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Tartamus (Tita) GV, Serban DE, Fogas CR, Tantau MV. Pediatric Inflammatory Bowel Disease in Romania: The First Epidemiological Study of the North-West Region (2000-2020). CHILDREN (BASEL, SWITZERLAND) 2025; 12:403. [PMID: 40310072 PMCID: PMC12025974 DOI: 10.3390/children12040403] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Received: 01/27/2025] [Revised: 03/11/2025] [Accepted: 03/20/2025] [Indexed: 05/02/2025]
Abstract
Background: Inflammatory bowel disease (IBD) represents a group of disorders with increasing incidence in the pediatric population worldwide. There are limited data on pediatric IBD (pIBD) epidemiology in Eastern Europe and none so far from Romania. The aim of our study was to evaluate incidence rates and time trends for pIBD in the north-west region of Romania and to compare them with results from studies on the same topic published worldwide. Methods: We performed a retrospective study of patients under 18 years of age diagnosed with pIBD in the time frame between 1 January 2000 and 31 December 2020 at the Emergency Clinical Hospital for Children in Cluj-Napoca. Age-adjusted incidence rates, annual percentage change (APC), average annual percentage chance (AAPC) and their corresponding 95% confidence intervals (CIs) were calculated for pIBD, Crohn's disease (CD) and ulcerative colitis (UC). Temporal trends were plotted using Joinpoint regression. Results: Ninety-four patients were identified. For the entire studied period, the incidence rate for pIBD was 0.79/105 (±0.74), 0.4/105 for CD (±0.42) and 0.34/105 for UC (±0.4). Time trends for incidence were positive, but statistical significance was found only for pIBD and CD. The APC observed for pIBD, CD and UC was 12 (95% CI: 6.5-17.7), 13.1 (95% CI: 8-18.5) and 5.67 (95% CI: 1.5-13.4), respectively. Comparison to other similar studies placed Romania among the countries with the lowest incidence of pIBD. Conclusions: Although pIBD incidence in our region appears to be low, there has been an important and significant increase in the incidence of overall pIBD and especially CD.
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Affiliation(s)
- Georgia Valentina Tartamus (Tita)
- 3rd Medical Discipline, Department of Internal Medicine, “Iuliu Hatieganu” University of Medicine and Pharmacy, 400012 Cluj-Napoca, Romania; (G.V.T.); (C.R.F.)
| | - Daniela Elena Serban
- Department of Mother and Child, 2nd Clinic of Pediatrics, Emergency Clinical Hospital for Children, “Iuliu Hatieganu” University of Medicine and Pharmacy, 400177 Cluj-Napoca, Romania
| | - Cristina Rebeca Fogas
- 3rd Medical Discipline, Department of Internal Medicine, “Iuliu Hatieganu” University of Medicine and Pharmacy, 400012 Cluj-Napoca, Romania; (G.V.T.); (C.R.F.)
| | - Marcel Vasile Tantau
- Department of Internal Medicine and Gastroenterology, “Prof. Dr. Octavian Fodor”, Regional Institute of Gastroenterology and Hepatology, “Iuliu Hatieganu” University of Medicine and Pharmacy, 400162 Cluj-Napoca, Romania;
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Xia Y, Sun Y, Du T, Sun C, Xu Y, Ge W, Liang L, Wang R, Sun M, Xiao B, Qiu W. Genetic Variants and Clinical Features of Patients With Glycogen Storage Disease Type Ib. JAMA Netw Open 2025; 8:e2461888. [PMID: 40009380 PMCID: PMC11866026 DOI: 10.1001/jamanetworkopen.2024.61888] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/24/2024] [Accepted: 12/22/2024] [Indexed: 02/27/2025] Open
Abstract
Importance Glycogen storage disease type Ib (GSDIb) is a rare metabolic disorder characterized by impaired glucose-6-phosphate transporter function with limited descriptions. Objective To describe the genetic and clinical features of Chinese patients with GSDIb. Design, Setting, and Participants This retrospective cohort study retrieved the medical records of 113 Chinese patients with GSDIb treated at a single institution in Shanghai from November 1, 2000, to June 30, 2024. Main Outcomes and Measures Biochemical parameters and clinical features (infections, inflammatory bowel disease [IBD], kidney and cardiovascular issues, growth and puberty) at baseline and the last follow-up, all-cause mortality, and mental and social development were assessed. Results A total of 113 patients (96 children [85%]; 67 males [59%]) had GSDIb genetically diagnosed at a median age of 1.4 (range, 0.0-35.5) years. Thirty-eight novel SLC37A4 variants were identified. Hypoglycemia and metabolic derangements were the primary concerns in patients aged 2 years or younger (45%). Short stature (75%), infections (75%), and recurrent epistaxis (31%) became more prevalent in patients aged 2.1 to 5.0 years. The frequency of respiratory tract infection decreased after a median age of 7.0 (IQR, 3.5-10.5) years. Forty-four patients (46%) developed IBD at a median age of 6.0 (IQR, 3.0-12.0) years, 14% of whom developed IBD-associated arthritis at a median age of 10.5 (IQR, 5.8-19.6) years. The mean (SD) difference between the z scores of actual height and target height (Δ height z scores) at the final visit (-2.11 [1.76]) did not significantly increase even after uncooked cornstarch treatment. The mean (SD) Δ height z scores of the patients with IBD (-2.99 [1.70]) were significantly lower than those without IBD (mean [SD], -1.36 [1.44]) (P < .001). Ten patients (9%) died due to complications of GSDIb, including metabolic derangements, sepsis, and/or severe pneumonia (n = 8), IBD (n = 1), and pulmonary hypertension (n = 1). Conclusions and Relevance In this cohort study, the genetic and clinical spectra of GSDIb were broadened, suggesting associations between GSDIb and bowel, growth, and survival outcomes. To date, no previous study reported IBD-associated arthritis.
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Affiliation(s)
- Yu Xia
- Department of Pediatric Endocrinology and Genetic Metabolism, Xinhua Hospital, Shanghai Institute of Pediatric Research, School of Medicine, Shanghai Jiao Tong University, Shanghai, China
| | - Yu Sun
- Department of Pediatric Endocrinology and Genetic Metabolism, Xinhua Hospital, Shanghai Institute of Pediatric Research, School of Medicine, Shanghai Jiao Tong University, Shanghai, China
- Department of Clinical Genetics, Xinhua Hospital, School of Medicine, Shanghai Jiao Tong University, Shanghai, China
| | - Taozi Du
- Department of Pediatric Endocrinology and Genetic Metabolism, Xinhua Hospital, Shanghai Institute of Pediatric Research, School of Medicine, Shanghai Jiao Tong University, Shanghai, China
| | - Chengkai Sun
- Department of Pediatric Endocrinology and Genetic Metabolism, Xinhua Hospital, Shanghai Institute of Pediatric Research, School of Medicine, Shanghai Jiao Tong University, Shanghai, China
| | - Ying Xu
- Department of Pediatric Internal Medicine, Ruijin Hospital, School of Medicine, Shanghai Jiao Tong University, Shanghai, China
| | - Wensong Ge
- Department of Gastroenterology, Xinhua Hospital, School of Medicine, Shanghai Jiao Tong University, Shanghai, China
| | - Lili Liang
- Department of Pediatric Endocrinology and Genetic Metabolism, Xinhua Hospital, Shanghai Institute of Pediatric Research, School of Medicine, Shanghai Jiao Tong University, Shanghai, China
| | - Ruifang Wang
- Department of Pediatric Endocrinology and Genetic Metabolism, Xinhua Hospital, Shanghai Institute of Pediatric Research, School of Medicine, Shanghai Jiao Tong University, Shanghai, China
| | - Manqing Sun
- Department of Pediatric Endocrinology and Genetic Metabolism, Xinhua Hospital, Shanghai Institute of Pediatric Research, School of Medicine, Shanghai Jiao Tong University, Shanghai, China
| | - Bing Xiao
- Department of Pediatric Endocrinology and Genetic Metabolism, Xinhua Hospital, Shanghai Institute of Pediatric Research, School of Medicine, Shanghai Jiao Tong University, Shanghai, China
- Department of Clinical Genetics, Xinhua Hospital, School of Medicine, Shanghai Jiao Tong University, Shanghai, China
| | - Wenjuan Qiu
- Department of Pediatric Endocrinology and Genetic Metabolism, Xinhua Hospital, Shanghai Institute of Pediatric Research, School of Medicine, Shanghai Jiao Tong University, Shanghai, China
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Chen X, Xiang X, Fan X, Xia W, Xiao Y, Wang S, Ye S, Kang M, Jing F, Wu X, Chen Y. Global Burden of Inflammatory Bowel Disease Among Children and Adolescents: A Comprehensive Analysis (1990-2019). Int J Public Health 2024; 69:1607440. [PMID: 39314257 PMCID: PMC11417169 DOI: 10.3389/ijph.2024.1607440] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/28/2024] [Accepted: 08/27/2024] [Indexed: 09/25/2024] Open
Abstract
Objective We summarize the global, regional, and national burden of inflammatory bowel disease (IBD) in children and adolescents from 1990 to 2019. Methods Based on the Global Burden of Disease Study 2019, the data of IBD in children and adolescents were analyzed by sex, age, year, and location. Joinpoint analysis was applied to assess the temporal trend of the disease burden. Results From 1990 to 2019, the incidence of IBD in children and adolescents increased by 22.8%, from 20,897.42 to 25,658.55 cases, especially in high SDI region. During the same period, the DALY numbers decreased by 53.5%, from 243,081.06 to 113,119.86, with all SDI regions experiencing a clear drop in DALYs except high SDI regions. In 2019, early-onset IBD incidence and DALY numbers were reported at 2,053.52 (95% UI: 1,575.62 to 2,677.49) and 73,797.46 (95% UI: 43,655.86 to 105,998.63), respectively. Conclusion Early-onset IBD in children and adolescents remains a significant global health concern. The disease burden has not improved in developed countries over the past 30 years, highlighting the need for targeted interventions.
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Affiliation(s)
- Xuejie Chen
- Department of Gastroenterology, Third Xiangya Hospital of Central South University, Changsha, Hunan, China
| | - Xin Xiang
- Department of Gastroenterology, Third Xiangya Hospital of Central South University, Changsha, Hunan, China
| | - Xiaofei Fan
- Department of Clinical Medicine, Shandong Medical College, Jinan, Shandong, China
| | - Weitong Xia
- Department of Gastroenterology, Third Xiangya Hospital of Central South University, Changsha, Hunan, China
| | - Yi Xiao
- Department of Gastroenterology, Third Xiangya Hospital of Central South University, Changsha, Hunan, China
| | - Sidan Wang
- Department of Gastroenterology, Third Xiangya Hospital of Central South University, Changsha, Hunan, China
| | - Shuyu Ye
- Department of Gastroenterology, Third Xiangya Hospital of Central South University, Changsha, Hunan, China
| | - Meng Kang
- Department of Gastroenterology, Third Xiangya Hospital of Central South University, Changsha, Hunan, China
| | - Fangmin Jing
- Department of Gastroenterology, Third Xiangya Hospital of Central South University, Changsha, Hunan, China
| | - Xing Wu
- Department of Gastroenterology, Third Xiangya Hospital of Central South University, Changsha, Hunan, China
| | - Yang Chen
- Department of Gastroenterology, Third Xiangya Hospital of Central South University, Changsha, Hunan, China
- Nursing Department, Third Xiangya Hospital of Central South University, Changsha, Hunan, China
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Tóbi L, Prehoda B, Balogh AM, Nagypál P, Kovács K, Miheller P, Iliás Á, Dezsőfi-Gottl A, Cseh Á. Transition is associated with lower disease activity, fewer relapses, better medication adherence, and lower lost-to-follow-up rate as opposed to self-transfer in pediatric-onset inflammatory bowel disease patients: results of a longitudinal, follow-up, controlled study. Therap Adv Gastroenterol 2024; 17:17562848241252947. [PMID: 39156978 PMCID: PMC11327998 DOI: 10.1177/17562848241252947] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/13/2023] [Accepted: 04/15/2024] [Indexed: 08/20/2024] Open
Abstract
BACKGROUND Despite the continuously rising rate of pediatric-onset inflammatory bowel diseases (PIBD), there are no consensus transitional guidelines or standardized practices. OBJECTIVES We aimed to examine: (1) the determinants of a successful transfer, (2) the effects of the transfer versus transition on the disease course and patient compliance, (3) the unique characteristics of PIBD patients, that need special attention in adult care. DESIGN Longitudinal, follow-up, controlled study conducted between 2001 and 2022, with retrospective data collection until 2018, thence prospective. METHODS Three hundred fifty-one PIBD patients enrolled in the study, of whom 152 were moved to adult care, with a mean post-transfer follow-up time of 3 years. Seventy-three patients took part in structured transition, whereas 79 self-transferred to adult care. The main outcome measures were disease activity (defined by PCDAI, PUCAI, CDAI, and Mayo-scores) and course, hospitalizations, surgeries, IBD-related complications, including anthropometry and bone density, patient compliance, medication adherence, and continuation of medical care. RESULTS Patients who underwent structured transition spent significantly more time in remission (83.6% ± 28.5% versus 77.5% ± 29.7%, p = 0.0339) and had better adherence to their medications (31.9% versus 16.4% non-adherence rate, p = 0.0455) in adult care, with self-transferred patients having a 1.59-fold increased risk of discontinuing their medical care and a 1.88-fold increased risk of experiencing a relapse. Post-transfer the compliance of patients deteriorated (38.5% versus 29%, p = 0.0002), with the highest lost-to-follow-up rate during the changing period between the healthcare systems (12.7%), in which female gender was a risk factor (p = 0.010). PIBD patients had experienced IBD-related complications (23.4%) and former surgeries (15%) upon arriving at adult care, with high rates of malnutrition, growth impairment, and poor bone health. CONCLUSION Structured transition plays a key role in ensuring the best disease course and lowering the lost-to-follow-up rate among PIBD patients. BRIEF SUMMARY Structured transition plays a key role in ensuring the best disease outcome among PIBD patients, as in our study it was associated with lower disease activity, fewer relapses, better medication adherence, and lower lost-to-follow-up rate as opposed to self-transfer.
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Affiliation(s)
- Luca Tóbi
- Pediatric Center, MTA Center of Excellence, Semmelweis University, Post Office Box 2, Budapest 1428, Hungary
| | - Bence Prehoda
- Pediatric Center, MTA Center of Excellence, Semmelweis University, Budapest, Hungary
| | - Anna M. Balogh
- Pediatric Center, MTA Center of Excellence, Semmelweis University, Budapest, Hungary
| | - Petra Nagypál
- Pediatric Center, MTA Center of Excellence, Semmelweis University, Budapest, Hungary
| | - Krisztián Kovács
- Department of Laboratory Medicine, Semmelweis University, Budapest, Hungary
| | - Pál Miheller
- Department of Surgery, Transplantation, and Gastroenterology, Semmelweis University, Budapest, Hungary
| | - Ákos Iliás
- Department of Internal Medicine and Oncology, Semmelweis University, Budapest, Hungary
| | - Antal Dezsőfi-Gottl
- Pediatric Center, MTA Center of Excellence, Semmelweis University, Budapest, Hungary
| | - Áron Cseh
- Pediatric Center, MTA Center of Excellence, Semmelweis University, Budapest, Hungary
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Ochoa B, McMahon L. Surgery for ulcerative colitis. Semin Pediatr Surg 2024; 33:151404. [PMID: 38615424 DOI: 10.1016/j.sempedsurg.2024.151404] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 04/16/2024]
Abstract
Ulcerative colitis (UC) has a more severe presentation and rapid progression in pediatric patients, resulting in a greater need for surgical intervention compared to adults. Though medical management of UC has advanced with new biologic therapies, surgery continues to play an important role when disease progresses in the form of worsened or persistent symptoms, hemodynamic instability, or sepsis. The goals of surgical management are to restore intestinal continuity with a functional pouch when possible. While the literature has been growing regarding studies of pediatric patients with UC, high level of evidence studies are limited and most recommendations are based on adult studies. Similar to adults, pediatric patients who have ileal pouches created require surveillance for recurrent disease and cancer surveillance. Unique issues for pediatric patients include monitoring of growth and appropriate transition to adult care after adolescence. This review includes indications for surgical management, overview of staged surgical approaches, and the technical details of the three-stage approach.
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Affiliation(s)
- Brielle Ochoa
- Division of Pediatric Surgery, Department of Surgery, Phoenix Children's, Phoenix, Arizona, USA
| | - Lisa McMahon
- Division of Pediatric Surgery, Department of Surgery, Phoenix Children's, Phoenix, Arizona, USA.
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Weidner J, Glauche I, Manuwald U, Kern I, Reinecke I, Bathelt F, Amin M, Dong F, Rothe U, Kugler J. Correlation of Socioeconomic and Environmental Factors With Incidence of Crohn Disease in Children and Adolescents: Systematic Review and Meta-Regression. JMIR Public Health Surveill 2024; 10:e48682. [PMID: 38526534 PMCID: PMC11002755 DOI: 10.2196/48682] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/03/2023] [Revised: 12/28/2023] [Accepted: 01/23/2024] [Indexed: 03/26/2024] Open
Abstract
BACKGROUND The worldwide incidence of Crohn disease (CD) in childhood and adolescence has an increasing trend, with significant differences between different geographic regions and individual countries. This includes an increase in the incidence of CD in countries and geographic regions where CD was not previously prevalent. In response to the increasing incidence, the pediatric care landscape is facing growing challenges. OBJECTIVE This systematic review and meta-analysis were undertaken to comprehensively delineate the incidence rates of CD in pediatric populations across different countries and to explore potential influencing factors. METHODS We performed a systematic review of PubMed and Embase (via Ovid) for studies from January 1, 1970, to December 31, 2019. In addition, a manual search was performed in relevant and previously published reviews. The results were evaluated quantitatively. For this purpose, random effects meta-analyses and meta-regressions were performed to investigate the overall incidence rate and possible factors influencing the incidence. RESULTS A qualitative synthesis of 74 studies was performed, with 72 studies included in the meta-analyses and 52 in the meta-regressions. The results of our meta-analysis showed significant heterogeneity between the individual studies, which cannot be explained by a sample effect alone. Our findings showed geographical differences in incidence rates, which increased with increasing distance from the equator, although no global temporal trend was apparent. The meta-regression analysis also identified geographic location, UV index, and Human Development Index as significant moderators associated with CD incidence. CONCLUSIONS Our results suggest that pediatric CD incidence has increased in many countries since 1970 but varies widely with geographic location, which may pose challenges to the respective health care systems. We identified geographic, environmental, and socioeconomic factors that contribute to the observed heterogeneity in incidence rates. These results can serve as a basis for future research. To this end, implementations of internationally standardized and interoperable registries combined with the dissemination of health data through federated networks based on a common data model, such as the Observational Medical Outcomes Partnership, would be beneficial. This would deepen the understanding of CD and promote evidence-based approaches to preventive and interventional strategies as well as inform public health policies aimed at addressing the increasing burden of CD in children and adolescents. TRIAL REGISTRATION PROSPERO International prospective register of systematic reviews CRD42020168644; https://www.crd.york.ac.uk/PROSPERO/display_record.php?RecordID=168644. INTERNATIONAL REGISTERED REPORT IDENTIFIER (IRRID) RR2-10.1136/bmjopen-2020-037669.
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Affiliation(s)
- Jens Weidner
- Institute for Medical Informatics and Biometry, Medical Faculty Carl Gustav Carus, TU Dresden, Dresden, Germany
| | - Ingmar Glauche
- Institute for Medical Informatics and Biometry, Medical Faculty Carl Gustav Carus, TU Dresden, Dresden, Germany
| | - Ulf Manuwald
- Faculty of Applied Social Sciences, University of Applied Sciences (FHD), Dresden, Germany
| | - Ivana Kern
- Institute and Policlinic for Occupational and Social Medicine, Department of Health Sciences/Public Health, Medical Faculty Carl Gustav Carus, TU Dresden, Dresden, Germany
| | - Ines Reinecke
- Institute for Medical Informatics and Biometry, Medical Faculty Carl Gustav Carus, TU Dresden, Dresden, Germany
| | - Franziska Bathelt
- Institute for Medical Informatics and Biometry, Medical Faculty Carl Gustav Carus, TU Dresden, Dresden, Germany
- Thiem-Research GmbH, Cottbus, Germany
| | - Makan Amin
- Institute and Policlinic for Occupational and Social Medicine, Department of Health Sciences/Public Health, Medical Faculty Carl Gustav Carus, TU Dresden, Dresden, Germany
- Department for Trauma Surgery and Orthopaedics, Park-Klinik Weissensee, Berlin, Germany
| | - Fan Dong
- Institute and Policlinic for Occupational and Social Medicine, Department of Health Sciences/Public Health, Medical Faculty Carl Gustav Carus, TU Dresden, Dresden, Germany
| | | | - Joachim Kugler
- Institute and Policlinic for Occupational and Social Medicine, Department of Health Sciences/Public Health, Medical Faculty Carl Gustav Carus, TU Dresden, Dresden, Germany
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Salguero MV, Deplewski D, Gokhale R, Wroblewski K, Sentongo T, Jan A, Kirschner BS. Growth After Menarche in Pediatric Inflammatory Bowel Disease. J Pediatr Gastroenterol Nutr 2023; 76:183-190. [PMID: 36705699 PMCID: PMC9889107 DOI: 10.1097/mpg.0000000000003667] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 01/28/2023]
Abstract
OBJECTIVES Growth impairment in pediatric patients with pediatric onset inflammatory bowel disease (IBD) is multifactorial. Reports on the effect of age at menarche on adult stature in this population are limited. This study investigated the impact of age at menarche, disease-associated factors, and mid-parental height on growth from menarche to final height (FHt) in pediatric patients with Crohn disease (CD) and ulcerative colitis (UC) and IBD unclassified (IBD-U). METHODS Subjects were enrolled from a prospectively maintained pediatric IBD database when IBD preceded menarche and dates of menarche and FHt measurements were recorded. RESULTS One hundred forty-six patients: CD 112 and UC 30/IBD-U 4. Mean age (years) at diagnosis (10.9 vs 10.1), menarche (14.4 vs 14.0), and FHt (19.6 vs 19.7). CD and UC/IBD-U patients showed significant association between Chronological age (CA) at menarche and FHt (cm, P < 0.001) but not FHt z score (FHt-Z) < -1.0 (P = 0.42). FHt-Z < -2.0 occurred in only 5 patients. Growth impairment (FHt-Z < -1.0) was associated with surgery before menarche (P = 0.03), jejunal disease (P = 0.003), low mid-parental height z score (MPH-Z) (P < 0.001), hospitalization for CD (P = 0.03) but not UC, recurrent corticosteroid, or anti-tumor necrosis factor alpha (anti-TNFα) therapy. CONCLUSIONS Early age of menarche was associated with greater potential for linear growth to FHt but not FHt-Z (P < 0.05). Surgery before menarche, jejunal disease, hospitalization for CD, low MPH, and weight z score were associated with FHt-Z < -1.0.
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Affiliation(s)
- Maria V. Salguero
- Section of Adult and Pediatric Endocrinology, University of Chicago, Chicago, IL 60637, USA
| | - Dianne Deplewski
- Section of Adult and Pediatric Endocrinology, University of Chicago, Chicago, IL 60637, USA
| | - Ranjana Gokhale
- Section of Pediatric Gastroenterology, Hepatology and Nutrition, University of Chicago Comer Children’s Hospital, Chicago, IL 60637, USA
| | - Kristen Wroblewski
- Department of Public Health Sciences, University of Chicago, Chicago, IL 60637, USA
| | - Timothy Sentongo
- Section of Pediatric Gastroenterology, Hepatology and Nutrition, University of Chicago Comer Children’s Hospital, Chicago, IL 60637, USA
| | - Aseel Jan
- Section of Pediatric Gastroenterology, Hepatology and Nutrition, University of Chicago Comer Children’s Hospital, Chicago, IL 60637, USA
| | - Barbara S. Kirschner
- Section of Pediatric Gastroenterology, Hepatology and Nutrition, University of Chicago Comer Children’s Hospital, Chicago, IL 60637, USA
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Kern I, Schoffer O, Richter T, Kiess W, Flemming G, Winkler U, Quietzsch J, Wenzel O, Zurek M, Manuwald U, Hegewald J, Li S, Weidner J, de Laffolie J, Zimmer KP, Kugler J, Laass MW, Rothe U. Current and projected incidence trends of pediatric-onset inflammatory bowel disease in Germany based on the Saxon Pediatric IBD Registry 2000–2014 –a 15-year evaluation of trends. PLoS One 2022; 17:e0274117. [PMID: 36084003 PMCID: PMC9462751 DOI: 10.1371/journal.pone.0274117] [Citation(s) in RCA: 9] [Impact Index Per Article: 3.0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/05/2022] [Accepted: 08/23/2022] [Indexed: 11/19/2022] Open
Abstract
Aims
An increasing number of children and adolescents worldwide suffer from inflammatory bowel disease (IBD) such as Crohn’s disease (CD) and ulcerative colitis (UC). The present work aims to investigate the incidence, prevalence and future trends of IBD in children and adolescents in Saxony, Germany.
Methods
The Saxon Pediatric IBD Registry collected data on patients up to 15 years of age from all 31 pediatric hospitals and pediatric gastroenterologists in Saxony over a 15-year period (2000–2014). In 2019, an independent survey estimated a registry completeness of 95.7%. Age-standardized incidence rates (ASR) per 100,000 person-years (PY) and prevalence per 100,000 children and adolescents were calculated. Evaluation was also been performed in sex and age subgroups. Joinpoint and Poisson regression were used for trend analyses and projections.
Results
532 patients with confirmed IBD during 2000–2014 were included in the epidemiological evaluation. 63.5% (n = 338) patients had CD, 33.1% (n = 176) had UC and 3.4% (n = 18) had unclassified IBD (IBD-U). The 15-year IBD prevalence was 111.8 [95%-CI: 102.3–121.3] per 100,000. The incidence ASR of IBD per 100,000 PY over the whole observation period was 7.5 [6.9–8.1]. ASR for the subtypes were 4.8 [4.3–5.3] for CD, 2.5 [2.1–2.9] for UC and 0.3 [0.1–0.4] for IBD-U. The trend analysis of ASR using the joinpoint regression confirmed a significant increase for incidence of IBD as well as CD. For IBD, the ASR per 100,000 PY increased from 4.6 [2.8–6.3] in 2000 to 8.2 [7.5–13.6] in 2014; projected incidence rates for IBD in Germany are 12.9 [6.5–25.5] in the year 2025 and 14.9 [6.7–32.8] in 2030, respectively. Thus, the number of new IBD diagnoses in Germany would more than triple (325%) in 2030 compared to 2000. The increase is expected to be faster in CD than UC, and be more in males than in females. The expected number of newly diagnosed children with IBD in Germany is projected to rise to about 1,584 [1,512–1,655] in 2025, and to about 1,918 [1,807–2,29] in 2030.
Conclusion
The incidence of IBD in children and adolescents in Saxony increased at a similar rate as in other developed countries during the observation period. Given this trend, the health care system must provide adequate resources for the care of these young patients in the future.
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Affiliation(s)
- Ivana Kern
- Department of Health Sciences/Public Health, Institute and Policlinic for Occupational and Social Medicine, Faculty of Medicine „Carl Gustav Carus”, TU Dresden, Dresden, Germany
- * E-mail:
| | - Olaf Schoffer
- Center for Evidence-based Healthcare, University Hospital and Faculty of Medicine “Carl Gustav Carus”, TU Dresden, Dresden, Germany
| | - Thomas Richter
- Clinic for Children and Adolescents, Hospital St. Georg, Leipzig, Germany
| | - Wieland Kiess
- Center for Pediatric Research, Department of Women and Child Health, Hospital for Children and Adolescents, University of Leipzig, Leipzig, Germany
| | - Gunter Flemming
- Center for Pediatric Research, Department of Women and Child Health, Hospital for Children and Adolescents, University of Leipzig, Leipzig, Germany
| | - Ulf Winkler
- Clinic for Children and Adolescents, Hospital Bautzen, Oberlausitz-Hospitals, Bautzen, Germany
| | - Jürgen Quietzsch
- Clinic for Children and Adolescents, DRK Hospital Lichtenstein, Lichtenstein, Germany
| | - Olaf Wenzel
- Clinic for Children and Adolescents, Helios Hospital Aue, Bad Schlema, Germany
| | - Marlen Zurek
- Clinic for Children and Adolescents, Hospital St. Georg, Leipzig, Germany
| | - Ulf Manuwald
- Department of Health Sciences/Public Health, Institute and Policlinic for Occupational and Social Medicine, Faculty of Medicine „Carl Gustav Carus”, TU Dresden, Dresden, Germany
| | - Janice Hegewald
- Department of Occupational, Social and Environmental Epidemiology, Institute and Policlinic for Occupational and Social Medicine, Faculty of Medicine „Carl Gustav Carus”, TU Dresden, Dresden, Germany
| | - Shi Li
- Department of Health Sciences/Public Health, Institute and Policlinic for Occupational and Social Medicine, Faculty of Medicine „Carl Gustav Carus”, TU Dresden, Dresden, Germany
| | - Jens Weidner
- Center for Medical Informatics, Institute for Medical Informatics and Biometry, Faculty of Medicine “Carl Gustav Carus”, TU Dresden, Dresden, Germany
| | - Jan de Laffolie
- Department of General Pediatrics, Children’s Gastroenterology/ Hepatology/ Nutrition, Justus-Liebig-University Gießen, CEDATA-GPGE Working Group, Gießen, Germany
| | - Klaus-Peter Zimmer
- Department of General Pediatrics, Children’s Gastroenterology/ Hepatology/ Nutrition, Justus-Liebig-University Gießen, CEDATA-GPGE Working Group, Gießen, Germany
| | - Joachim Kugler
- Department of Health Sciences/Public Health, Institute and Policlinic for Occupational and Social Medicine, Faculty of Medicine „Carl Gustav Carus”, TU Dresden, Dresden, Germany
| | - Martin W. Laass
- University Hospital for Children and Adolescents, Faculty of Medicine “Carl Gustav Carus”, TU Dresden, Dresden, Germany
| | - Ulrike Rothe
- Department of Health Sciences/Public Health, Institute and Policlinic for Occupational and Social Medicine, Faculty of Medicine „Carl Gustav Carus”, TU Dresden, Dresden, Germany
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9
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Khan MR, Ulrich JA, Hull NC, Inoue A, Harmsen WS, Faubion WA, Fletcher JG, Absah I. Perianal magnetic resonance imaging findings and their potential impact on outcome in children with perianal fistulizing Crohn disease. Pediatr Radiol 2021; 51:2481-2491. [PMID: 34490496 DOI: 10.1007/s00247-021-05158-w] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/30/2020] [Revised: 05/25/2021] [Accepted: 07/20/2021] [Indexed: 11/24/2022]
Abstract
BACKGROUND Children with perianal fistulizing Crohn disease require intensive medical management but also have a higher risk for subsequent surgical interventions. OBJECTIVE We performed a retrospective study to identify patient factors and perianal anatomical features by pelvic MR that are associated with surgical interventions in these children. MATERIALS AND METHODS We included children with Crohn disease and perianal fistula who underwent pelvic MR with available, archived images and collected demographic, clinical and laboratory data. Radiologists reviewed pelvic MR exams and identified Park classification and additional anatomical features of perianal fistulas, including fistula branching, horseshoe ramifications, abscess, inflammatory mass, supralevator extension, anal sphincter damage, proctitis and posterior anal space involvement. We performed univariate and subsequent multivariate analysis to determine features associated with subsequent surgical intervention. RESULTS Ninety-nine children with Crohn disease underwent pelvic MR. In this cohort, 69 children had no surgical interventions prior to baseline MRI, with subsequent median clinical follow-up of 5.5 years. Univariate analysis demonstrated that branching (P=0.009), supralevator extension (P=0.015) and anal sphincter damage (P=0.031) were significantly associated with subsequent surgical intervention. Age at baseline MRI was also associated with intervention (hazard ratio [HR] every 5 years: 2.13; 95% confidence interval [CI]: 1.18-3.83; P=0.012). A multivariable model identified only fistula branching (HR: 2.31; 95% CI: 1.28-4.15; P=0.005) and age (HR: 5.18; CI: 1.57-17.14; P=0.007) as independent predictors of subsequent surgery. No demographic, clinical or laboratory parameter predicted subsequent surgical intervention. CONCLUSION Age and anatomical MR features indicating fistula complexity (branching, supralevator extension) and sphincter damage confer a higher risk of subsequent surgical intervention in children with perianal Crohn disease.
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Affiliation(s)
- Muhammad Rehan Khan
- Division of Pediatric Gastroenterology, Hepatology and Nutrition, University of Illinois College of Medicine at Peoria, Children's Hospital of Illinois, 530 NE Glen Oak Ave., Peoria, IL, 61637, USA. .,Department of Pediatrics & Child Health, Aga Khan University, Karachi, Pakistan.
| | | | - Nathan C Hull
- Department of Radiology, Mayo Clinic, Rochester, MN, USA
| | - Akitoshi Inoue
- Department of Radiology, Mayo Clinic, Rochester, MN, USA
| | - William S Harmsen
- Department of Biomedical Statistics and informatics, Mayo Clinic, Rochester, MN, USA
| | - William A Faubion
- Division of Gastroenterology and Hepatology, Mayo Clinic, Rochester, MN, USA
| | | | - Imad Absah
- Division of Gastroenterology and Hepatology, Mayo Clinic, Rochester, MN, USA.,Division of Pediatric Gastroenterology and Hepatology, Mayo Clinic, Rochester, MN, USA
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10
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Yao J, Jiang X, You JHS. Proactive therapeutic drug monitoring of adalimumab for pediatric Crohn's disease patients: A cost-effectiveness analysis. J Gastroenterol Hepatol 2021; 36:2397-2407. [PMID: 33326123 DOI: 10.1111/jgh.15373] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/07/2020] [Revised: 12/04/2020] [Accepted: 12/10/2020] [Indexed: 01/15/2023]
Abstract
BACKGROUND AND AIM Recent clinical findings showed proactive therapeutic drug monitoring (TDM) of adalimumab (ADL) to improve sustained remission rate in pediatric patients with Crohn's disease (CD). The present study aimed to evaluate the potential cost-effectiveness of proactive versus reactive TDM of ADL in pediatric patients with CD from the perspective of the US health-care provider. METHODS A Markov model was constructed to estimate outcomes of proactive versus reactive TDM of ADL in a hypothetical cohort of pediatric CD patients who were in remission on ADL maintenance treatment. Model inputs were derived from published literature and public data. Model outcomes included CD-related direct medical cost and quality-adjusted life-years (QALYs). Sensitivity analyses were performed to examine the robustness of base-case results. RESULTS When compared with the reactive TDM group, the proactive TDM group saved 0.1960 QALYs at lower cost by USD2021 over a 3-year time frame in base-case analysis. One-way sensitivity analysis showed the ADL drug cost to be the most influential factor. Probabilistic sensitivity analysis of 10 000 Monte-Carlo simulations found the proactive TDM group to gain 0.1958 QALYs (95% confidence interval [CI] 0.1950-0.1966; P < 0.001) and save USD2037 (95%CI USD1943-2131; P < 0.001). CONCLUSIONS Proactive TDM for ADL seems to gain higher QALYs at lower cost in pediatric CD patients.
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Affiliation(s)
- Jiaqi Yao
- School of Pharmacy, Faculty of Medicine, The Chinese University of Hong Kong, Hong Kong, China
| | - Xinchan Jiang
- School of Pharmacy, Faculty of Medicine, The Chinese University of Hong Kong, Hong Kong, China
| | - Joyce H S You
- School of Pharmacy, Faculty of Medicine, The Chinese University of Hong Kong, Hong Kong, China
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11
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Telemedicine and Integrated Multidisciplinary Care for Pediatric IBD Patients: A Review. CHILDREN-BASEL 2021; 8:children8050347. [PMID: 33924754 PMCID: PMC8147085 DOI: 10.3390/children8050347] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Download PDF] [Subscribe] [Scholar Register] [Received: 03/15/2021] [Revised: 04/23/2021] [Accepted: 04/27/2021] [Indexed: 11/16/2022]
Abstract
The global COVID-19 pandemic brought with it an unprecedented, widespread implementation of telemedicine services, requiring pediatric inflammatory bowel disease (IBD) providers to shift in-person clinic visits to a virtual platform. With the passing of the one-year anniversary of the global pandemic, telemedicine continues to be offered and utilized. Although it remains unclear as to the extent to which telemedicine services will be used in the future, it is critical to understand how integrated multidisciplinary treatment-the standard of care in pediatric IBD-is delivered through a virtual platform. This paper provides an overview of the existing literature examining integrated multidisciplinary care for pediatric IBD provided via telemedicine. The author also presents one integrated multidisciplinary IBD program's response to the global pandemic and subsequent transition to telemedicine. Challenges around implementation and directions for future research in this area are also discussed.
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12
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Rana T, Korolkova OY, Rachakonda G, Williams AD, Hawkins AT, James SD, Sakwe AM, Hui N, Wang L, Yu C, Goodwin JS, Izban MG, Offodile RS, Washington MK, Ballard BR, Smoot DT, Shi XZ, Forbes DS, Shanker A, M’Koma AE. Linking bacterial enterotoxins and alpha defensin 5 expansion in the Crohn's colitis: A new insight into the etiopathogenetic and differentiation triggers driving colonic inflammatory bowel disease. PLoS One 2021; 16:e0246393. [PMID: 33690604 PMCID: PMC7942995 DOI: 10.1371/journal.pone.0246393] [Citation(s) in RCA: 4] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/14/2020] [Accepted: 01/17/2021] [Indexed: 02/05/2023] Open
Abstract
Evidence link bacterial enterotoxins to apparent crypt-cell like cells (CCLCs), and Alpha Defensin 5 (DEFA5) expansion in the colonic mucosa of Crohn's colitis disease (CC) patients. These areas of ectopic ileal metaplasia, positive for Paneth cell (PC) markers are consistent with diagnosis of CC. Retrospectively, we: 1. Identified 21 patients with indeterminate colitis (IC) between 2000-2007 and were reevaluation their final clinical diagnosis in 2014 after a followed-up for mean 8.7±3.7 (range, 4-14) years. Their initial biopsies were analyzed by DEFA5 bioassay. 2. Differentiated ulcer-associated cell lineage (UACL) analysis by immunohistochemistry (IHC) of the CC patients, stained for Mucin 6 (MUC6) and DEFA5. 3. Treated human immortalized colonic epithelial cells (NCM460) and colonoids with pure DEFA5 on the secretion of signatures after 24hr. The control colonoids were not treated. 4. Treated colonoids with/without enterotoxins for 14 days and the spent medium were collected and determined by quantitative expression of DEFA5, CCLCs and other biologic signatures. The experiments were repeated twice. Three statistical methods were used: (i) Univariate analysis; (ii) LASSO; and (iii) Elastic net. DEFA5 bioassay discriminated CC and ulcerative colitis (UC) in a cohort of IC patients with accuracy. A fit logistic model with group CC and UC as the outcome and the DEFA5 as independent variable differentiator with a positive predictive value of 96 percent. IHC staining of CC for MUC6 and DEFA5 stained in different locations indicating that DEFA5 is not co-expressed in UACL and is therefore NOT the genesis of CC, rather a secretagogue for specific signature(s) that underlie the distinct crypt pathobiology of CC. Notably, we observed expansion of signatures after DEFA5 treatment on NCM460 and colonoids cells expressed at different times, intervals, and intensity. These factors are key stem cell niche regulators leading to DEFA5 secreting CCLCs differentiation 'the colonic ectopy ileal metaplasia formation' conspicuously of pathogenic importance in CC.
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Affiliation(s)
- Tanu Rana
- Department of Biochemistry, Cancer Biology, Neuroscience and Pharmacology, Meharry Medical College School of Medicine, Nashville, Tennessee, United States of America
| | - Olga Y. Korolkova
- Department of Biochemistry, Cancer Biology, Neuroscience and Pharmacology, Meharry Medical College School of Medicine, Nashville, Tennessee, United States of America
| | - Girish Rachakonda
- Department of Microbiology and Immunology, Meharry Medical College School of Medicine, Nashville, Tennessee, United States of America
| | - Amanda D. Williams
- Department of Biology, Lipscomb University, Nashville, Tennessee, United States of America
| | - Alexander T. Hawkins
- Division of General Surgery, Section of Colon and Rectal Surgery, Vanderbilt University School of Medicine, Nashville, Tennessee, United States of America
| | - Samuel D. James
- Department of Pathology, Anatomy and Cell Biology, Meharry Medical College School of Medicine, Nashville General Hospital, Nashville, Tennessee, United States of America
- Department of Pathology, Microbiology, and Immunology, Tennessee Valley Health Systems VA Medical Center, Vanderbilt University School of Medicine, Nashville, Tennessee, United States of America
| | - Amos M. Sakwe
- Department of Biochemistry, Cancer Biology, Neuroscience and Pharmacology, Meharry Medical College School of Graduate Studies and Research, Nashville, Tennessee, United States of America
| | - Nian Hui
- Department of Biostatistics, Vanderbilt University School of Medicine, Nashville, Tennessee, United States of America
| | - Li Wang
- Department of Biostatistics, Vanderbilt University School of Medicine, Nashville, Tennessee, United States of America
| | - Chang Yu
- Department of Biostatistics, Vanderbilt University School of Medicine, Nashville, Tennessee, United States of America
| | - Jeffrey S. Goodwin
- Department of Biochemistry, Cancer Biology, Neuroscience and Pharmacology, Meharry Medical College School of Medicine, Nashville, Tennessee, United States of America
| | - Michael G. Izban
- Department of Pathology, Anatomy and Cell Biology, Meharry Medical College School of Medicine, Nashville General Hospital, Nashville, Tennessee, United States of America
| | - Regina S. Offodile
- Department of Professional and Medical Education, Meharry Medical College School of Medicine, Nashville General Hospital, Nashville, Tennessee, United States of America
| | - Mary K. Washington
- Department of Pathology, Microbiology and Immunology, Vanderbilt University School of Medicine, Nashville, Tennessee, United States of America
| | - Billy R. Ballard
- Department of Pathology, Anatomy and Cell Biology, Meharry Medical College School of Medicine, Nashville General Hospital, Nashville, Tennessee, United States of America
| | - Duane T. Smoot
- Department of Medicine, Meharry Medical College School of Medicine, Nashville General Hospital, Nashville, Tennessee, United States of America
| | - Xuan-Zheng Shi
- Department of Medicine, University of Texas Medical Branch (UTMB) in Galveston, Galveston, Texas, United States of America
| | - Digna S. Forbes
- Department of Pathology, Anatomy and Cell Biology, Meharry Medical College School of Medicine, Nashville General Hospital, Nashville, Tennessee, United States of America
| | - Anil Shanker
- Department of Biochemistry, Cancer Biology, Neuroscience and Pharmacology, Meharry Medical College School of Medicine, Nashville, Tennessee, United States of America
| | - Amosy E. M’Koma
- Department of Biochemistry, Cancer Biology, Neuroscience and Pharmacology, Meharry Medical College School of Medicine, Nashville, Tennessee, United States of America
- Division of General Surgery, Section of Colon and Rectal Surgery, Vanderbilt University School of Medicine, Nashville, Tennessee, United States of America
- Department of Pathology, Anatomy and Cell Biology, Meharry Medical College School of Medicine, Nashville General Hospital, Nashville, Tennessee, United States of America
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13
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Aloi M, Bramuzzo M, Norsa L, Arrigo S, Distante M, Miele E, Romano C, Giobbi C, Panceri R, Cucchiara S, Alvisi P. Disease Activity Patterns in the First 5 Years After Diagnosis in Children With Ulcerative Colitis: A Population-Based Study. J Crohns Colitis 2021; 15:367-374. [PMID: 33022055 DOI: 10.1093/ecco-jcc/jjaa203] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/08/2023]
Abstract
BACKGROUND The aim of this study was to define clusters of activity in a population-based cohort during the first 5 years after diagnosis in children with ulcerative colitis [UC] and to identify early prognostic risk factors. METHODS All UC patients from the SIGENP IBD registry with a complete follow-up of at least 5 years were included. Active disease was defined every 6 months in the presence of at least one of the following: clinical activity [Paediatric Ulcerative Colitis Activity Index ≥ 35]; endoscopic activity [Mayo score ≥ 1]; faecal calprotectin > 250 µg/g; hospitalization; surgery; or treatment escalation. Formula-based clusters were generated based on four published questionnaire-based activity patterns in adults, plus one additional cluster. RESULTS In total, 226 patients were identified. Forty-two [19%] had moderate-severe chronically active disease, 31 [14%] chronic-intermittent, 75 [33%] quiescent, 54 [24%] active disease in the first 2 years after the diagnosis, then sustained remission, and 24 [11%] a remission in the first 2 years then an active disease. Mild disease onset along with a lower clinical severity not requiring the use of corticosteroids at 6 months were related to a quiescent disease course at the next follow-up (logistic model area under the curve 0.86 [95% confidence interval 0.78-0.94]; positive predictive value 67%; negative predictive value 70%). Eight per cent of patients needed surgery, none in the quiescent group [p = 0.04]. CONCLUSIONS More than one-third of children with UC present with a chronically active or intermittent course during the first 5 years of follow-up. A significant group of patients has active disease in the first 2 years and then sustained remission. Interestingly, after initial treatment, one-third of patients have well-controlled disease throughout.
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Affiliation(s)
- M Aloi
- Pediatric Gastroenterology and Liver Unit, Sapienza University of Rome, Rome, Italy
| | - M Bramuzzo
- Institute for Maternal and Child Health IRCCS 'Burlo Garofalo', Trieste, Italy
| | - L Norsa
- Pediatric Hepatology Gastroenterology and Transplantation, ASST Papa Giovanni XXIII, Bergamo, Italy
| | - S Arrigo
- Pediatric Gastroenterology Unit, Institute 'Giannina Gaslini', Genoa, Italy
| | - M Distante
- Pediatric Gastroenterology and Liver Unit, Sapienza University of Rome, Rome, Italy
| | - E Miele
- Department of Translational Medical Science, Section of Pediatrics, University of Naples 'Federico II', Naples, Italy
| | - C Romano
- Pediatric Gastroenterology and Endoscopy, University of Messina, Messina, Italy
| | - C Giobbi
- Pediatric Gastroenterology and Liver Unit, Sapienza University of Rome, Rome, Italy
| | - R Panceri
- Department of Pediatrics, University of Milano-Bicocca Fond., MBBM/Hosp., San Gerardo Monza, Italy
| | - S Cucchiara
- Pediatric Gastroenterology and Liver Unit, Sapienza University of Rome, Rome, Italy
| | - P Alvisi
- Pediatric Department, Maggiore Hospital, Bologna, Italy
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14
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Kern I, Schoffer O, Kiess W, Henker J, Laaß MW, Winkler U, Quietzsch J, Wenzel O, Zurek M, Büttner K, Fischer P, de Laffolie J, Manuwald U, Stange T, Zenker R, Weidner J, Zimmer KP, Kunath H, Kugler J, Richter T, Rothe U. Incidence trends of pediatric onset inflammatory bowel disease in the years 2000-2009 in Saxony, Germany-first results of the Saxon Pediatric IBD Registry. PLoS One 2021; 16:e0243774. [PMID: 33395450 PMCID: PMC7781364 DOI: 10.1371/journal.pone.0243774] [Citation(s) in RCA: 14] [Impact Index Per Article: 3.5] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/23/2020] [Accepted: 11/28/2020] [Indexed: 02/06/2023] Open
Abstract
Aims In developed countries, the incidence of inflammatory bowel disease (IBD) such as Crohn's disease (CD) and ulcerative colitis (UC) is increasing. Therefore, we aimed to investigate the incidence rates and trends over time in the population of children and adolescents in one of the federal states of Germany, in Saxony. Methods Over the 10-year period 2000–2009 all 31 children’s hospitals and pediatric gastroenterologists, respectively in Saxony reported all IBD patients up to 15 years of age to the Saxon Pediatric IBD Registry. The completeness of the registry was estimated as 96.7% by independent surveys in the years 2005–2009. Incidence rates were presented as age-standardized incidence rates (ASR) regarding New European Standard Population 1990 per 100,000 person-years (PY) with 95% confidence intervals [CI]. Joinpoint and linear regression was used for trend analyses. Results 344 patients with confirmed IBD between 2000–2009 were included in the epidemiological evaluation: 212 (61.6%) patients with CD, 122 (35.6%) with UC and 10 (2.9%) with unclassified IBD (IBD-U). The ASR per 100,000 PY over the whole observation period was 7.2 [6.4–7.9] for IBD, 4.4 [3.8–5.0] for CD, 2.6 [2.1–3.0] for UC and 0.2 [0.1–0.3] for IBD-U. For IBD, the ASR per 100,000 PY increased from 4.6 [2.8–6.3] in 2000 to 10.5 [7.5–13.6] in 2009. The incidence trend analysis of ASRs using the joinpoint regression confirmed a significant increase of IBD as well as UC. The mean age at first diagnosis decreased significantly during the observation period from 11.5 (11.0–13.4) in 2000 to 9.6 (5.1–13.5) years in 2009. The median of the diagnostic latency among IBD patients was 3 months. Conclusion The incidence of IBD in children and adolescents in Saxony was slightly higher than the average of other countries in the same time period and followed the trend towards a general increase of IBD. The age at diagnosis was subject to a very unfavorable downward trend.
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Affiliation(s)
- Ivana Kern
- Department of Health Sciences/Public Health, Institute and Policlinic for Occupational and Social Medicine, Faculty of Medicine “Carl Gustav Carus”, TU Dresden, Dresden, Germany
- * E-mail:
| | - Olaf Schoffer
- Center for Evidence-Based Healthcare, University Hospital and Faculty of Medicine “Carl Gustav Carus”, TU Dresden, Dresden, Germany
| | - Wieland Kiess
- Department of Women and Child Health, Hospital for Children and Adolescents, Center for Pediatric Research, University of Leipzig, Leipzig, Germany
| | - Jobst Henker
- Children's Center Dresden-Friedrichstadt, Dresden, Germany
| | - Martin W. Laaß
- Faculty of Medicine “Carl Gustav Carus”, University Hospital for Children and Adolescents, TU Dresden, Dresden, Germany
| | - Ulf Winkler
- Clinic for Children and Adolescents, Hospital Bautzen, Oberlausitz-Hospitals, Bautzen, Germany
| | - Jürgen Quietzsch
- Clinic for Children and Adolescents, DRK Hospital Lichtenstein, Lichtenstein, Germany
| | - Olaf Wenzel
- Clinic for Children and Adolescents, Helios Hospital Aue, Aue, Germany
| | - Marlen Zurek
- Clinic for Children and Adolescents, Hospital St. Georg, Leipzig, Germany
| | - Katrin Büttner
- Medical Care Centre—Polyclinic Spremberg, Spremberg, Germany
| | - Peter Fischer
- General Pediatrics for Children and Adolescents, Naunhof, Germany
| | - Jan de Laffolie
- Department of General Pediatrics, Children's Gastroenterology/Hepatology/Nutrition, Justus-Liebig-University Gießen, CEDATA-GPGE Working Group, Gießen, Germany
| | - Ulf Manuwald
- Department of Health Sciences/Public Health, Institute and Policlinic for Occupational and Social Medicine, Faculty of Medicine “Carl Gustav Carus”, TU Dresden, Dresden, Germany
| | - Thoralf Stange
- Institute for Medical Informatics and Biometry, Faculty of Medicine “Carl Gustav Carus”, TU Dresden, Dresden, Germany
| | - Ronny Zenker
- Department of General Practice, Medical Clinic 3, Faculty of Medicine “Carl Gustav Carus”, TU Dresden, Dresden, Germany
| | - Jens Weidner
- Department of General Practice, Medical Clinic 3, Faculty of Medicine “Carl Gustav Carus”, TU Dresden, Dresden, Germany
| | - Klaus-Peter Zimmer
- Department of General Pediatrics, Children's Gastroenterology/Hepatology/Nutrition, Justus-Liebig-University Gießen, CEDATA-GPGE Working Group, Gießen, Germany
| | - Hildebrand Kunath
- Faculty of Medicine “Carl Gustav Carus“, TU Dresden, Dresden, Germany
| | - Joachim Kugler
- Department of Health Sciences/Public Health, Institute and Policlinic for Occupational and Social Medicine, Faculty of Medicine “Carl Gustav Carus”, TU Dresden, Dresden, Germany
| | - Thomas Richter
- Clinic for Children and Adolescents, Hospital St. Georg, Leipzig, Germany
| | - Ulrike Rothe
- Department of Health Sciences/Public Health, Institute and Policlinic for Occupational and Social Medicine, Faculty of Medicine “Carl Gustav Carus”, TU Dresden, Dresden, Germany
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15
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Colman RJ, Dhaliwal J, Rosen MJ. Predicting Therapeutic Response in Pediatric Ulcerative Colitis-A Journey Towards Precision Medicine. Front Pediatr 2021; 9:634739. [PMID: 33681110 PMCID: PMC7925616 DOI: 10.3389/fped.2021.634739] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/28/2020] [Accepted: 01/08/2021] [Indexed: 12/12/2022] Open
Abstract
Ulcerative colitis (UC) is a disabling disease, characterized by chronic inflammation of the colon, with a rising prevalence worldwide in the pediatric age group. Although UC presents in children with varying severity, disease extent, and comorbidities, initial treatment is essentially uniform, consisting of 5-aminosalicylate drugs with corticosteroid induction for those with moderately to severely active disease. With the advent of anti-tumor necrosis factor (TNF) biologic therapy and several new biologics and small-molecule drugs for UC, precision medicine approaches to treatment are needed to more rapidly achieve sustained remission, restore quality of life, normalize development, and limit exposure to toxic corticosteroids in children with UC. Here, we review available data on clinical, biochemical, histopathologic, and molecular predictors of treatment response in UC. We also address known predictors and special treatment considerations in specific relevant scenarios such as very-early-onset UC, acute severe UC, ileal pouch anal anastomosis, and UC with concomitant primary sclerosing cholangitis. The review concludes with a prediction of how machine learning will integrate multimodal patient data to bring precision medicine to the bedside of children with UC in the future.
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Affiliation(s)
- Ruben J Colman
- Division of Gastroenterology, Hepatology and Nutrition, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, United States
| | - Jasbir Dhaliwal
- Division of Gastroenterology, Hepatology and Nutrition, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, United States.,Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, OH, United States
| | - Michael J Rosen
- Division of Gastroenterology, Hepatology and Nutrition, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, United States.,Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, OH, United States
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16
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Jezernik G, Mičetić-Turk D, Potočnik U. Molecular Genetic Architecture of Monogenic Pediatric IBD Differs from Complex Pediatric and Adult IBD. J Pers Med 2020; 10:E243. [PMID: 33255894 PMCID: PMC7712254 DOI: 10.3390/jpm10040243] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/20/2020] [Revised: 11/20/2020] [Accepted: 11/24/2020] [Indexed: 12/19/2022] Open
Abstract
Inflammatory bowel disease (IBD) manifests as a complex disease resulting from gene-environment interactions or as a monogenic disease resulting from deleterious mutations. While monogenic IBD is predominantly pediatric, only one-quarter of complex IBD is pediatric. In this study, we were the first to systematically compare genetic architecture between monogenic and complex pediatric and adult IBD on genetic and molecular pathway levels. Genes reported as causal for monogenic pediatric IBD and related syndromes and as risk factors for pediatric and adult complex IBD were analyzed using CytoScape and ClueGO software tools to elucidate significantly enriched Gene Ontology (GO) terms. Despite the small overlap (seven genes) between monogenic IBD genes (85) and complex IBD loci (240), GO analysis revealed several enriched GO terms shared between subgroups (13.9%). Terms Th17 cell differentiation and Jak/STAT signaling were enriched in both monogenic and complex IBD subgroups. However, primary immunodeficiency and B-cell receptor signaling pathway were specifically enriched only for pediatric subgroups, confirming existing clinical observations and experimental evidence of primary immunodeficiency in pediatric IBD patients. In addition, comparative analysis identified patients below 6 years of age to significantly differ from complex pediatric and adult IBD and could be considered a separate entity.
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Affiliation(s)
- Gregor Jezernik
- Faculty of Medicine, University of Maribor, Taborska Ulica 8, 2000 Maribor, Slovenia; (G.J.); (D.M.-T.)
| | - Dušanka Mičetić-Turk
- Faculty of Medicine, University of Maribor, Taborska Ulica 8, 2000 Maribor, Slovenia; (G.J.); (D.M.-T.)
| | - Uroš Potočnik
- Faculty of Medicine, University of Maribor, Taborska Ulica 8, 2000 Maribor, Slovenia; (G.J.); (D.M.-T.)
- Faculty of Chemistry and Chemical Engineering, University of Maribor, Smetanova Ulica 17, 2000 Maribor, Slovenia
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17
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Grossman A, Mauer E, Gerber LM, Long MD, Kappelman MD, Gupta N. Black/African American Patients with Pediatric Crohn's Disease Report Less Anxiety and Fatigue than White Patients. J Pediatr 2020; 225:146-151. [PMID: 32544479 DOI: 10.1016/j.jpeds.2020.05.042] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/18/2020] [Revised: 05/18/2020] [Accepted: 05/20/2020] [Indexed: 01/10/2023]
Abstract
OBJECTIVES To compare patient-reported outcomes in black/African American patients with white patients participating in IBD Partners Kids & Teens, in order to identify possible racial healthcare disparities in pediatric inflammatory bowel disease (IBD) as future targets for improvement. STUDY DESIGN This was a cross-sectional analysis comparing patient-reported outcomes in black/African American patients with white patients, aged 9-18 years, with IBD participating in the IBD Partners Kids & Teens cohort from August 2013 to April 2018. Secondary outcomes included number of IBD-related hospitalizations and surgeries, current medication use, and disease activity. RESULTS We included 401 patients with Crohn's disease (white = 378 [94%]; black/African American = 23 [6%]). For children with Crohn's disease, black/African American patients compared with white patients reported less anxiety (40.7 vs 47.5, P = .001) and fatigue (44.3 vs 48.4, P = .047) despite more frequently reported treatment with biologics (91% vs 61%, P = .006) and antibiotics (17% vs 5%, P = .03) and history of hospitalizations (81% vs 52%, P = .02). CONCLUSIONS Black/African American children with Crohn's disease were less likely to report anxiety or fatigue than white patients, despite an apparent more severe disease course reflected by greater reported frequency of treatment with biologics and antibiotics and history of hospitalizations.
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Affiliation(s)
| | - Elizabeth Mauer
- Department of Healthcare Policy and Research, Weill Cornell Medicine, New York, NY
| | - Linda M Gerber
- Department of Healthcare Policy and Research, Weill Cornell Medicine, New York, NY
| | - Millie D Long
- Department of Internal Medicine, University of North Carolina at Chapel Hill, Chapel Hill, NC; Center for Gastrointestinal Biology and Disease, Chapel Hill, NC
| | - Michael D Kappelman
- Department of Pediatrics, University of North Carolina at Chapel Hill, Chapel Hill, NC; Center for Gastrointestinal Biology and Disease, Chapel Hill, NC
| | - Neera Gupta
- Department of Pediatrics, Weill Cornell Medicine, New York, NY.
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18
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A baseline assessment of enhanced recovery protocol implementation at pediatric surgery practices performing inflammatory bowel disease operations. J Pediatr Surg 2020; 55:1996-2006. [PMID: 32713714 PMCID: PMC7606356 DOI: 10.1016/j.jpedsurg.2020.06.021] [Citation(s) in RCA: 19] [Impact Index Per Article: 3.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/28/2020] [Revised: 05/17/2020] [Accepted: 06/07/2020] [Indexed: 02/08/2023]
Abstract
BACKGROUND Enhanced recovery protocols (ERPs) have been used to improve patient outcomes and resource utilization after surgery. These evidence-based interventions include patient education, standardized anesthesia protocols, and limited fasting, but their use among pediatric populations is lagging. We aimed to determine baseline recovery practices within pediatric surgery departments participating in an ERP implementation trial for elective inflammatory bowel disease (IBD) operations. METHODS To measure baseline ERP adherence, we administered a survey to a staff surgeon in each of the 18 participating sites. The survey assessed demographics of each department and utilization of 21 recovery elements during patient encounter phases. Mixed-methods analysis was used to evaluate predictors and barriers to ERP element implementation. RESULTS The assessment revealed an average of 6.3 ERP elements being practiced at each site. The most commonly practiced elements were using minimally invasive techniques (100%), avoiding intraabdominal drains (89%), and ileus prophylaxis (72%). The preoperative phase had the most elements with no adherence including patient education, optimizing medical comorbidities, and avoiding prolonged fasting. There was no association with number of elements utilized and total number of surgeons in the department, annual IBD surgery volume, and hospital size. Lack of buy-in from colleagues, electronic medical record adaptation, and resources for data collection and analysis were identified barriers. CONCLUSIONS Higher intervention utilization for IBD surgery was associated with elements surgeons directly control such as use of laparoscopy and avoiding drains. Elements requiring system-level changes had lower use. The study characterizes the scope of ERP utilization and the need for effective tools to improve adoption. LEVEL OF EVIDENCE Level III. TYPE OF STUDY Mixed-methods survey.
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19
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Stapersma L, van den Brink G, van der Ende J, Szigethy EM, Groeneweg M, de Bruijne FH, Hillegers MHJ, Escher JC, Utens EMWJ. Psychological Outcomes of a Cognitive Behavioral Therapy for Youth with Inflammatory Bowel Disease: Results of the HAPPY-IBD Randomized Controlled Trial at 6- and 12-Month Follow-Up. J Clin Psychol Med Settings 2020; 27:490-506. [PMID: 31506853 PMCID: PMC7462914 DOI: 10.1007/s10880-019-09649-9] [Citation(s) in RCA: 10] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/07/2023]
Abstract
Youth with inflammatory bowel disease (IBD) often experience psychological difficulties, such as anxiety and depression. This randomized controlled study tested whether a 3-month disease-specific cognitive behavioral therapy (CBT) in addition to standard medical care versus standard medical care only was effective in improving these youth's psychological outcomes. As this study was aimed at prevention, we included 70 youth (10-25 years) with IBD and symptoms of subclinical anxiety and/or depression, and measured psychological outcomes at 6- and 12-month follow-up. In general, participants in both groups showed improvements in anxiety, depression, health-related quality of life, social functioning, coping, and illness perceptions, sustained until 12 months follow-up. Overall, we found no differences between those receiving additional CBT and those receiving standard medical care only. We assume that this can be explained by the perceived low burden (both somatically and psychologically) or heightened awareness of psychological difficulties and IBD. ClinicalTrials.gov NCT02265588.
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Affiliation(s)
- Luuk Stapersma
- Department of Child and Adolescent Psychiatry/Psychology, Erasmus MC-Sophia Children's Hospital, P.O. Box 2060, 3000 CB, Rotterdam, The Netherlands
| | - Gertrude van den Brink
- Department of Pediatric Gastroenterology, Erasmus MC-Sophia Children's Hospital, Rotterdam, The Netherlands
| | - Jan van der Ende
- Department of Child and Adolescent Psychiatry/Psychology, Erasmus MC-Sophia Children's Hospital, P.O. Box 2060, 3000 CB, Rotterdam, The Netherlands
| | - Eva M Szigethy
- Department of Psychiatry, University of Pittsburgh, Pittsburgh, PA, USA
| | - Michael Groeneweg
- Department of Pediatrics, Maasstad Hospital, Rotterdam, The Netherlands
| | | | - Manon H J Hillegers
- Department of Child and Adolescent Psychiatry/Psychology, Erasmus MC-Sophia Children's Hospital, P.O. Box 2060, 3000 CB, Rotterdam, The Netherlands
| | - Johanna C Escher
- Department of Pediatric Gastroenterology, Erasmus MC-Sophia Children's Hospital, Rotterdam, The Netherlands
| | - Elisabeth M W J Utens
- Department of Child and Adolescent Psychiatry/Psychology, Erasmus MC-Sophia Children's Hospital, P.O. Box 2060, 3000 CB, Rotterdam, The Netherlands.
- Research Institute of Child Development and Education, University of Amsterdam, Amsterdam, The Netherlands.
- Academic Center for Child Psychiatry the Bascule/Department of Child and Adolescent Psychiatry, Academic Medical Center, Amsterdam, The Netherlands.
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20
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Extraintestinal Manifestations Are Associated With Disease Severity in Pediatric Onset Inflammatory Bowel Disease. J Pediatr Gastroenterol Nutr 2020; 71:40-45. [PMID: 32187141 DOI: 10.1097/mpg.0000000000002707] [Citation(s) in RCA: 13] [Impact Index Per Article: 2.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 01/04/2023]
Abstract
OBJECTIVES The aim of this study was to investigate a possible association between extraintestinal manifestations (EIM) and a more severe disease course in pediatric onset inflammatory bowel disease (pIBD). METHODS This study compares the disease course of pIBD patients (IBD diagnosis <15 years of age) with and without EIM in a population-based cohort from Denmark. Patients diagnosed with pIBD between 1998 and 2008 were included in the study and followed until December 31, 2014. Data on phenotype, treatment, relapses, and the temporal relationship between IBD relapses and activity of EIM were collected at end of follow-up by manual revision of patient charts. RESULTS Of 333 pIBD patients, 14 (4.2%) had EIM at time of diagnosis and 47 (14.1%) developed EIM during follow-up. Median follow-up time was 9.6 years for patients with EIM and 8.8 years for patients without. In ulcerative colitis, EIM were associated with an increased risk of biological treatment and surgery (hazard ratio: 2.6; 95% confidence interval [CI]: 1.3-5.5, P = 0.008 and 2.9 [95% CI: 1.1-7.7, P = 0.03], respectively). In Crohn disease, EIM were associated with an increased relapse rate (1.3 [95% CI: 1.1-1.5], P = 0.001). Lastly, we found a positive temporal relationship between relapse of IBD and EIM activity. CONCLUSION The presence of EIM is associated with a more severe disease course in pIBD. This should be considered when deciding treatment options, as a more aggressive treatment approach could be warranted in patients with EIM. However, prospective studies are needed to fully evaluate this.
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21
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Davies M, Dodd S, Coultate M, Ross A, Pears G, Gnaneswaran B, Tzivinikos C, Konidari A, Cheng J, Auth MK, Cameron F, Tamhne S, Renji E, Nair M, Baillie C, Collins P, Smith PJ, Subramanian S. From Paris to Montreal: disease regression is common during long term follow-up of paediatric Crohn's disease. Scand J Gastroenterol 2020; 55:148-153. [PMID: 31928099 DOI: 10.1080/00365521.2019.1710765] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/04/2023]
Abstract
Introduction: Paediatric Crohn's disease (PCD) often presents with extensive and a frequent pan-enteric phenotype at onset. However, its long term evolution into adulthood, especially since the widespread use of biological agents, is not well characterised. We conducted a single centre cohort study of all PCD patients transitioned to adult care to assess the long term disease evolution in the era of biologic therapy.Methods: We conducted a retrospective observational, study of all PCD patients who were subsequently transferred to the care of an adult gastroenterology unit and had a minimum follow up of 2 years. We examined the case notes for evolution of disease location and behaviour. Disease location and behaviour was characterised using Paris classification at diagnosis and Montreal classification at last follow-up. In addition, we examined variables associated with complicated disease behaviour and the need for CD related intestinal resection.Results: In total, 132 patients were included with a median age at diagnosis of 13 (IQR 11-14) and a median follow up of 11 years (range 4-14). At diagnosis, 23 (17.4%), 39 (29.6%) and 70 (53%) patients had ileal, colonic and ileocolonic disease respectively. In addition, 31 (23.5%) patients had L4a or L4b disease at diagnosis (proximal or distal to the ligament of treitz respectively) and 13 patients (9.8%) had both whilst 27 (20.4%) patients had perianal disease. At diagnosis, 27 (20.4%) patients had complicated disease behaviour but 83 (62.9)% of patients had an extensive 'pan-enteric' phenotype. Of these patients only 55 (66.3%) retained the pan-enteric phenotype at last follow-up (p = .0002). Disease extension was noted in 25 (18.9%) of patients and regression was noted in 47 (35.6%) of patients, whereas upper GI disease was noted in significantly fewer patients at last follow-up (21, 15.9%) (p = .0001). More patients had complicated disease behaviour (46 patients, 34.9%, p = .0018) at last follow-up. There was a high exposure to both thiopurines 121 (91.7%) and biologics 84 (63.6%). The cumulative probability (95% CI) of surgery was 0.05 (0.02, 0.11) at 1 year, 0.17 (0.11, 0.24) at 3 years and 0.22 (0.15, 0.30) at 5 years. Neither disease location nor behaviour were associated with the need for intestinal resectional surgery.Conclusions: Over the course of an extended follow-up period, there appeared to be changes in both disease location and behaviour in PCD. Interestingly, a significant proportion of patients had disease involution which may be related to a high rate of exposure to thiopurines and biologics. We were unable to identify any variables associated with complicated disease course or the need for intestinal surgery.
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Affiliation(s)
- Mike Davies
- Department of Gastroenterology, Royal Liverpool, and Broadgreen University Hospital NHS Trust, Liverpool, UK
| | - Susanna Dodd
- Department of Biostatistics, University of Liverpool, Liverpool, UK
| | - Morwenna Coultate
- Department of Gastroenterology, Royal Liverpool, and Broadgreen University Hospital NHS Trust, Liverpool, UK
| | - Andrew Ross
- Department of Gastroenterology, Royal Liverpool, and Broadgreen University Hospital NHS Trust, Liverpool, UK
| | - George Pears
- Department of Gastroenterology, Royal Liverpool, and Broadgreen University Hospital NHS Trust, Liverpool, UK
| | - Bruno Gnaneswaran
- Department of Gastroenterology, Royal Liverpool, and Broadgreen University Hospital NHS Trust, Liverpool, UK
| | - Christos Tzivinikos
- Pediatric Gastroenterology, Hepatology and Nutrition, Alder Hey Children's NHS Foundation Trust, Liverpool, UK
| | - Anastasia Konidari
- Pediatric Gastroenterology, Hepatology and Nutrition, Alder Hey Children's NHS Foundation Trust, Liverpool, UK
| | - Jeng Cheng
- Pediatric Gastroenterology, Hepatology and Nutrition, Alder Hey Children's NHS Foundation Trust, Liverpool, UK
| | - Marcus Kh Auth
- Pediatric Gastroenterology, Hepatology and Nutrition, Alder Hey Children's NHS Foundation Trust, Liverpool, UK.,Department of Cellular and Molecular Physiology, University of Liverpool, Liverpool, UK
| | - Fiona Cameron
- Pediatric Gastroenterology, Hepatology and Nutrition, Alder Hey Children's NHS Foundation Trust, Liverpool, UK
| | - Sarang Tamhne
- Pediatric Gastroenterology, Hepatology and Nutrition, Alder Hey Children's NHS Foundation Trust, Liverpool, UK
| | - Elizabeth Renji
- Pediatric Gastroenterology, Hepatology and Nutrition, Alder Hey Children's NHS Foundation Trust, Liverpool, UK
| | - Manjula Nair
- Pediatric Gastroenterology, Hepatology and Nutrition, Alder Hey Children's NHS Foundation Trust, Liverpool, UK
| | - Colin Baillie
- Department of Surgery, Alder Hey Children's NHS Foundation Trust, Liverpool, UK
| | - Paul Collins
- Department of Gastroenterology, Royal Liverpool, and Broadgreen University Hospital NHS Trust, Liverpool, UK
| | - Philip J Smith
- Department of Gastroenterology, Royal Liverpool, and Broadgreen University Hospital NHS Trust, Liverpool, UK
| | - Sreedhar Subramanian
- Department of Gastroenterology, Royal Liverpool, and Broadgreen University Hospital NHS Trust, Liverpool, UK.,Department of Cellular and Molecular Physiology, University of Liverpool, Liverpool, UK
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22
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Long-term outcome of immunomodulator use in pediatric patients with inflammatory bowel disease. Dig Liver Dis 2020; 52:164-172. [PMID: 31640916 DOI: 10.1016/j.dld.2019.09.008] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/28/2019] [Revised: 09/02/2019] [Accepted: 09/09/2019] [Indexed: 12/11/2022]
Abstract
OBJECTIVES In the era where new biologicals are entering the market, the place of immunomodulators in the treatment of pediatric inflammatory bowel disease (IBD) needs to be reassessed. METHODS All children with Crohn's disease (CD) or ulcerative colitis (UC) followed at our center over the last 10 years were reviewed. Children who received conventional therapy (including 5-aminosalicylates, steroids, thiopurines and methotrexate) since diagnosis were included. Primary outcome was steroid-free clinical remission without need for rescue therapy (biologics or surgery) at 6 and 12 months after diagnosis and at last follow-up. Cox proportional hazard modelling was performed to determine variables at diagnosis associated with outcomes. RESULTS In total, 176 IBD patients (121 CD, 55 UC) were identified with a median follow-up of 4.6 [2.0-8.1] years. Remission rates were 79.6% at month 6, but decreased to 60.2% at month 12, and 31.8% at last follow-up. Higher CRP [1.006 (1.001-1.011)], lower albumin [1.050 (1.012-1.086)] and growth impairment [1.214 (1.014-1.373)] in CD patients and higher PUCAI score [1.038 (1.006-1.072)] and low iron [1.023 (1.003-1.043)] in UC patients were associated with treatment failure (all p < 0.05). CONCLUSION Only 32% pediatric IBD patients will remain free of biologics or surgery 5-years after diagnosis. Especially children with a high disease burden at diagnosis were more likely to fail conventional therapy.
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23
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Abstract
The landscape of pediatric inflammatory bowel disease is rapidly evolving. The therapeutic advances seen in the adult arena are rapidly being adopted by pediatric gastroenterologists and evaluated in both controlled trials and real-world experience. Though anti-tumor necrosis factor agents have been the primary therapy over the last decade, recently there has been an expansion of therapeutic targets and alternative mechanism of action drugs with a focus on individualized and personalized therapy. By reviewing epidemiology, pathophysiology, and goals of treatment, we hope to frame the discussion of current and novel therapeutics for the pediatric gastroenterologist. As scientific discovery continues to push the envelope in defining our understanding of pediatric inflammatory bowel disease, the current era of therapeutics gives us hope that a cure may be realized soon.
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Affiliation(s)
- Bhaskar Gurram
- University of Texas Southwestern Medical Center, Dallas, TX, 75390, USA
| | - Ashish S. Patel
- University of Texas Southwestern Medical Center, Dallas, TX, 75390, USA
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24
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Lund K, Larsen MD, Knudsen T, Kjeldsen J, Nielsen RG, Nørgård BM. The impact of anti-TNFα therapy on colectomy rates and corticosteroid treatment among 3001 paediatric and adolescent patients with ulcerative colitis-a nationwide study from 1995 to 2015. Aliment Pharmacol Ther 2019; 50:1077-1085. [PMID: 31579961 DOI: 10.1111/apt.15510] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/02/2019] [Revised: 06/07/2019] [Accepted: 08/29/2019] [Indexed: 01/08/2023]
Abstract
BACKGROUND The long-term effects of anti-TNFα therapy in ulcerative colitis are debatable. AIM To examine whether anti-TNFα therapy changed the colectomy proportion and reduced the use of corticosteroids. METHODS A nationwide cohort study among patients (age 0-20) diagnosed with ulcerative colitis through 1995-2015 from Danish health registries. We calculated the cumulative 5-year risk of colectomy after diagnosis and used a Cox regression model for comparison between a historical pre-anti-TNFα cohort 1 (1995-2003) and a cohort 2 for the era of anti-TNFα (2004-2015). Based on anti-TNFα users, defined as patients who had at least four anti-TNFα treatments within 4 months, we examined the subsequent need for corticosteroids. RESULTS We identified 3001 patients from 1995 to 2015. The 5-year cumulative proportion of colectomy in cohort 2 was 9.7% (95% confidence interval [CI] 8.4-11.1) and 12.3% (95% CI 10.4-14.6) in cohort 1. The adjusted 5-year hazard ratio (HR) was 0.76 (95% CI 0.60-0.96) for colectomy in cohort 2 compared to cohort 1. A total of 334 patients received anti-TNFα treatments, and 16.8% (56/334) were prescribed corticosteroids in the subsequent 3-month period. Corticosteroid treatment declined with follow-up after 6 and 12 months, 5.4% and 1.2%, respectively. CONCLUSION In patient's ≤20 years, the HR for colectomy within a period of 5 years from the time of diagnosis was reduced in the era of anti-TNFα compared to a historical cohort. In patients treated with anti-TNFα, prescriptions of corticosteroids were virtually ceased after 12 months.
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Affiliation(s)
- Ken Lund
- Center for Clinical Epidemiology, Odense University Hospital, Odense, Denmark.,Research Unit of Clinical Epidemiology, Department of Clinical Research, University of Southern Denmark, Odense, Denmark
| | - Michael Due Larsen
- Center for Clinical Epidemiology, Odense University Hospital, Odense, Denmark.,Research Unit of Clinical Epidemiology, Department of Clinical Research, University of Southern Denmark, Odense, Denmark
| | - Torben Knudsen
- Department of Medicine, Hospital of Southwest Jutland, Esbjerg, Denmark.,Institute for Regional Health Science, Center Southwest Jutland, University of Southern Denmark, Esbjerg, Denmark
| | - Jens Kjeldsen
- Department of Medical Gastroenterology S, Odense University Hospital, Odense, Denmark.,Research Unit of Medical Gastroenterology, Department of Clinical Research, University of Southern Denmark, Odense, Denmark
| | - Rasmus Gaardskjaer Nielsen
- Hans Christian Andersen Children's Hospital, Odense University Hospital, Odense, Denmark.,Research Unit of Pediatric, Department of Clinical Research, University of Southern Denmark, Odense, Denmark
| | - Bente Mertz Nørgård
- Center for Clinical Epidemiology, Odense University Hospital, Odense, Denmark.,Research Unit of Clinical Epidemiology, Department of Clinical Research, University of Southern Denmark, Odense, Denmark
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25
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van den Brink G, van Gaalen MAC, de Ridder L, van der Woude CJ, Escher JC. Health Care Transition Outcomes in Inflammatory Bowel Disease: A Multinational Delphi Study. J Crohns Colitis 2019; 13:1163-1172. [PMID: 30766997 PMCID: PMC7142327 DOI: 10.1093/ecco-jcc/jjz044] [Citation(s) in RCA: 22] [Impact Index Per Article: 3.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/23/2022]
Abstract
BACKGROUND Transition programmes are designed to prepare adolescent inflammatory bowel disease [IBD] patients for transfer to adult care. It is still unclear which outcome parameters define 'successful transition'. Therefore, this study aimed to identify outcomes important for success of transition in IBD. METHODS A multinational Delphi study in patients, IBD nurses, and paediatric and adult gastroenterologists was conducted. In stage 1, panellists commented on an outcome list. In stage 2, the refined list was graded from 1 to 9 [least to very important], by an expert and a patient panel. In stage 3, the expert panel ranked important outcomes from 1 to 10 [least to most important]. Descriptive statistics and Mann-Whitney U-tests were performed. RESULTS The final item list developed in stage 1 was tested by the expert [n = 74 participants, 52.7% paediatric] and patient panel [n = 61, aged 16-25 years, 49.2% male]. Respectively, ten and 11 items were found to be important by the expert and patient panel. Both panels agreed on eight of these items, of which six reflected self-management skills. In stage 3, the expert panel formed a top-ten list. The three most important items were: decision-making regarding IBD [mean score 6.7], independent communication [mean score 6.3] and patient satisfaction [mean score 5.8]. CONCLUSION This is the first study identifying outcomes that IBD healthcare providers and patients deem important factors for successful transition. Self-management skills were considered more important than IBD-specific items. This is a first step to further define success of transition in IBD and subsequently evaluate the efficacy of different transition models.
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Affiliation(s)
- Gertrude van den Brink
- Department of Paediatric Gastroenterology, Erasmus MC-Sophia Children’s Hospital, Rotterdam, The Netherlands
| | - Martha A C van Gaalen
- Department of Paediatric Gastroenterology, Erasmus MC-Sophia Children’s Hospital, Rotterdam, The Netherlands
| | - Lissy de Ridder
- Department of Paediatric Gastroenterology, Erasmus MC-Sophia Children’s Hospital, Rotterdam, The Netherlands
| | | | - Johanna C Escher
- Department of Paediatric Gastroenterology, Erasmus MC-Sophia Children’s Hospital, Rotterdam, The Netherlands,Corresponding author: Professor J. C. Escher, MD, PhD, Department of Paediatric Gastroenterology, Erasmus MC-Sophia, Wytemaweg 80, 3015 CN Rotterdam, The Netherlands. Tel: +31-10-7036076;
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26
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Stapersma L, van den Brink G, van der Ende J, Bodelier AG, van Wering HM, Hurkmans PCWM, Mearin ML, van der Meulen-de Jong AE, Escher JC, Utens EMWJ. Illness Perceptions and Depression Are Associated with Health-Related Quality of Life in Youth with Inflammatory Bowel Disease. Int J Behav Med 2019; 26:415-426. [PMID: 31183787 PMCID: PMC6652166 DOI: 10.1007/s12529-019-09791-6] [Citation(s) in RCA: 27] [Impact Index Per Article: 4.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Download PDF] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/12/2022]
Abstract
BACKGROUND In youth with inflammatory bowel disease (IBD), health-related quality of life (HRQOL) has been shown to be affected by individual disease factors and specific psychological factors. The innovative aim of this study is to examine the combined impact of psychological factors (illness perceptions, cognitive coping, anxiety, and depression) on HRQOL, over and above the associations of demographic and disease factors with HRQOL in youth with IBD. METHOD Data on clinical disease activity, illness perceptions, cognitive coping, anxiety, depression, and HRQOL were prospectively collected in 262 consecutive youth (age 10-20, 46.6% male) with confirmed IBD. Multiple linear regression analyses tested the associations of demographic, disease, and psychological variables with HRQOL in separate groups for Crohn's disease (CD; N = 147) and ulcerative colitis and IBD unclassified (UC/IBD-U; N = 115), using age-specific validated instruments. RESULTS In both disease groups, more negative illness perceptions (ß = - .412; ß = - .438, p < .001) and more depression (ß = - .454; ß = - .279, p < .001) were related to lower HRQOL. In the UC/IBD-U group, more anxiety was related to lower HRQOL (ß = - .201, p = .001). The model with the psychological variables explained a large and significant amount of variance in both groups: 74% and 83%, respectively (p < .001). CONCLUSION In 10-20-year-old IBD patients, negative illness perceptions and depression were significantly and more strongly associated with lower HRQOL than demographic and disease factors. Thus, it is important to integrate psychological factors in the treatment for IBD patients. To improve HRQOL in young IBD patients, psychological interventions should be targeted at negative illness perceptions and depression.
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Affiliation(s)
- Luuk Stapersma
- Department of Child and Adolescent Psychiatry/Psychology, Erasmus MC-Sophia Children's Hospital, Wytemaweg 8, 3015 CN, Rotterdam, The Netherlands
| | - Gertrude van den Brink
- Department of Pediatric Gastroenterology, Erasmus MC-Sophia Children's Hospital, Rotterdam, The Netherlands
| | - Jan van der Ende
- Department of Child and Adolescent Psychiatry/Psychology, Erasmus MC-Sophia Children's Hospital, Wytemaweg 8, 3015 CN, Rotterdam, The Netherlands
| | | | | | | | - M Luisa Mearin
- Department of Pediatrics, Leiden University Medical Center, Leiden, The Netherlands
| | | | - Johanna C Escher
- Department of Pediatric Gastroenterology, Erasmus MC-Sophia Children's Hospital, Rotterdam, The Netherlands
| | - Elisabeth M W J Utens
- Department of Child and Adolescent Psychiatry/Psychology, Erasmus MC-Sophia Children's Hospital, Wytemaweg 8, 3015 CN, Rotterdam, The Netherlands.
- Research Institute of Child Development and Education, University of Amsterdam, Amsterdam, The Netherlands.
- Academic Center for Child Psychiatry the Bascule/Department of Child and Adolescent Psychiatry, Academic Medical Center, Amsterdam, The Netherlands.
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27
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Muntean A, Stoica I, McMahon SV, Mortell A, Gillick J, Sweeney BT. Colectomies in children with inflammatory bowel disease: a national referral centre experience. Pediatr Surg Int 2019; 35:691-698. [PMID: 30847603 DOI: 10.1007/s00383-019-04467-4] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Accepted: 03/02/2019] [Indexed: 12/15/2022]
Abstract
OBJECTIVES Few studies have reported on the utility and efficacy of laparoscopic colectomy in the paediatric population. We aim to compare the characteristics and outcomes of children with inflammatory bowel disease (IBD) who underwent open vs laparoscopic treatment at our centre. METHODS A 10-year retrospective review was performed. Collected data included demographics, indication for surgery, operative characteristics, histopathology results, post-operative course and peri-operative complications. RESULTS A total of 58 patients underwent subtotal colectomy for IBD, with 38 laparoscopic procedures. The cohort included 33 males and 25 females with a mean age at surgery of 12.9 ± 3.3 years. The pre-operative diagnosis was ulcerative colitis in n = 41, Crohn's disease in n = 5 and indeterminate colitis in n = 11. There was an 84.2% concordance between the pre-operative and the post-operative histological diagnoses. Overall, 34 (58.6%) patients had complications, of which 19 (32.7%) patients required return to theatre. The complication rate was significantly smaller for laparoscopic compared to open procedures (42.1% vs 75%) as well as for elective compared with emergency colectomies (38.4% vs 75%). Four patients (10.5%) required conversion to open approach. CONCLUSIONS Laparoscopic approach is feasible and safe in most paediatric patients with IBD and has a lower complication rate and better recovery than open procedures, especially when performed in an elective setting.
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Affiliation(s)
- Ancuta Muntean
- Department of Paediatric Surgery, Our Lady's Children Hospital, Crumlin, Dublin, Ireland. .,Department of Paediatric Surgery, Temple Street Children's Hospital, Dublin, Ireland.
| | - Ionica Stoica
- Department of Paediatric Surgery, Our Lady's Children Hospital, Crumlin, Dublin, Ireland.,Department of Paediatric Surgery, Temple Street Children's Hospital, Dublin, Ireland
| | | | - Alan Mortell
- Department of Paediatric Surgery, Our Lady's Children Hospital, Crumlin, Dublin, Ireland.,Department of Paediatric Surgery, Temple Street Children's Hospital, Dublin, Ireland
| | - John Gillick
- Department of Paediatric Surgery, Our Lady's Children Hospital, Crumlin, Dublin, Ireland.,Department of Paediatric Surgery, Temple Street Children's Hospital, Dublin, Ireland
| | - Brian Terence Sweeney
- Department of Paediatric Surgery, Our Lady's Children Hospital, Crumlin, Dublin, Ireland
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Abuquteish D, Putra J. Upper gastrointestinal tract involvement of pediatric inflammatory bowel disease: A pathological review. World J Gastroenterol 2019; 25:1928-1935. [PMID: 31086461 PMCID: PMC6487385 DOI: 10.3748/wjg.v25.i16.1928] [Citation(s) in RCA: 30] [Impact Index Per Article: 5.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/19/2019] [Revised: 04/03/2019] [Accepted: 04/10/2019] [Indexed: 02/06/2023] Open
Abstract
Upper gastrointestinal (UGI) tract involvement of inflammatory bowel disease (IBD) is commonly seen in pediatric patients. Upper endoscopy is included in the routine workup of children with suspected IBD to enhance the diagnosis and management of these patients. Currently, childhood IBD is classified into ulcerative colitis (UC), atypical UC, Crohn's disease (CD) and IBD unclassified. Histologic confirmation of UGI tract involvement, in particular the presence of epithelioid (non-caseating) granulomas, is helpful in confirming the diagnosis of IBD and its classification. Herein, we reviewed selected IBD-associated UGI tract manifestations in children. Lymphocytic esophagitis, seen predominantly in CD, is histologically characterized by increased intraepithelial lymphocytes (> 20 in one high-power field) in a background of mucosal injury with absence of granulocytes. Focally enhanced gastritis is a form of gastric inflammation in pediatric IBD marked by a focal lymphohistiocytic pit inflammation with or without granulocytes and plasma cells in a relatively normal background gastric mucosa. Duodenal inflammation seen in children with IBD includes cryptitis, villous flattening, increased intraepithelial lymphocytes, and lamina propria eosinophilia. Finally, epithelioid granulomas not associated with ruptured gland/crypt are a diagnostic feature of CD. The clinicopathologic correlation and differential diagnosis of each microscopic finding are discussed. Clinicians and pathologists should be cognizant of the utility and limitations of these histologic features.
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Affiliation(s)
- Dua Abuquteish
- Division of Pathology, Department of Paediatric Laboratory Medicine, Hospital for Sick Children, Toronto, ON M5G 1X8, Canada
| | - Juan Putra
- Division of Pathology, Department of Paediatric Laboratory Medicine, Hospital for Sick Children, Toronto, ON M5G 1X8, Canada
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29
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Perler B, Ungaro R, Baird G, Mallette M, Bright R, Shah S, Shapiro J, Sands BE. Presenting symptoms in inflammatory bowel disease: descriptive analysis of a community-based inception cohort. BMC Gastroenterol 2019; 19:47. [PMID: 30940072 PMCID: PMC6446285 DOI: 10.1186/s12876-019-0963-7] [Citation(s) in RCA: 80] [Impact Index Per Article: 13.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/11/2019] [Accepted: 03/18/2019] [Indexed: 12/20/2022] Open
Abstract
BACKGROUND Few data are currently available on the initial presenting symptoms of patients with inflammatory bowel disease (IBD). METHODS We evaluated the initial symptom presentation of patients with IBD in the Ocean State Crohn's and Colitis Area Registry (OSCCAR), a community-based inception cohort that enrolled Rhode Island IBD patients at time of diagnosis with longitudinal follow up. A 41-question symptom inventory was administered at time of enrollment to capture symptoms experienced during the 4 weeks preceding diagnosis of IBD. Frequencies of presenting symptoms were calculated. Principal component analysis (PCA) with promax rotation was used to examine possible symptom profiles among Crohn's disease (CD) and ulcerative colitis (UC) patients, respectively. Using the Scree plot, the 4-component solution was found to be optimal for both CD and UC. RESULTS A total of 233 CD and 150 UC patients were included. The most common presenting symptoms in CD were tiredness/fatigue (80.6%) and abdominal pain (80.4%) while passage of blood with bowel movements (BM) (86.6%) and loose/watery BMs (86.5%) were most common in UC. The 5 symptoms with greatest differences between UC and CD were passage of blood with BM (UC 86.6%/CD 45.3%), urgent BM (UC 82.5%/CD 63.9%), passage of mucus with BM (UC 67.7%/CD 36.9%), passage of blood from the anus (UC 59.7%/CD 32.1%), and anxiety about distance from bathroom (UC 59%/CD 38.7%). The PCA analysis yielded a 4 symptom components solution for CD and UC. CONCLUSION The most common presenting symptoms in CD are fatigue and abdominal pain while in UC bloody BM and diarrhea are most common. Distinct symptom phenotypes are seen with PCA analysis. Our study demonstrates symptomatic similarities and differences between CD and UC and suggests that patients may also be classified by symptom phenotype at time of diagnosis.
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Affiliation(s)
- Bryce Perler
- Department of Medicine, Warren Alpert Medical School of Brown University, Providence, USA
| | - Ryan Ungaro
- Dr. Henry D. Janowitz Division of Gastroenterology, Icahn School of Medicine at Mount Sinai, New York, USA
| | - Grayson Baird
- Lifespan Biostatistics Core, Rhode Island Hospital, Providence, USA
| | - Meaghan Mallette
- Department of Gastroenterology, Warren Alpert Medical School of Brown University, Providence, USA
| | - Renee Bright
- Department of Gastroenterology, Warren Alpert Medical School of Brown University, Providence, USA
| | - Samir Shah
- Department of Gastroenterology, Warren Alpert Medical School of Brown University, Providence, USA
| | - Jason Shapiro
- Department of Pediatric Gastroenterology, Warren Alpert Medical School of Brown University, Providence, USA
| | - Bruce E. Sands
- Dr. Henry D. Janowitz Division of Gastroenterology, Icahn School of Medicine at Mount Sinai, New York, USA
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Long-term outcome of pediatric-onset Crohn's disease: A population-based cohort study. Dig Liver Dis 2019; 51:496-502. [PMID: 30611597 DOI: 10.1016/j.dld.2018.11.033] [Citation(s) in RCA: 43] [Impact Index Per Article: 7.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/30/2018] [Revised: 11/23/2018] [Accepted: 11/26/2018] [Indexed: 12/11/2022]
Abstract
BACKGROUND Pediatric-onset Crohn's disease (CD) may represent a more severe form of disease. The aim of this study was to describe long-term outcome and identify associated risk factors of complicated behavior in a large population-based pediatric-onset CD cohort. PATIENTS AND METHODS Cases included all patients recorded in the EPIMAD registry diagnosed with definite or probable CD between January 1988 and December 2004, under the age of 17 years at the time of diagnosis, with at least two years of follow-up. RESULTS Five hundred and thirty-five patients were included. Median follow-up was 11.1 years [IQR, 7.3-15.0]. At the end of follow-up, 8% (n = 44) of patients had pure ileal disease (L1), 8% (n = 44) had pure colonic disease (L2), and 83% (n = 439) had ileocolonic disease (L3). L4 disease and perianal disease were observed in 42% (n = 227) and 16% (n = 85) of patients, respectively. At the end of follow-up, 58% (n = 308) of patients presented complicated disease behavior (B2, 39% and B3, 19%), and 42% (n = 163) of patients with inflammatory behavior at diagnosis had evolved to complicated behavior. During follow-up, 86% of patients (n = 466) received at least one course of corticosteroids, 67% (n = 357) of patients had been exposed to immunosuppressants and 35% (n = 187) of patients received at least one anti-TNF agent. Forty-three percent (n = 230) of patients underwent at least one intestinal resection. The overall mortality rate was 0.93% and the SMR was 1.6 [0.5-3.8] (p = 0.20). Five cancers were reported with a crude cancer incidence rate of 1.1% and an SIR of 3.3 [1.2-7.0] (p = 0.01). In a multivariate Cox model, ileal (HR, 1.87 [1.09-3.21], p = 0.022) or ileocolonic (HR, 1.54 [1.01-2.34], p = 0.042) and perianal lesions at diagnosis (HR, 1.81 [1.13- 2.89], p = 0.013) were significantly associated with complicated behavior. CONCLUSION About 80% of patients with pediatric-onset CD presented extensive ileocolonic disease during follow-up. The majority of patients evolved to complicated behavior. Surgery, cancer and mortality were observed in 43%, 0.9% and 0.9% of patients, respectively.
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Pediatric Chronic Inflammatory Bowel Disease in a German Statutory Health INSURANCE-Incidence Rates From 2009 to 2012. J Pediatr Gastroenterol Nutr 2019; 68:244-250. [PMID: 30672768 DOI: 10.1097/mpg.0000000000002162] [Citation(s) in RCA: 21] [Impact Index Per Article: 3.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/13/2022]
Abstract
OBJECTIVE The incidence of pediatric inflammatory bowel disease (PIBD) varies over time and geographic region. We attempted to generate incidence rates form German health insurance data. METHODS We used health care data for 2009-2015 provided by BARMER, a major statutory health insurance company in Germany, insuring approximately 8% of the pediatric population. We applied a Canadian case definition for PIBD based on International Classification of Diseases coding, documentation of (ileo)colonoscopy and the number of PIBD related visits, without external validation for Germany. An internal validation of the specificity of the diagnosis by checking whether the identified incident cases had also prescriptions of PIPD specific drugs was performed. RESULTS In 2012, 187 pediatric patients were newly diagnosed, accounting for an overall PIBD incidence of 17.41 (95% CI 15.08-20.10) per 100,000 insured children and adolescents from 0 to 17.9 years per year compared with 13.65/100,000 (95% CI 11.63-16.01) in 2009. The age-specific incidence showed a steep increase as of the age of 7 years. The PIBD prevalence in 2012 was 66.29/100,000. CONCLUSIONS In conclusion, the incidence of PIBD in 0 to 17.9-year-olds in Germany with health BARMER health insurance in 2012 is among the highest reported in the literature.
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Duricova D, Sarter H, Savoye G, Leroyer A, Pariente B, Armengol-Debeir L, Bouguen G, Ley D, Turck D, Templier C, Buche S, Peyrin-Biroulet L, Gower-Rousseau C, Fumery M. Impact of Extra-Intestinal Manifestations at Diagnosis on Disease Outcome in Pediatric- and Elderly-Onset Crohn's Disease: A French Population-Based Study. Inflamm Bowel Dis 2019; 25:394-402. [PMID: 30085159 DOI: 10.1093/ibd/izy254] [Citation(s) in RCA: 18] [Impact Index Per Article: 3.0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/27/2018] [Indexed: 12/16/2022]
Abstract
BACKGROUND Extraintestinal manifestations (EIM) have been associated with more severe course of inflammatory bowel disease. The aim was to study the frequency of EIM in pediatric- and elderly-onset Crohn's disease (CD) and the factors associated with EIM and their impact on long-term disease outcome. METHODS Pediatric- (age at diagnosis younger than 17 years) and elderly-onset CD patients (age at diagnosis 60 years or older) from a prospective population-based registry (EPIMAD) were recruited. Data on EIM and clinical factors at diagnosis and at maximal follow-up were collected. RESULTS We included 535 pediatric- and 370 elderly-onset patients (median age 14.5 and 69.9 years; median follow-up 11.1 and 5.9 years). Extraintestinal manifestations presented in 23.5% of childhood-onset and 4.9% of elderly-onset individuals at diagnosis, while in 29.8% and 5.9% of patients, EIM developed newly during the follow-up (hazard ration [HR] 4.4, 95% CI, 2.7-7.0, P < 0.001). The most frequently involved organ in both age cohorts, either at diagnosis or during disease course, were joints (pediatric: 11.2% and 22.6%; elderly: 3.2% and 3.5%, respectively) followed by skin (pediatric: 15.9% and 13.6%; elderly: 2.7% and 2.7%, respectively). Extraintestinal manifestations at diagnosis were associated with increased risk for corticosteroids (HR 1.42, 95% CI, 1.14-1.78 and HR 3.38, 95% CI, 1.88-6.08) and immunosuppressive therapy (HR 1.30, 95% CI, 1.02-1.65 and HR 4.24, 95% CI, 1.91-9.42), in both age populations. CONCLUSIONS Extraintestinal manifestations occurred at lower frequency in elderly-onset compared with pediatric-onset patients. In both age populations, presence of EIM at diagnosis independently increased the need for corticosteroid and immunosuppressive treatment.
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Affiliation(s)
- Dana Duricova
- Public Health, Epidemiology and Economic Health, Register Epimad, Maison Régionale de la Recherche Clinique, Lille University and Hospital, Lille, France.,IBD clinical and research centre, ISCARE, Prague, Czech Republic
| | - Hélène Sarter
- Public Health, Epidemiology and Economic Health, Register Epimad, Maison Régionale de la Recherche Clinique, Lille University and Hospital, Lille, France.,Lille Inflammation Research International Centre LIRIC - UMR 995 Inserm Lille 2 University, CHRU de Lille, Lille, France
| | - Guillaume Savoye
- Gastroenterology Unit, Epimad Registry, Hôpital Charles Nicolle, Rouen University Hospital, Rouen, France
| | - Ariane Leroyer
- Public Health, Epidemiology and Economic Health, Register Epimad, Maison Régionale de la Recherche Clinique, Lille University and Hospital, Lille, France
| | - Benjamin Pariente
- Gastroenterology Unit, Hôpital Huriez, Lille University Hospital, Lille, France
| | - Laura Armengol-Debeir
- Gastroenterology Unit, Epimad Registry, Hôpital Charles Nicolle, Rouen University Hospital, Rouen, France
| | | | - Delphine Ley
- Lille Inflammation Research International Centre LIRIC - UMR 995 Inserm Lille 2 University, CHRU de Lille, Lille, France.,Division of Gastroenterology, Hepatology and Nutrition, Department of Pediatrics, Lille University Jeanne de Flandre Children's Hospital, University of Lille, Lille, France
| | - Dominique Turck
- Lille Inflammation Research International Centre LIRIC - UMR 995 Inserm Lille 2 University, CHRU de Lille, Lille, France.,Division of Gastroenterology, Hepatology and Nutrition, Department of Pediatrics, Lille University Jeanne de Flandre Children's Hospital, University of Lille, Lille, France
| | - Carole Templier
- Dermatology Unit, Hôpital Huriez, Lille University Hospital, Lille, France
| | - Sebastien Buche
- Dermatology Unit, Hôpital Huriez, Lille University Hospital, Lille, France
| | | | - Corinne Gower-Rousseau
- Public Health, Epidemiology and Economic Health, Register Epimad, Maison Régionale de la Recherche Clinique, Lille University and Hospital, Lille, France.,Lille Inflammation Research International Centre LIRIC - UMR 995 Inserm Lille 2 University, CHRU de Lille, Lille, France
| | - Mathurin Fumery
- Lille Inflammation Research International Centre LIRIC - UMR 995 Inserm Lille 2 University, CHRU de Lille, Lille, France.,Gastroenterology Unit, Epimad Registry, CHU Amiens Sud, l, Amiens University Hospital, Amiens, France
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Everhov ÅH, Sachs MC, Malmborg P, Nordenvall C, Myrelid P, Khalili H, Elmberg M, Ekbom A, Askling J, Jakobsson G, Halfvarson J, Ludvigsson JF, Olén O. Changes in inflammatory bowel disease subtype during follow-up and over time in 44,302 patients. Scand J Gastroenterol 2019; 54:55-63. [PMID: 30700170 DOI: 10.1080/00365521.2018.1564361] [Citation(s) in RCA: 45] [Impact Index Per Article: 7.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/08/2023]
Abstract
AIM To investigate inflammatory bowel disease (IBD) register-based subtype classifications over a patient's disease course and over time. METHODS We examined International Classification of Diseases coding in patients with ≥2 IBD diagnostic listings in the National Patient Register 2002-2014 (n = 44,302). RESULTS 18% of the patients changed diagnosis (17% of adults, 29% of children) during a median follow-up of 3.8 years. Of visits with diagnoses of Crohn's disease (CD) or ulcerative colitis (UC), 97% were followed by the same diagnosis, whereas 67% of visits with diagnosis IBD-unclassified (IBD-U) were followed by another IBD-U diagnosis. Patients with any diagnostic change changed mostly once (47%) or twice (31%), 39% from UC to CD, 33% from CD to UC and 30% to or from IBD-U. Using a classification algorithm based on the first two diagnoses ('incident classification'), suited for prospective cohort studies, the proportion adult patients with CD, UC, and IBD-U 2002-2014 were 29%, 62%, and 10% (43%, 45%, and 12% in children). A classification model incorporating additional information from surgeries and giving weight to the last 5 years of visits ('prevalent classification'), suited for description of a study population at end of follow-up, classified 31% of adult cases as CD, 58% as UC and 11% as IBD-U (44%, 38%, and 18% in children). CONCLUSIONS IBD subtype changed in 18% during follow-up. The proportion with CD increased and UC decreased from definition at start to end of follow-up. IBD-U was more common in children.
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Affiliation(s)
- Åsa H Everhov
- a Department of Clinical Science and Education , Södersjukhuset, Karolinska Institutet , Stockholm , Sweden
- b Clinical Epidemiology Unit, Department of Medicine Solna , Karolinska Institutet , Stockholm , Sweden
| | - Michael C Sachs
- c Institute of Environmental Medicine, Karolinska Institutet , Stockholm , Sweden
| | - Petter Malmborg
- a Department of Clinical Science and Education , Södersjukhuset, Karolinska Institutet , Stockholm , Sweden
- b Clinical Epidemiology Unit, Department of Medicine Solna , Karolinska Institutet , Stockholm , Sweden
| | - Caroline Nordenvall
- d Department of Molecular Medicine and Surgery , Karolinska Institutet , Stockholm , Sweden
- e Center for Digestive Disease, Div. of Coloproctology , Karolinska University Hospital , Stockholm , Sweden
| | - Pär Myrelid
- f Division of Surgery, Department of Clinical and Experimental Medicine , Faulty of Health Sciences , Linköping, Sweden
- g University and Department of Surgery , County Council of Östergötland Linköping , Linköping, Sweden
| | - Hamed Khalili
- b Clinical Epidemiology Unit, Department of Medicine Solna , Karolinska Institutet , Stockholm , Sweden
- h Gastroenterology Unit, Crohn's and Colitis Center, Massachusetts General Hospital, Harvard Medical School , Boston , MA , USA
| | - Maria Elmberg
- b Clinical Epidemiology Unit, Department of Medicine Solna , Karolinska Institutet , Stockholm , Sweden
| | - Anders Ekbom
- b Clinical Epidemiology Unit, Department of Medicine Solna , Karolinska Institutet , Stockholm , Sweden
| | - Johan Askling
- b Clinical Epidemiology Unit, Department of Medicine Solna , Karolinska Institutet , Stockholm , Sweden
| | - Gustav Jakobsson
- i Department Medical Epidemiology and Biostatistics , Karolinska Institutet , Stockholm , Sweden
| | - Jonas Halfvarson
- j Department of Gastroenterology, Faculty of Medicine and Health , Örebro University , Örebro , Sweden
| | - Jonas F Ludvigsson
- i Department Medical Epidemiology and Biostatistics , Karolinska Institutet , Stockholm , Sweden
- j Department of Gastroenterology, Faculty of Medicine and Health , Örebro University , Örebro , Sweden
- k Department of Pediatrics , Örebro University Hospital, Örebro University , Örebro , Sweden
- l Division of Epidemiology and Public Health , School of Medicine, University of Nottingham , Nottingham , UK
- m Department of Medicine , Columbia University College of Physicians and Surgeons , New York , NY , USA
| | - Ola Olén
- a Department of Clinical Science and Education , Södersjukhuset, Karolinska Institutet , Stockholm , Sweden
- b Clinical Epidemiology Unit, Department of Medicine Solna , Karolinska Institutet , Stockholm , Sweden
- n Department of pediatric gastroenterology and nutrition , Sachs' Children and Youth Hospital , Stockholm , Sweden
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Kim J, Ye BD. Successful Transition from Pediatric to Adult Care in Inflammatory Bowel Disease: What is the Key? Pediatr Gastroenterol Hepatol Nutr 2019; 22:28-40. [PMID: 30671371 PMCID: PMC6333582 DOI: 10.5223/pghn.2019.22.1.28] [Citation(s) in RCA: 16] [Impact Index Per Article: 2.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/23/2018] [Revised: 11/27/2018] [Accepted: 11/28/2018] [Indexed: 12/13/2022] Open
Abstract
The incidence of pediatric-onset inflammatory bowel disease (IBD) is on the rise, accounting for up to 25% of IBD cases. Pediatric IBD often has extensive bowel involvement with aggressive and rapidly progressing behavior compared to adult IBD. Because IBD has a high morbidity rate and can have a lifelong impact, successful transition from pediatric to adult care is important to maintain the continuity of care. Furthermore, successful transition facilitates appropriate development and psychosocial well-being among patients, as well as comprehensive and harmonious healthcare delivery amongst stakeholders. However, there are various obstacles related to patients, family, providers, and organizations that interfere with successful transition. Successful transition requires a flexible and tailored plan that is made according to the patient's developmental abilities and situation. This plan should be established through periodic interviews with the patient and family and through close collaboration with other care providers. Through a stepwise approach to the transition process, patients' knowledge and self-management skills can be improved. After preparation for the transition is completed and the obstacles are overcome, patients can be gradually moved to adult care. Finally, successful transition can increase patients' adherence to therapy, maintain the appropriate health status, improve patients' self-management, and promote self-reliance among patients.
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Affiliation(s)
- Jeongseok Kim
- Department of Gastroenterology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Byong Duk Ye
- Department of Gastroenterology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea.,Inflammatory Bowel Disease Center, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
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Abstract
OBJECTIVES We examined the fecal virome and bacterial community composition of children with Crohn disease (CD), ulcerative colitis (UC), and healthy controls to test the hypothesis that unique patterns of viral organisms and/or presence of bacterial pathogens may be identified that could contribute to the pathogenesis of pediatric inflammatory bowel disease (IBD). METHODS Fecal samples from 24 children (mean 12.2 years) with CD (n = 7) or UC (n = 5) and similar aged controls (n = 12) were processed to determine individual viromes. Viral sequences were identified through translated protein sequence similarity search. Bacterial microbiota were determined by sequencing of the V4 region of the 16S rRNA gene. RESULTS Only a few human viruses were detected, so virome analyses focused on bacterial viruses. The relative abundance of Caudovirales was greater than that of Microviridae phages in both IBD and healthy controls. Caudovirales phages were more abundant in CD (mean 80.8%) than UC (48.8%) (P = 0.05) but not controls. The richness of viral strains in Microviridae but not Caudovirales was higher in controls than CD (P = 0.05) but not UC cases. No other measure of phage abundance, richness, or Shannon diversity showed significant difference between the 2 IBD and control groups. Bacterial microbiota analysis revealed that IBD diagnosis, albumin, hemoglobin, erythrocyte sedimentation rate, and probiotic supplementation correlated to the composition of gut bacterial microbiota. CONCLUSIONS Minor patterns in gut virome and bacterial community composition distinguish pediatric IBD patients from healthy controls. Probiotics are associated with bacterial microbiota composition. These exploratory results need confirmation in larger studies.
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Kim J, Ye BD. Successful transition from pediatric to adult care in inflammatory bowel disease: what is the key? Intest Res 2018; 17:24-35. [PMID: 30419636 PMCID: PMC6361010 DOI: 10.5217/ir.2018.00128] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/17/2018] [Accepted: 10/07/2018] [Indexed: 12/13/2022] Open
Abstract
The incidence of pediatric-onset inflammatory bowel disease (IBD) is on the rise, accounting for up to 25% of IBD cases. Pediatric IBD often has extensive bowel involvement with aggressive and rapidly progressing behavior compared to adult IBD. Because IBD has a high morbidity rate and can have a lifelong impact, successful transition from pediatric to adult care is important to maintain the continuity of care. Furthermore, successful transition facilitates appropriate development and psychosocial well-being among patients, as well as comprehensive and harmonious healthcare delivery amongst stakeholders. However, there are various obstacles related to patients, family, providers, and organizations that interfere with successful transition. Successful transition requires a flexible and tailored plan that is made according to the patient’s developmental abilities and situation. This plan should be established through periodic interviews with the patient and family and through close collaboration with other care providers. Through a stepwise approach to the transition process, patients’ knowledge and self-management skills can be improved. After preparation for the transition is completed and the obstacles are overcome, patients can be gradually moved to adult care. Finally, successful transition can increase patients’ adherence to therapy, maintain the appropriate health status, improve patients’ self-management, and promote self-reliance among patients.
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Affiliation(s)
- Jeongseok Kim
- Department of Gastroenterology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Byong Duk Ye
- Department of Gastroenterology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea.,Department of Inflammatory Bowel Disease Center, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
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37
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Mikocka-Walus A, Knowles SR. Editorial: anxiety and depression in inflammatory bowel disease. Aliment Pharmacol Ther 2018; 48:686-687. [PMID: 30132938 DOI: 10.1111/apt.14912] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/28/2022]
Affiliation(s)
- A Mikocka-Walus
- School of Psychology, Deakin University Geelong, Melbourne, Vic., Australia
| | - S R Knowles
- Faculty of Health, Arts and Design, Swinburne University of Technology, Melbourne, Vic., Australia
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Sýkora J, Pomahačová R, Kreslová M, Cvalínová D, Štych P, Schwarz J. Current global trends in the incidence of pediatric-onset inflammatory bowel disease. World J Gastroenterol 2018; 24:2741-2763. [PMID: 29991879 PMCID: PMC6034144 DOI: 10.3748/wjg.v24.i25.2741] [Citation(s) in RCA: 253] [Impact Index Per Article: 36.1] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/29/2018] [Revised: 06/01/2018] [Accepted: 06/25/2018] [Indexed: 02/06/2023] Open
Abstract
AIM To perform a comprehensive review and provide an up-to-date synopsis of the incidence and trends of inflammatory bowel disease (IBD). METHODS We systematically searched the MEDLINE (source PubMed), EMBASE and Cochrane Library databases in accordance with the Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines (period: 1985-2018) to identify studies reporting population-based data on the incidence of pediatric-onset (< 19 years at diagnosis) IBD in full manuscripts. Two authors carried out screening and data extraction. Choropleth interactive maps and temporal trends were used to illustrate the international differences and incidences of and changes in IBD and subtypes. RESULTS In total, one hundred forty studies reporting data from 38 countries were considered in this review. The highest annual pediatric incidences of IBD were 23/100000 person-years in Europe, 15.2/100000 in North America, and 11.4/100000 in Asia/the Middle East and Oceania. The highest annual incidences of Crohn's disease (CD) were 13.9/100000 in North America and 12.3/100000 in Europe. The highest annual incidences of ulcerative colitis (UC) were 15.0/100000 in Europe and 10.6/100000 in North America. The highest annual incidences of IBD-unclassified (IBD-U) were 3.6/100000 in Europe and 2.1/100000 in North America. In the time-trend analyses, 67% of CD, 46% of UC and 11% of IBD-U studies reported an increasing incidence (P < 0.05). The risk of IBD is increasing among first-generation of migrant populations. CONCLUSION Globally, the incidence of IBD varies greatly by geographical areas. The steadily increasing incidence of pediatric IBD over time indicates its emergence as a global disease, suggesting that studies should investigate the environmental risk factors among pediatric cohorts.
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Affiliation(s)
- Josef Sýkora
- Division of Gastroenterology, Hepatology and Nutrition, Department of Paediatrics, Charles University in Prague, Faculty of Medicine in Pilsen, Faculty Hospital, Pilsen 304 60, Czech Republic
| | - Renáta Pomahačová
- Division of Gastroenterology, Hepatology and Nutrition, Department of Paediatrics, Charles University in Prague, Faculty of Medicine in Pilsen, Faculty Hospital, Pilsen 304 60, Czech Republic
| | - Marcela Kreslová
- Division of Gastroenterology, Hepatology and Nutrition, Department of Paediatrics, Charles University in Prague, Faculty of Medicine in Pilsen, Faculty Hospital, Pilsen 304 60, Czech Republic
| | - Dominika Cvalínová
- Division of Gastroenterology, Hepatology and Nutrition, Department of Paediatrics, Charles University in Prague, Faculty of Medicine in Pilsen, Faculty Hospital, Pilsen 304 60, Czech Republic
| | - Přemysl Štych
- Department of Applied Geoinformatics and Cartography, Charles University in Prague, Faculty of Science, Prague 128 43, Czech Republic
| | - Jan Schwarz
- Division of Gastroenterology, Hepatology and Nutrition, Department of Paediatrics, Charles University in Prague, Faculty of Medicine in Pilsen, Faculty Hospital, Pilsen 304 60, Czech Republic
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Sýkora J, Pomahačová R, Kreslová M, Cvalínová D, Štych P, Schwarz J. Current global trends in the incidence of pediatric-onset inflammatory bowel disease. World J Gastroenterol 2018. [PMID: 29991879 DOI: 10.3748/wjg.v24.i25.2741.pmid:29991879;pmcid:pmc6034144] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 05/14/2023] Open
Abstract
AIM To perform a comprehensive review and provide an up-to-date synopsis of the incidence and trends of inflammatory bowel disease (IBD). METHODS We systematically searched the MEDLINE (source PubMed), EMBASE and Cochrane Library databases in accordance with the Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines (period: 1985-2018) to identify studies reporting population-based data on the incidence of pediatric-onset (< 19 years at diagnosis) IBD in full manuscripts. Two authors carried out screening and data extraction. Choropleth interactive maps and temporal trends were used to illustrate the international differences and incidences of and changes in IBD and subtypes. RESULTS In total, one hundred forty studies reporting data from 38 countries were considered in this review. The highest annual pediatric incidences of IBD were 23/100000 person-years in Europe, 15.2/100000 in North America, and 11.4/100000 in Asia/the Middle East and Oceania. The highest annual incidences of Crohn's disease (CD) were 13.9/100000 in North America and 12.3/100000 in Europe. The highest annual incidences of ulcerative colitis (UC) were 15.0/100000 in Europe and 10.6/100000 in North America. The highest annual incidences of IBD-unclassified (IBD-U) were 3.6/100000 in Europe and 2.1/100000 in North America. In the time-trend analyses, 67% of CD, 46% of UC and 11% of IBD-U studies reported an increasing incidence (P < 0.05). The risk of IBD is increasing among first-generation of migrant populations. CONCLUSION Globally, the incidence of IBD varies greatly by geographical areas. The steadily increasing incidence of pediatric IBD over time indicates its emergence as a global disease, suggesting that studies should investigate the environmental risk factors among pediatric cohorts.
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Affiliation(s)
- Josef Sýkora
- Division of Gastroenterology, Hepatology and Nutrition, Department of Paediatrics, Charles University in Prague, Faculty of Medicine in Pilsen, Faculty Hospital, Pilsen 304 60, Czech Republic
| | - Renáta Pomahačová
- Division of Gastroenterology, Hepatology and Nutrition, Department of Paediatrics, Charles University in Prague, Faculty of Medicine in Pilsen, Faculty Hospital, Pilsen 304 60, Czech Republic
| | - Marcela Kreslová
- Division of Gastroenterology, Hepatology and Nutrition, Department of Paediatrics, Charles University in Prague, Faculty of Medicine in Pilsen, Faculty Hospital, Pilsen 304 60, Czech Republic
| | - Dominika Cvalínová
- Division of Gastroenterology, Hepatology and Nutrition, Department of Paediatrics, Charles University in Prague, Faculty of Medicine in Pilsen, Faculty Hospital, Pilsen 304 60, Czech Republic
| | - Přemysl Štych
- Department of Applied Geoinformatics and Cartography, Charles University in Prague, Faculty of Science, Prague 128 43, Czech Republic
| | - Jan Schwarz
- Division of Gastroenterology, Hepatology and Nutrition, Department of Paediatrics, Charles University in Prague, Faculty of Medicine in Pilsen, Faculty Hospital, Pilsen 304 60, Czech Republic.
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Martinelli M, Giugliano FP, Russo M, Giannetti E, Andreozzi M, Bruzzese D, Perrone L, Staiano A, Miraglia Del Giudice E, Miele E, Marzuillo P, Strisciuglio C. The Changing Face of Pediatric Ulcerative Colitis: A Population-based Cohort Study. J Pediatr Gastroenterol Nutr 2018; 66:903-908. [PMID: 29135823 DOI: 10.1097/mpg.0000000000001833] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/06/2023]
Abstract
OBJECTIVES The aims of this retrospective study were to describe ulcerative colitis (UC) phenotype at diagnosis and follow-up and to identify possible predictors of severe disease course. METHODS This was a retrospective, single-center study. We reviewed the charts of patients with UC diagnosed between 2 and 18 years at our referral center from January 2007 to January 2016. Laboratory and clinical features at diagnosis, such as disease extent, atypical phenotypes, extraintestinal manifestations, and therapies, and pattern changes during the follow-up, including relapse rate, disease extension, and the cumulative risk for colectomy were collected. RESULTS One hundred eleven patients were enrolled. Atypical phenotypes were identified at diagnosis in 55 out of 111 patients (49.5%). Extraintestinal manifestations were detected in 16 out of 111 (14.4%) at the diagnosis. During the follow-up 60 out of 111 (54%) patients needed to start azathioprine, 9 out of 111 (8.1%) patients started biologic therapy and 10 out of 111 (patients underwent surgery, resulting in a cumulative risk of 8% at 5 years and 16% at 10 years. Steroid refractoriness (hazard ratio: 13.9) and starting of biologic therapy (hazard ratio: 25.3) represented the best predictors for surgery. The cumulative probability of first relapse was 47% at 6 months and 63% at 1 year. Disease extension was reported in 21 out of 70 patients (30%). CONCLUSION Pediatric UC is associated with a severe phenotype and a high percentage of atypical features. Surgery rate seems to be decreased from early reports.
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Affiliation(s)
- Massimo Martinelli
- Department of Translational Medical Science, Section of Pediatrics, University of Naples "Federico II"
| | - Francesca P Giugliano
- Department of Translational Medical Science, Section of Pediatrics, University of Naples "Federico II"
| | - Marina Russo
- Department of Translational Medical Science, Section of Pediatrics, University of Naples "Federico II"
| | - Eleonora Giannetti
- Department of Translational Medical Science, Section of Pediatrics, University of Naples "Federico II"
| | - Marialuisa Andreozzi
- Department of Translational Medical Science, Section of Pediatrics, University of Naples "Federico II"
| | | | - Laura Perrone
- Department of Woman, Child and General and Specialized Surgery, Second University of Naples, Naples, Italy
| | - Annamaria Staiano
- Department of Translational Medical Science, Section of Pediatrics, University of Naples "Federico II"
| | | | - Erasmo Miele
- Department of Translational Medical Science, Section of Pediatrics, University of Naples "Federico II"
| | - Pierluigi Marzuillo
- Department of Woman, Child and General and Specialized Surgery, Second University of Naples, Naples, Italy
| | - Caterina Strisciuglio
- Department of Woman, Child and General and Specialized Surgery, Second University of Naples, Naples, Italy
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Alreheili KM, Alsaleem KA, Almehaidib AI. Natural history and outcome of inflammatory bowel diseases in children in Saudi Arabia: A single-center experience. Saudi J Gastroenterol 2018; 24:171-176. [PMID: 29676289 PMCID: PMC5985636 DOI: 10.4103/sjg.sjg_490_17] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/12/2022] Open
Abstract
Background/Aim Inflammatory bowel disease (IBD) is a chronic gastrointestinal disorder which includes ulcerative colitis (UC), Crohn's disease (CD), and indeterminate colitis (IC). The natural history of pediatric IBDs is poorly understood and generally unpredictable. We aim to study the natural history of IBD in Saudi children including the extraintestinal manifestations, changes in diagnosis, disease behavior, medical management, and surgical outcome. Patients and Methods A retrospective review of all the charts of children less than 14 years of age who were diagnosed as IBD and followed up in King Faisal Specialist Hospital and Research Center (KFSH and RC) from January 2001 to December 2011 was performed. Results Sixty-six children were diagnosed with IBD, 36 patients (54.5%) had CD, 27 patients (41%) had UC, and 3 patients (4.5%) had IC. Change in the diagnosis from UC to CD was made in 5 patients (7.6%). Extraintestinal manifestations were documented in 32% of all patients, and the most common was bone involvement (osteopenia/osteoporosis) in 16.7% of the patients. Arthritis (13.6%) was the second most common manifestation. Sclerosing cholangitis was reported in 2.8% in CD compared to 14.8% in UC. At the time of data collection, 8 patients (12%) were off therapy, 38 patients (57.6) were on 5-ASA, 31 patients (47%) were on azathioprine, and 12 patients (18.2%) were receiving anti-TNF. Of the children with CD, 10 patients (27.8%) underwent 1 or more major operations. Of the children with UC, 18.5% underwent 1 or more major intraabdominal procedures. Conclusions Many issues in pediatric IBD can predict the natural history of the disease including growth failure, complications, need for more aggressive medical treatment, and/or surgery. More studies are needed from the region focusing on factors that may affect the natural history and disease progression.
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Affiliation(s)
- Khalid M. Alreheili
- Department of Pediatrics, Division of Gastroenterology, Maternity and Children's Hospital, Madinah, Saudi Arabia
| | - Khalid A. Alsaleem
- Department of Pediatrics, Division of Gastroenterology, King Faisal Specialist Hospital and Research Centre, Riyadh, Saudi Arabia
| | - Ali I. Almehaidib
- Department of Pediatrics, Division of Gastroenterology, King Faisal Specialist Hospital and Research Centre, Riyadh, Saudi Arabia
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Rinawi F, Assa A, Hartman C, Mozer Glassberg Y, Nachmias Friedler V, Rosenbach Y, Silbermintz A, Zevit N, Shamir R. Long-term Extent Change of Pediatric-Onset Ulcerative Colitis. J Clin Gastroenterol 2018; 52:326-332. [PMID: 28067753 DOI: 10.1097/mcg.0000000000000741] [Citation(s) in RCA: 8] [Impact Index Per Article: 1.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 01/08/2023]
Abstract
BACKGROUND Data describing extent change (progression or regression) in pediatric-onset ulcerative colitis (UC) are scarce. GOAL We aimed to describe extent change in pediatric-onset UC during long-term follow-up and to assess predictors of extent change. STUDY Medical charts of pediatric-onset UC patients with at least 5-year follow-up were analyzed retrospectively. Disease extent was determined using the Paris classification. It was examined at diagnosis and during follow-up at different time points. The impact of possible predictors on extent change including age at diagnosis, gender, clinical manifestations, disease, severity indices, and different therapeutic regimens during disease course was assessed. RESULTS Patients (n=134, 55% males) were followed for a median duration of 13.1 (range, 5 to 28) years. Median age at diagnosis was 13.1 (range, 2 to 17.8) years. Of 134 patients, 40.5% had extensive or pancolitis, 33.5% left-sided colitis, and 26% had proctitis at diagnosis. On follow-up (n=117), 45% had unchanged disease extent, 35% experienced extent progression, whereas 20% experienced regression of disease extent. The multivariate Cox models demonstrated that among children with left-sided disease at diagnosis, presence of extraintestinal manifestations (hazard ratio, 5.19; P=0.022), and higher pediatric UC activity index (hazard ratio, 8.77; P=0.008) were associated with extent progression to extensive disease. Predictors of extent regression have not been identified. CONCLUSIONS Disease extent changes significantly over time in pediatric-onset UC. In our cohort, presence of extraintestinal manifestation and higher pediatric UC activity index score at diagnosis were associated with progression from limited to extensive disease during follow-up.
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Affiliation(s)
- Firas Rinawi
- Institute of Gastroenterology, Nutrition and Liver Diseases, Schneider Children's Medical Center of Israel, Petach-Tikva
| | - Amit Assa
- Institute of Gastroenterology, Nutrition and Liver Diseases, Schneider Children's Medical Center of Israel, Petach-Tikva.,Sackler Faculty of Medicine, Tel-Aviv University, Tel Aviv, Israel
| | - Corina Hartman
- Institute of Gastroenterology, Nutrition and Liver Diseases, Schneider Children's Medical Center of Israel, Petach-Tikva.,Sackler Faculty of Medicine, Tel-Aviv University, Tel Aviv, Israel
| | - Yael Mozer Glassberg
- Institute of Gastroenterology, Nutrition and Liver Diseases, Schneider Children's Medical Center of Israel, Petach-Tikva.,Sackler Faculty of Medicine, Tel-Aviv University, Tel Aviv, Israel
| | - Vered Nachmias Friedler
- Institute of Gastroenterology, Nutrition and Liver Diseases, Schneider Children's Medical Center of Israel, Petach-Tikva
| | - Yoram Rosenbach
- Institute of Gastroenterology, Nutrition and Liver Diseases, Schneider Children's Medical Center of Israel, Petach-Tikva
| | - Ari Silbermintz
- Institute of Gastroenterology, Nutrition and Liver Diseases, Schneider Children's Medical Center of Israel, Petach-Tikva
| | - Noam Zevit
- Institute of Gastroenterology, Nutrition and Liver Diseases, Schneider Children's Medical Center of Israel, Petach-Tikva.,Sackler Faculty of Medicine, Tel-Aviv University, Tel Aviv, Israel
| | - Raanan Shamir
- Institute of Gastroenterology, Nutrition and Liver Diseases, Schneider Children's Medical Center of Israel, Petach-Tikva.,Sackler Faculty of Medicine, Tel-Aviv University, Tel Aviv, Israel
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43
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Cabrera JM, Sato TT. Medical and Surgical Management of Pediatric Ulcerative Colitis. Clin Colon Rectal Surg 2018; 31:71-79. [PMID: 29487489 PMCID: PMC5825852 DOI: 10.1055/s-0037-1609021] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/17/2022]
Abstract
Inflammatory bowel disease (IBD) describes a spectrum of idiopathic, lifelong, and progressive intestinal inflammatory conditions that includes Crohn's disease, ulcerative colitis, and indeterminate colitis. A worldwide increase in the incidence of IBD has been observed. In comparison to adults, IBD occurring during childhood and adolescence has several unique clinical characteristics and surgical management issues. In this article, we provide an overview contrasting these important differences.
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Affiliation(s)
- José M. Cabrera
- Division of Pediatric Gastroenterology, Department of Pediatrics, Children's Hospital of Wisconsin, Medical College of Wisconsin, Milwaukee, Wisconsin
| | - Thomas T. Sato
- Division of Pediatric Surgery, Department of Surgery, Children's Hospital of Wisconsin, Medical College of Wisconsin, Children's Corporate Center, Milwaukee, Wisconsin
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44
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Lopez RN, Appleton L, Gearry RB, Day AS. Rising Incidence of Paediatric Inflammatory Bowel Disease in Canterbury, New Zealand, 1996-2015. J Pediatr Gastroenterol Nutr 2018; 66:e45-e50. [PMID: 28727653 DOI: 10.1097/mpg.0000000000001688] [Citation(s) in RCA: 22] [Impact Index Per Article: 3.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/19/2022]
Abstract
BACKGROUND The incidence of paediatric inflammatory bowel disease (IBD) around the world is increasing. Canterbury, New Zealand, has one of the highest Crohn disease (CD) incidence rates published. The present study aimed to document the incidence of paediatric IBD in Canterbury between 1996 and 2015. METHODS All patients diagnosed with IBD in Canterbury, while younger than 16 years, between January 1, 1996 and December 31, 2015 were identified. Demographic and disease phenotypic details were collected and entered into a secure database. Age-specific population data for Canterbury were obtained and annual incidence rates were then calculated. RESULTS The mean annual incidence rate over the 20-year period was 7.18/100,000 (95% confidence interval 5.55-8.81) children. There was a 4-fold increase in the incidence of paediatric IBD in Canterbury between 1996 and 2015. The ratio of CD to ulcerative colitis (UC) diagnosed was 8.4:1. Disease phenotype of CD and UC, based on the Paris classification, were comparable with other studies. CONCLUSIONS The incidence of paediatric IBD in Canterbury has increased dramatically during the last 2 decades. Some of the observed incidence rates are among the highest documented anywhere in the world. The preponderance of CD over UC in the present study is the highest published.
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Affiliation(s)
| | | | - Richard B Gearry
- Department of Medicine, University of Otago, Christchurch, New Zealand
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Nordenvall C, Rosvall O, Bottai M, Everhov ÅH, Malmborg P, Smedby KE, Ekbom A, Askling J, Ludvigsson JF, Myrelid P, Olén O. Surgical Treatment in Childhood-onset Inflammatory Bowel Disease-A Nationwide Register-based Study of 4695 Incident Patients in Sweden 2002-2014. J Crohns Colitis 2018; 12:157-166. [PMID: 29029152 DOI: 10.1093/ecco-jcc/jjx132] [Citation(s) in RCA: 17] [Impact Index Per Article: 2.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/14/2017] [Accepted: 09/19/2017] [Indexed: 12/30/2022]
Abstract
BACKGROUND AND AIMS The incidence of childhood-onset [< 18 years] inflammatory bowel disease [IBD] is increasing worldwide, and some studies suggest that it represents a more severe disease phenotype. Few nationwide, population-based studies have evaluated the surgical burden in patients with childhood-onset IBD, and whether the improved medical treatment has influenced the need for gastrointestinal surgery. The aim was to examine whether the surgical treatment at any age of patients with childhood-onset IBD has changed over time. METHODS In a nationwide cohort study we identified 4695 children [< 18 years] diagnosed with incident IBD in 2002-2014 through the Swedish Patient Register [ulcerative colitis: n = 2295; Crohn's disease: n = 2174; inflammatory bowel disease-unclassified: n = 226]. Abdominal [intestinal resections and colectomies] and perianal surgeries were identified through the Swedish Patient Register. The cumulative incidences of surgeries were calculated using the Kaplan-Meier method. RESULTS In the cohort, 44% were females and 56% males. The median age at inflammatory bowel disease diagnosis was 15 years and the maximum age at end of follow-up was 31 years. The 3-year cumulative incidence of intestinal surgery was 5% in patients with ulcerative colitis and 7% in patients with Crohn's disease, and lower in children aged < 6 years at inflammatory bowel disease diagnosis [3%] than in those aged 15-17 years at diagnosis [7%]. Calendar period of inflammatory bowel disease diagnosis was not associated with risk of surgery. CONCLUSION Over the past 13 years, the risk of surgery in childhood-onset inflammatory bowel disease has remained unchanged.
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Affiliation(s)
- Caroline Nordenvall
- Department of Molecular Medicine and Surgery, Karolinska Institutet, Stockholm, Sweden.,Center for Digestive Disease, Karolinska University Hospital, Stockholm, Sweden
| | - Oda Rosvall
- Department of Clinical and Experimental Medicine, Linköping University, and Department of Surgery, County Council of Östergötland, Linköping, Sweden
| | - Matteo Bottai
- Institute of Environmental Medicine, Karolinska Institutet, Stockholm, Sweden
| | - Åsa H Everhov
- Department of Clinical Science and Education Södersjukhuset, Karolinska Institutet, Stockholm, Sweden.,Department of Medicine Solna, Karolinska Institutet, Stockholm, Sweden
| | - Petter Malmborg
- Sachs' Children and Youth Hospital, Stockholm South General Hospital, Stockholm, Sweden.,Department of Women's and Children's Health, Karolinska Institutet, Stockholm, Sweden
| | - Karin E Smedby
- Department of Medicine Solna, Karolinska Institutet, Stockholm, Sweden
| | - Anders Ekbom
- Department of Medicine Solna, Karolinska Institutet, Stockholm, Sweden
| | - Johan Askling
- Department of Medicine Solna, Karolinska Institutet, Stockholm, Sweden
| | - Jonas F Ludvigsson
- Department of Medical Epidemiology and Biostatistics, Karolinska Institutet, Stockholm, Sweden.,Department of Pediatrics, Örebro University Hospital, Örebro, Sweden.,Division of Epidemiology and Public Health, University of Nottingham, Nottingham, UK.,Department of Medicine, Columbia University College of Physicians and Surgeons, New York, NY, USA
| | - Pär Myrelid
- Department of Clinical and Experimental Medicine, Linköping University, and Department of Surgery, County Council of Östergötland, Linköping, Sweden
| | - Ola Olén
- Department of Clinical Science and Education Södersjukhuset, Karolinska Institutet, Stockholm, Sweden.,Department of Medicine Solna, Karolinska Institutet, Stockholm, Sweden.,Sachs' Children and Youth Hospital, Stockholm South General Hospital, Stockholm, Sweden
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Brooks AJ, Smith PJ, Lindsay JO. Monitoring adolescents and young people with inflammatory bowel disease during transition to adult healthcare. Frontline Gastroenterol 2018; 9:37-44. [PMID: 29484159 PMCID: PMC5824770 DOI: 10.1136/flgastro-2016-100747] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/28/2016] [Revised: 12/13/2016] [Accepted: 12/15/2016] [Indexed: 02/04/2023] Open
Abstract
The transition of adolescents and young people (AYP) with inflammatory bowel disease (IBD) from paediatric to adult healthcare requires coordination between healthcare care providers to achieve optimum outcomes. Transition into adulthood is a time of major challenges physically, developmentally, emotionally and psychosocially for AYP living with IBD. Healthcare professionals must monitor the AYP progress proactively with attention to each of these parameters throughout the transition period to ensure that milestones are attained, and skills for self-management are formed. Thus, achieving the desired goals in both clinical and pastoral areas requires intensive monitoring from a multidisciplinary team across healthcare providers.
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Affiliation(s)
- Alenka J Brooks
- Academic Department of Gastroenterology, Sheffield Teaching Hospitals NHS Foundation Trust, Royal Hallamshire Hospital, Sheffield, UK
| | - Philip J Smith
- Centre for Gastroenterology and Hepatology, Royal Free Hospital, London, UK
| | - James O Lindsay
- Bart's Health NHS Trust, The Royal London Hospital, London, UK
- Centre for Immunology and Infectious Disease, Blizard Institute, Barts and the London School of Medicine, Queen Mary University of London, London, UK
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47
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Protásio BKPF, Barbosa CMPMM, Neufeld CB, Buck LD, Laund LDSL, Toporovski MS, Visoni TC. Specificities of presentation of Crohn's disease in childhood. EINSTEIN-SAO PAULO 2017; 16:eRC4070. [PMID: 29236792 PMCID: PMC5968813 DOI: 10.1590/s1679-45082017rc4070] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/03/2017] [Accepted: 07/26/2017] [Indexed: 11/22/2022] Open
Abstract
The incidence of inflammatory bowel disease in the pediatric population has increased in the last years. The most common form of inflammatory bowel disease is Crohn's disease and, according to its form and age of presentation, it is possible to predict the evolution of the disease.
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48
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Jung D, Lee S, Jeong I, Oh SH, Kim KM. Short-Term Outcome of Infliximab Therapy in Pediatric Crohn's Disease: A Single-Center Experience. Pediatr Gastroenterol Hepatol Nutr 2017; 20:236-243. [PMID: 29302505 PMCID: PMC5750378 DOI: 10.5223/pghn.2017.20.4.236] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/09/2017] [Revised: 10/01/2017] [Accepted: 10/24/2017] [Indexed: 02/06/2023] Open
Abstract
PURPOSE Studies on the efficacy of infliximab (IFX) in a large population of pediatric patients with Crohn's disease (CD) are limited, and prognostic factors are not well-known. The aim of this study was to evaluate outcomes of IFX in pediatric patients with CD and to identify factors associated with poor prognosis. METHODS We retrospectively analyzed medical data of 594 pediatric patients with CD between 1987 and 2013 in a tertiary center. Of these, 156 children treated with IFX were enrolled and were followed up for at least a year with intact data. Outcomes of induction and maintenance, classified as failure or clinical response, were evaluated on the tenth and 54th week of IFX therapy. RESULTS We treated 156 pediatric patients with CD with IFX, and the median duration of IFX therapy was 47 months. For IFX induction therapy, 134 (85.9%) patients experienced clinical response on the 10th week. Among the 134 patients who showed response to induction, 111 (82.8%) patients maintained the clinical response on the 54th week. In multivariate analysis, low hematocrit (p=0.046) at the time of IFX initiation was associated with the failure of IFX induction. For IFX maintenance therapy, longer duration from the initial diagnosis to IFX therapy (p=0.017) was associated with maintenance failure on the 54th week. CONCLUSION We have shown the acceptable outcomes of IFX in a large cohort of pediatric CD patients in Korea. Hematocrit and early introduction of IFX may be prognostic factors for the outcomes of IFX.
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Affiliation(s)
- Dai Jung
- Department of Pediatrics, Asan Medical Center Children's Hospital, University of Ulsan College of Medicine, Seoul, Korea
| | - Sunghee Lee
- Department of Pediatrics, Asan Medical Center Children's Hospital, University of Ulsan College of Medicine, Seoul, Korea
| | - Insook Jeong
- Department of Pediatrics, Asan Medical Center Children's Hospital, University of Ulsan College of Medicine, Seoul, Korea
| | - Seak Hee Oh
- Department of Pediatrics, Asan Medical Center Children's Hospital, University of Ulsan College of Medicine, Seoul, Korea
| | - Kyung Mo Kim
- Department of Pediatrics, Asan Medical Center Children's Hospital, University of Ulsan College of Medicine, Seoul, Korea
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49
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Yu YR, Rodriguez JR. Clinical presentation of Crohn's, ulcerative colitis, and indeterminate colitis: Symptoms, extraintestinal manifestations, and disease phenotypes. Semin Pediatr Surg 2017; 26:349-355. [PMID: 29126502 DOI: 10.1053/j.sempedsurg.2017.10.003] [Citation(s) in RCA: 222] [Impact Index Per Article: 27.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/14/2022]
Abstract
The incidence of inflammatory bowel disease (IBD) is rising with 25% of IBD diagnosed in children under 18 years of age. The clinical presentation of IBD in children is often vague leading to initial misdiagnosis as infectious colitis or irritable bowel syndrome. When IBD is identified, overlap in histologic and endoscopic features may lead to difficulty distinguishing Crohn's disease from ulcerative colitis, resulting in a higher frequency of the diagnosis indeterminate colitis or IBD unspecified. Recognizing the common and the atypical presentation of pediatric IBD and extraintestinal manifestations will aid in expeditious referral and early diagnosis. Activity severity scoring tools and more specific classification systems for pediatric IBD direct therapeutic algorithms and allow for improved longitudinal assessment since disease severity and location have been shown to be associated with outcome.
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Affiliation(s)
- Yangyang R Yu
- Michael E. DeBakey Department of Surgery, Texas Children's Hospital, Houston, Texas
| | - J Ruben Rodriguez
- Michael E. DeBakey Department of Surgery, Texas Children's Hospital, Houston, Texas.
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50
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Duricova D, Leroyer A, Savoye G, Sarter H, Pariente B, Aoucheta D, Armengol-Debeir L, Ley D, Turck D, Peyrin-Biroulet L, Gower-Rousseau C, Fumery M, Antonietti M, Aouakli A, Armand A, Aroichane I, Assi F, Aubet JP, Auxenfants E, Ayafi-Ramelot F, Bankovski D, Barbry B, Bardoux N, Baron P, Baudet A, Bazin B, Bebahani A, Becqwort JP, Benet V, Benali H, Benguigui C, Ben Soussan E, Bental A, Berkelmans I, Bernet J, Bernou K, Bernou-Dron C, Bertot P, Bertiaux-Vandaële N, Bertrand V, Billoud E, Biron N, Bismuth B, Bleuet M, Blondel F, Blondin V, Bohon P, Boniface E, Bonnière P, Bonvarlet E, Bonvarlet P, Boruchowicz A, Bostvironnois R, Boualit M, Bouche B, Boudaillez C, Bourgeaux C, Bourgeois M, Bourguet A, Bourienne A, Branche J, Bray G, Brazier F, Breban P, Brihier H, Brung-Lefebvre V, Bulois P, Burgiere P, Butel J, Canva JY, Canva-Delcambre V, Capron JP, Cardot F, Carpentier P, Cartier E, Cassar JF, Cassagnou M, Castex JF, Catala P, Cattan S, Catteau S, Caujolle B, Cayron G, Chandelier C, Chantre M, Charles J, Charneau T, Chavance-Thelu M, Chirita D, Choteau A, Claerbout JF, Clergue PY, Coevoet H, Cohen G, Collet R, Colombel JF, Coopman S, Corvisart J, Cortot A, Couttenier F, et alDuricova D, Leroyer A, Savoye G, Sarter H, Pariente B, Aoucheta D, Armengol-Debeir L, Ley D, Turck D, Peyrin-Biroulet L, Gower-Rousseau C, Fumery M, Antonietti M, Aouakli A, Armand A, Aroichane I, Assi F, Aubet JP, Auxenfants E, Ayafi-Ramelot F, Bankovski D, Barbry B, Bardoux N, Baron P, Baudet A, Bazin B, Bebahani A, Becqwort JP, Benet V, Benali H, Benguigui C, Ben Soussan E, Bental A, Berkelmans I, Bernet J, Bernou K, Bernou-Dron C, Bertot P, Bertiaux-Vandaële N, Bertrand V, Billoud E, Biron N, Bismuth B, Bleuet M, Blondel F, Blondin V, Bohon P, Boniface E, Bonnière P, Bonvarlet E, Bonvarlet P, Boruchowicz A, Bostvironnois R, Boualit M, Bouche B, Boudaillez C, Bourgeaux C, Bourgeois M, Bourguet A, Bourienne A, Branche J, Bray G, Brazier F, Breban P, Brihier H, Brung-Lefebvre V, Bulois P, Burgiere P, Butel J, Canva JY, Canva-Delcambre V, Capron JP, Cardot F, Carpentier P, Cartier E, Cassar JF, Cassagnou M, Castex JF, Catala P, Cattan S, Catteau S, Caujolle B, Cayron G, Chandelier C, Chantre M, Charles J, Charneau T, Chavance-Thelu M, Chirita D, Choteau A, Claerbout JF, Clergue PY, Coevoet H, Cohen G, Collet R, Colombel JF, Coopman S, Corvisart J, Cortot A, Couttenier F, Crinquette JF, Crombe V, Dadamessi I, Dapvril V, Davion T, Dautreme S, Debas J, Degrave N, Dehont F, Delatre C, Delcenserie R, Delette O, Delgrange T, Delhoustal L, Delmotte JS, Demmane S, Deregnaucourt G, Descombes P, Desechalliers JP, Desmet P, Desreumaux P, Desseaux G, Desurmont P, Devienne A, Devouge E, Devred M, Devroux A, Dewailly A, Dharancy S, Di Fiore A, Djeddi D, Djedir R, Dreher-Duwat ML, Dubois R, Dubuque C, Ducatillon P, Duclay J, Ducrocq B, Ducrot F, Ducrotté P, Dufilho A, Duhamel C, Dujardin D, Dumant-Forest C, Dupas JL, Dupont F, Duranton Y, Duriez A, El Achkar K, El Farisi M, Elie C, Elie-Legrand MC, Elkhaki A, Eoche M, Evrard D, Evrard JP, Fatome A, Filoche B, Finet L, Flahaut M, Flamme C, Foissey D, Fournier P, Foutrein-Comes MC, Foutrein P, Fremond D, Frere T, Fumery M, Gallet P, Gamblin C, Ganga-Zandzou S, Gerard R, Geslin G, Gheyssens Y, Ghossini N, Ghrib S, Gilbert T, Gillet B, Godard D, Godard P, Godchaux JM, Godchaux R, Goegebeur G, Goria O, Gottrand F, Gower P, Grandmaison B, Groux M, Guedon C, Guillard JF, Guillem L, Guillemot F, Guimber D, Haddouche B, Hakim S, Hanon D, Hautefeuille V, Heckestweiller P, Hecquet G, Hedde JP, Hellal H, Henneresse PE, Heyman B, Heraud M, Herve S, Hochain P, Houssin-Bailly L, Houcke P, Huguenin B, Iobagiu S, Ivanovic A, Iwanicki-Caron I, Janicki E, Jarry M, Jeu J, Joly JP, Jonas C, Katherin F, Kerleveo A, Khachfe A, Kiriakos A, Kiriakos J, Klein O, Kohut M, Kornhauser R, Koutsomanis D, Laberenne JE, Laffineur G, Lagarde M, Lannoy P, Lapchin J, Lapprand M, Laude D, Leblanc R, Lecieux P, Leclerc N, Le Couteulx C, Ledent J, Lefebvre J, Lefiliatre P, Legrand C, Le Grix A, Lelong P, Leluyer B, Lenaerts C, Lepileur L, Leplat A, Lepoutre-Dujardin E, Leroi H, Leroy MY, Lesage JP, Lesage X, Lesage J, Lescanne-Darchis I, Lescut J, Lescut D, Leurent B, Levy P, Lhermie M, Lion A, Lisambert B, Loire F, Louf S, Louvet A, Luciani M, Lucidarme D, Lugand J, Macaigne O, Maetz D, Maillard D, Mancheron H, Manolache O, Marks-Brunel AB, Marti R, Martin F, Martin G, Marzloff E, Mathurin P, Mauillon J, Maunoury V, Maupas JL, Mesnard B, Metayer P, Methari L, Meurisse B, Meurisse F, Michaud L, Mirmaran X, Modaine P, Monthe A, Morel L, Mortier PE, Moulin E, Mouterde O, Mudry J, Nachury M, N’Guyen Khac E, Notteghem B, Ollevier V, Ostyn A, Ouraghi A, Ouvry D, Paillot B, Panien-Claudot N, Paoletti C, Papazian A, Parent B, Pariente B, Paris JC, Patrier P, Paupart L, Pauwels B, Pauwels M, Petit R, Piat M, Piotte S, Plane C, Plouvier B, Pollet E, Pommelet P, Pop D, Pordes C, Pouchain G, Prades P, Prevost A, Prevost JC, Quesnel B, Queuniet AM, Quinton JF, Rabache A, Rabelle P, Raclot G, Ratajczyk S, Rault D, Razemon V, Reix N, Revillon M, Richez C, Robinson P, Rodriguez J, Roger J, Roux JM, Rudelli A, Saber A, Savoye G, Schlosseberg P, Segrestin M, Seguy D, Serin M, Seryer A, Sevenet F, Shekh N, Silvie J, Simon V, Spyckerelle C, Talbodec N, Techy A, Thelu JL, Thevenin A, Thiebault H, Thomas J, Thorel JM, Tielman G, Tode M, Toisin J, Tonnel J, Touchais JY, Touze Y, Tranvouez JL, Triplet C, Turck D, Uhlen S, Vaillant E, Valmage C, Vanco D, Vandamme H, Vanderbecq E, Vander Eecken E, Vandermolen P, Vandevenne P, Vandeville L, Vandewalle A, Vandewalle C, Vaneslander P, Vanhoove JP, Vanrenterghem A, Varlet P, Vasies I, Verbiese G, Vernier-Massouille G, Vermelle P, Verne C, Vezilier-Cocq P, Vigneron B, Vincendet M, Viot J, Voiment YM, Wacrenier A, Waeghemaecker L, Wallez JY, Wantiez M, Wartel F, Weber J, Willocquet JL, Wizla N, Wolschies E, Zalar A, Zaouri B, Zellweger A, Ziade C. Extra-intestinal Manifestations at Diagnosis in Paediatric- and Elderly-onset Ulcerative Colitis are Associated With a More Severe Disease Outcome: A Population-based Study. J Crohns Colitis 2017; 11:1326-1334. [PMID: 28981648 DOI: 10.1093/ecco-jcc/jjx092] [Show More Authors] [Citation(s) in RCA: 17] [Impact Index Per Article: 2.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/06/2017] [Accepted: 07/05/2017] [Indexed: 02/08/2023]
Abstract
BACKGROUND AND AIMS Data on extra-intestinal manifestations [EIM] and their impact on the disease course of ulcerative colitis [UC] in population-based cohorts are scarce, particularly in paediatric- and elderly-onset UC patients. The aims of this population-based study were to assess: 1] the occurrence of EIM in paediatric- and elderly-onset UC; 2] the factors associated with EIM; and 3] their impact on long-term disease outcome. METHODS Paediatric-onset [< 17 years at diagnosis] and elderly-onset UC patients [> 60 years at diagnosis] from a French prospective population-based registry [EPIMAD] were included. Data on EIM and other clinical factors at diagnosis and at maximal follow-up were collected. RESULTS In all, 158 paediatric- and 470 elderly-onset patients were included [median age at diagnosis 14.5 and 68.8 years, median follow-up 11.2 and 6.2 years, respectively]. EIM occurred in 8.9% of childhood- and 3% of elderly-onset patients at diagnosis and in 16.7% and 2.2% of individuals during follow-up [p < 0.01], respectively. The most frequent EIM was joint involvement [15.8% of paediatric onset and 2.6% of elderly-onset]. Presence of EIM at diagnosis was associated with more severe disease course [need for immunosuppressants or biologic therapy or colectomy] in both paediatric- and elderly-onset UC (hazard ratio [HR] = 2.0, 95% confidence interval [CI]: 1.0-4.2; and HR = 2.8, 0.9-7.9, respectively). Extensive colitis was another independent risk factor in both age groups. CONCLUSIONS Elderly-onset UC patients had lower risk of EIM either at diagnosis or during follow-up than paediatric-onset individuals. EIM at diagnosis predicted more severe disease outcome, including need for immunosuppressive or biologic therapy or surgery, in both paediatric- and elderly-onset UC.
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Affiliation(s)
- Dana Duricova
- Public Health, Epidemiology and Economic Health, Registre EPIMAD, Lille University and Hospital, Lille, France
| | - Ariane Leroyer
- Public Health, Epidemiology and Economic Health, Registre EPIMAD, Lille University and Hospital, Lille, France
| | - Guillaume Savoye
- Gastroenterology Unit, EPIMAD Registry, Rouen University Hospital, Rouen, France
| | - Hélène Sarter
- Public Health, Epidemiology and Economic Health, Registre EPIMAD, Lille University and Hospital, Lille, France.,Lille Inflammation Research International Center LIRIC-UMR 995 Inserm Lille 2 University, Lille, France
| | - Benjamin Pariente
- Gastroenterology Unit, Hôpital Huriez, Lille University Hospital, Lille, France
| | - Djamila Aoucheta
- Associated Medical Director, Immunology, MSD France, Courbevoie cedex, France
| | | | - Delphine Ley
- Lille Inflammation Research International Center LIRIC-UMR 995 Inserm Lille 2 University, Lille, France.,Division of Gastroenterology, Hepatology and Nutrition, Lille University Jeanne de Flandre Children's Hospital, University of Lille, Lille, France
| | - Dominique Turck
- Lille Inflammation Research International Center LIRIC-UMR 995 Inserm Lille 2 University, Lille, France.,Division of Gastroenterology, Hepatology and Nutrition, Lille University Jeanne de Flandre Children's Hospital, University of Lille, Lille, France
| | | | - Corinne Gower-Rousseau
- Public Health, Epidemiology and Economic Health, Registre EPIMAD, Lille University and Hospital, Lille, France.,Lille Inflammation Research International Center LIRIC-UMR 995 Inserm Lille 2 University, Lille, France
| | - Mathurin Fumery
- Lille Inflammation Research International Center LIRIC-UMR 995 Inserm Lille 2 University, Lille, France.,Gastroenterology Unit, EPIMAD Registry, Amiens University Hospital, Amiens, France
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