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Mills KC, Majumder S. What Is the Latest in Autoimmune Pancreatitis. Gastroenterol Clin North Am 2025; 54:245-258. [PMID: 39880531 DOI: 10.1016/j.gtc.2024.08.021] [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: 01/02/2025]
Abstract
Autoimmune pancreatitis (AIP) is a steroid-responsive fibroinflammatory disorder with 2 clinically distinct subtypes known as type 1 autoimmune and type 2 autoimmune pancreatitis. Type 1 AIP is considered the pancreatic manifestation of immunoglobulin G4-related disease, a systemic disease often presenting with other organ involvement. Advances in understanding the unique clinical presentation, imaging findings, histopathology, and clinical course of this relatively uncommon disease have led to international consensus regarding diagnosis and treatment. While corticosteroids remain the mainstay of treatment, several emerging novel therapies have been explored primarily in the context or relapsing and refractory cases.
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Affiliation(s)
- Krystal C Mills
- Division of Gastroenterology and Hepatology, Mayo Clinic, Rochester, MN, USA
| | - Shounak Majumder
- Division of Gastroenterology and Hepatology, Mayo Clinic, Rochester, MN, USA.
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Li M, Liu Y, Wang J, Wang Y, Yang Y, Yang A. Neutrophil extracellular DNA traps activate the TLR9 signaling pathway of pancreatic ductal epithelial cells in patients with type 2 autoimmune pancreatitis. Int Immunopharmacol 2025; 144:113673. [PMID: 39616853 DOI: 10.1016/j.intimp.2024.113673] [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: 09/23/2024] [Revised: 11/05/2024] [Accepted: 11/17/2024] [Indexed: 12/15/2024]
Abstract
The presence of neutrophil infiltration around the pancreatic ducts has been found to be associated with type 2 autoimmune pancreatitis (AIP). However, the functional role and clinical significance of neutrophil migration in the progression of pancreatitis is not fully understood. Here, we found that neutrophil extracellular traps (NETs) are abundant around the pancreatic duct in patients with type 2 AIP. We also observed an increased expression of toll-like receptor 9 (TLR9) in pancreatic ductal epithelial cells (HPDEC) in type 2 AIP patients compared to other pancreatic diseases. TLR9 acts as the DNA component of NETs (NET-DNA) receptor in HPDEC, which senses extracellular DNA and subsequently activates the NF-κB pathway to promote neutrophil recruitment and induce NET formation. In addition, our results indicated that the hydroxychloroquine (HCQ), acting as a TLR9 antagonist, could effectively inhibit the activation of inflammatory pathways, reduce neutrophil migration and block the positive feedback loop. The intervention positions HCQ acts as a potential target drug for the clinical treatment of type 2 AIP.
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Affiliation(s)
- Meizi Li
- Department of Gastroenterology, Peking Union Medical College Hospital (PUMCH), Chinese Academy of Medical Sciences & Peking Union Medical College (CAMS & PUMC), Beijing 100730, China
| | - Yixiao Liu
- Department of Gastroenterology, Peking Union Medical College Hospital (PUMCH), Chinese Academy of Medical Sciences & Peking Union Medical College (CAMS & PUMC), Beijing 100730, China
| | - Junmin Wang
- State Key Laboratory of Common Mechanism Research for Major Diseases, Institute of Basic Medical Sciences, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100005, China
| | - Yuyang Wang
- State Key Laboratory of Common Mechanism Research for Major Diseases, Institute of Basic Medical Sciences, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100005, China
| | - Yingyun Yang
- Department of Gastroenterology, Peking Union Medical College Hospital (PUMCH), Chinese Academy of Medical Sciences & Peking Union Medical College (CAMS & PUMC), Beijing 100730, China.
| | - Aiming Yang
- Department of Gastroenterology, Peking Union Medical College Hospital (PUMCH), Chinese Academy of Medical Sciences & Peking Union Medical College (CAMS & PUMC), Beijing 100730, China.
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Li MZ, Guo T, Feng YL, Zhang SY, Bai XY, Wu X, Xu K, Yang AM. Diabetes mellitus in patients with type 1 autoimmune pancreatitis at diagnosis and after corticosteroid therapy. Hepatobiliary Pancreat Dis Int 2024; 23:393-398. [PMID: 37121837 DOI: 10.1016/j.hbpd.2023.04.006] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/07/2022] [Accepted: 04/14/2023] [Indexed: 05/02/2023]
Abstract
BACKGROUND A high prevalence of diabetes mellitus (DM) coexisting with autoimmune pancreatitis (AIP) is observed. However, evidence on the circumstances under which corticosteroid therapy (CST) for AIP improves or worsens DM is scarce. This study aimed to demonstrate and identify predictors of DM control under the influence of CST. METHODS Patients diagnosed with type 1 AIP were enrolled from a prospectively maintained cohort and were classified into three groups according to the chronology in which AIP and DM were diagnosed: pre-existing DM (pDM), concurrent DM (cDM), and non-DM (nDM). The responses of DM to CST were assessed when corticosteroid was ceased or tapered to a maintenance dose and classified as 'improvement' and 'non-improvement' (including 'no change' and 'exacerbation'). RESULTS Among 101 patients with type 1 AIP, 52 (51.5%) patients were complicated with DM at the time of AIP diagnosis, with 36 patients in the cDM group and 16 patients in the pDM group. The incidences of diffuse pancreatic swelling (72.2%) and pancreatic body/tail involvement (91.7%) were significantly higher in the cDM group than in both the pDM and nDM groups. Of the 52 patients with DM, CST was administered in 48 cases. Multivariate logistic analysis identified that elevated serum gamma-glutamyl transferase (GGT) level at AIP diagnosis [odds ratio (OR) = 0.032, 95% confidence interval (CI): 0.003-0.412, P = 0.008] and pancreatic atrophy after CST (OR = 0.027, 95% CI: 0.003-0.295, P = 0.003) were negatively associated with DM control improvement. CONCLUSIONS Patients with diffuse pancreatic swelling and pancreatic body/tail involvement in pancreatitis tended to be complicated with cDM at AIP diagnosis. CST exerted a beneficial effect on the clinical course of DM in nearly half of the AIP patients complicated with DM at diagnosis, particularly in those without elevated serum GGT levels at diagnosis and who did not experience pancreatic atrophy after CST.
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Affiliation(s)
- Mei-Zi Li
- Department of Gastroenterology, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, Beijing 100730, China
| | - Tao Guo
- Department of Gastroenterology, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, Beijing 100730, China
| | - Yun-Lu Feng
- Department of Gastroenterology, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, Beijing 100730, China
| | - Sheng-Yu Zhang
- Department of Gastroenterology, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, Beijing 100730, China
| | - Xiao-Yin Bai
- Department of Gastroenterology, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, Beijing 100730, China
| | - Xi Wu
- Department of Gastroenterology, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, Beijing 100730, China
| | - Kai Xu
- Department of Radiology, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, Beijing 100730, China
| | - Ai-Ming Yang
- Department of Gastroenterology, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, Beijing 100730, China.
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Datta D, Selvakumar B, Goel AD, Chhibber S, Varshney VK, Kumar R. Diagnostic performance of F-18 FDG PET/CT in differentiating autoimmune pancreatitis from pancreatic cancer: a systemic review and meta-analysis. Ann Nucl Med 2024; 38:619-629. [PMID: 38750330 DOI: 10.1007/s12149-024-01934-4] [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: 01/28/2024] [Accepted: 04/18/2024] [Indexed: 06/14/2024]
Abstract
OBJECTIVES This study aims to evaluate the utility of F-18 FDG PET/CT in the non-invasive diagnosis of autoimmune pancreatitis (AIP) and differentiating it from pancreatic cancer (CaP) based on the amount and pattern of FDG uptake, as well as involvement of extra-pancreatic sites. METHODS A systematic search was conducted using PubMed, Scopus, Cochrane Library and Google Scholar. Only those studies that compared the findings of F-18 FDG PET/CT in terms of SUVmax, pattern of FDG uptake and presence of FDG-avid extra-pancreatic sites in both AIP and CaP were included. Studies were qualitatively assessed for risk of bias and publication bias. The diagnostic performance of parameters on PET/CT was examined through pooled sensitivity, specificity, diagnostic odd's ratio (DOR) and summary receiver operator characteristic (SROC) curve analysis. RESULTS Six studies were included with a total of 580 patients. 178 patients had AIP (Age 18-90 years, male, M: female, F ratio-8.4:1) and 402 patients had CaP (Age 22-88 years, M:F ratio-1.5:1). Type of AIP was reported in only 3 studies, with the included cases predominantly being type 1 AIP. All studies were retrospective with heterogeneity and a risk on patient selection and index test. The FDG uptake, expressed as SUVmax, was lower in AIP with a weighted mean difference of -3.11 (95% confidence interval, CI: -5.28 to -0.94). To diagnose AIP, the pooled sensitivity, specificity and DOR of diffuse pattern of FDG uptake were 0.59 (95% CI: 0.51-0.66), 0.89 (95% CI: 0.86-0.92) and 21.07 (95% CI: 5.07-88.32), respectively, with an area under curve (AUC) of 0.717 on SROC analysis. The pooled sensitivity, specificity and DOR of FDG-avid extra pancreatic sites were 0.55 (95% CI: 0.45-0.65), 0.58 (95% CI: 0.52-0.64) and 2.33 (95% CI: 1.40-3.89), respectively, with an AUC of 0.632. CONCLUSION On F-18 FDG PET/CT, a pancreatic lesion of AIP has a lower SUVmax value than CaP. A diffuse pattern of FDG uptake and presence of an extra-pancreatic FDG-avid site are nearly 21 times and twice more likely in AIP than CaP, respectively.
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Affiliation(s)
- Deepanksha Datta
- Department of Nuclear Medicine, All India Institute of Medical Sciences, Jodhpur, Rajasthan, India
| | - B Selvakumar
- Department of Surgical Gastroenterology, All India Institute of Medical Sciences, Basni Industrial Area Phase 2, Jodhpur, Rajasthan, 342005, India.
| | - Akhil Dhanesh Goel
- Department of Community and Family Medicine, All India Institute of Medical Sciences, Jodhpur, Rajasthan, India
| | | | - Vaibhav Kumar Varshney
- Department of Surgical Gastroenterology, All India Institute of Medical Sciences, Basni Industrial Area Phase 2, Jodhpur, Rajasthan, 342005, India
| | - Rajesh Kumar
- Department of Nuclear Medicine, All India Institute of Medical Sciences, Jodhpur, Rajasthan, India
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Metelli F, Manfredi G, Pagano N, Buscarini E, Crinò SF, Armellini E. The Role of Endoscopic Ultrasound and Ancillary Techniques in the Diagnosis of Autoimmune Pancreatitis: A Comprehensive Review. Diagnostics (Basel) 2024; 14:1233. [PMID: 38928649 PMCID: PMC11202526 DOI: 10.3390/diagnostics14121233] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/09/2024] [Revised: 06/04/2024] [Accepted: 06/07/2024] [Indexed: 06/28/2024] Open
Abstract
Autoimmune pancreatitis (AIP) is a unique form of chronic pancreatitis with a multifactorial pathogenesis. Historically, it has been classified as type 1 and type 2, according to its clinical and histological features. The diagnosis of AIP is challenging and relies on a combination of clinical, histopathologic, serologic, and imaging characteristics. In the available guidelines, the imaging hallmarks of AIP are based on cross-sectional imaging and cholangiopancreatography retrograde endoscopic findings. Endoscopic ultrasound (EUS) is generally used for pancreatic tissue acquisition to rule out pancreatic cancer and diagnose AIP with limited accuracy. Several papers reported the reliability of EUS for providing informative morphologic features of AIP. Nowadays, the improvement in the resolution of EUS conventional images and the development of new ancillary technologies have further increased the diagnostic yield of EUS: contrast-enhanced EUS and EUS elastography are non-invasive and real-time techniques that strongly support the diagnosis and management of pancreatic diseases. In this review article, we will present the role of conventional EUS and ancillary diagnostic techniques in the diagnosis of AIP to support clinicians and endosonographers in managing this condition.
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Affiliation(s)
- Flavio Metelli
- Gastroenterology and Endoscopy Department, ASST Maggiore Hospital Crema, 26013 Crema, Italy; (F.M.); (G.M.); (E.B.)
| | - Guido Manfredi
- Gastroenterology and Endoscopy Department, ASST Maggiore Hospital Crema, 26013 Crema, Italy; (F.M.); (G.M.); (E.B.)
| | - Nico Pagano
- Gastroenterology Unit, Department of Oncological and Specialty Medicine, University Hospital Maggiore della Carità, 28100 Novara, Italy;
| | - Elisabetta Buscarini
- Gastroenterology and Endoscopy Department, ASST Maggiore Hospital Crema, 26013 Crema, Italy; (F.M.); (G.M.); (E.B.)
| | - Stefano Francesco Crinò
- Diagnostic and Interventional Endoscopy of Pancreas, Pancreas Institute, University of Verona, 37134 Verona, Italy;
| | - Elia Armellini
- Gastroenterology and Endoscopy Unit, ASST-Bergamoest, 24068 Seriate, Italy
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Cong P, Yu YN, Wang XM, Zhang YF. Thickness of the Hyperechoic Capsule-like Rim Around Pancreatic Lesions Measured by Ultrasound for Differentiating Between Type 1 Autoimmune Pancreatitis and Pancreatic Adenocarcinoma. IRANIAN JOURNAL OF RADIOLOGY 2023; 20. [DOI: 10.5812/ijradiol-137889] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Subscribe] [Scholar Register] [Received: 05/31/2023] [Revised: 11/15/2023] [Accepted: 11/20/2023] [Indexed: 01/02/2025]
Abstract
Background: Autoimmune pancreatitis (AIP) is often misdiagnosed as pancreatic adenocarcinoma (PAC), resulting in unnecessary surgical interventions. On computed tomography (CT) scans, the capsule-like rim is an essential radiological characteristic for differentiating AIP from PAC. It presents as a hypoattenuating halo surrounding the pancreas. However, this characteristic is infrequently observed in ultrasonography. Objectives: The aim of this study was to assess the accuracy of the thickness measurement of the capsule-like structure surrounding lesions during ultrasonography in order to distinguish between AIP and PAC. Patients and Methods: This case-control study was conducted on 19 patients with type 1 AIP (AIP1) as the case group and 37 patients with PAC as the controls. The ultrasound images of these patients were obtained from our institute's database. The thickest part of the hyperechoic capsule-like structure around lesions was identified and measured on the workstation retrospectively. The difference in the thickness of the capsule-like structure between AIP1 and PAC was compared in all lesions and mass lesions, respectively. The optimal cut-off thickness was determined by the maximum Youden index (calculated as sensitivity + specificity - 1). A P-value of < 0.05 (or < 0.05/3 after applying the Bonferroni correction) was considered statistically significant. Results: All lesions appeared hypoechoic, and there were no significant differences in gender, age, abdominal pain symptoms, jaundice, or weight loss between the case and control groups (P > 0.05). However, there was a significant difference regarding the involved pancreatic location (P = 0.008). Among the lesions, 46 were mass lesions. The hyperechoic capsule-like rim was thicker in the case group compared to the control group for all lesions (mean = 0.40 ± 0.12 vs. 0.32 ± 0.09 cm, P = 0.006) and also for mass lesions (mean = 0.41 ± 0.13 vs. 0.31 ± 0.09 cm, P = 0.006). The cut-off thickness for AIP1 was estimated at 0.41 cm, according to the maximum Youden index in both all lesions and mass lesions. The sensitivity, specificity, accuracy, positive predictive value, negative predictive value, and odds ratio for all lesions were 0.58, 0.86, 0.77, 0.69, 0.80, and 8.80 (95% confidence interval [CI]: 2.37 – 32.64), respectively. In mass lesions, the corresponding values were 0.58, 0.88, 0.80, 0.64, 0.86, and 10.50 (95% CI: 2.23 – 49.52), respectively. Conclusion: Patients with a hyperechoic capsule-like rim thickness of ≥0.41 cm during ultrasonography are more likely to have AIP1. This finding holds valuable clinical significance in differentiating between AIP1 and PAC.
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Agboola AA, Mohamed KH, Syed M, Shiwlani S, Butt R, Reza RR, Haseeb M, Nasir H. Type 1 Autoimmune Pancreatitis Masquerading as Pancreatic Head Carcinoma. Cureus 2023; 15:e47471. [PMID: 38022068 PMCID: PMC10662655 DOI: 10.7759/cureus.47471] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 10/22/2023] [Indexed: 12/01/2023] Open
Abstract
Obstructive jaundice is a joint clinical presentation with many etiologies, including pancreatic cancer and autoimmune pancreatitis (AIP). Differentiating between these two conditions is pivotal due to the divergent management approaches and prognoses. In this case report, we present a case of a 49-year-old female patient who presented with weight loss, intermittent chronic abdominal pain, and jaundice. She was initially suspected of having pancreatic cancer because of clinical presentation and imaging findings. However, she was ultimately diagnosed with Type 1 AIP due to histopathology findings and elevated immunoglobulin G4. This case highlights the complexities in diagnosis, the role of advanced imaging techniques and tissue sampling, and the lessons learned regarding managing this challenging clinical scenario.
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Affiliation(s)
| | - Khalid H Mohamed
- Neurology, Sheffield Teaching Hospitals NHS Foundation Trust, Sheffield, GBR
| | - Maria Syed
- Surgery, Aga Khan University Hospital, Karachi, PAK
| | | | - Rowaida Butt
- Family Medicine, Avalon University School of Medicine, Ohio, USA
| | | | - Muhammad Haseeb
- Internal Medicine, Allama Iqbal Medical College, Lahore, PAK
- Internal Medicine, Bahria International Hospital, Lahore, PAK
| | - Hira Nasir
- Internal Medicine, Mayo Hospital, Lahore, PAK
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Gong L, Shu B, Yu F, Zhang X, Chen J, Peng J. Main Diagnostic Criteria Usually Does Not Work for Autoimmune Pancreatitis Wrongly Presuming Malignancy. Gastroenterol Res Pract 2023; 2023:6652881. [PMID: 39291275 PMCID: PMC11407881 DOI: 10.1155/2023/6652881] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/28/2023] [Revised: 07/21/2023] [Accepted: 08/16/2023] [Indexed: 09/19/2024] Open
Abstract
Background Autoimmune pancreatitis (AIP) usually responds dramatically to steroid therapy. Occasionally, however, misdiagnosed patients have undergone pancreaticoduodenectomy. This study is aimed at providing useful information to improve the accuracy of diagnosis before surgery and thus avoid unnecessary resections in patients with AIP. Methods From January 2015 to February 2020, a series of patients were enrolled, having undergone pancreaticoduodenectomy for presumed malignancy. AIP diagnoses were confirmed by postoperative pathology. The demographic and clinical data of the AIP patients were evaluated. The main diagnostic criteria (HISORt, Asian, and ICDC) for AIP were applied to assess whether and how unnecessary surgery could have been avoided. Results A total of 124 cases of pancreaticoduodenectomy were performed for presumed malignancy. Six patients were diagnosed with benign disease and five with AIP. The prevalences of benign disease and AIP were 4.8% and 4%, respectively. Four patients were female and 1 male, with a mean age of 60.0 years old. Jaundice, pain, and weight loss were observed in 100%, 20%, and 40% of AIP patients, respectively. The radiologic features of the AIP patients were a diffusely enlarged gland (40.0%), a focally enlarged gland (40.0%), pancreatic ductal dilatation (60.0%), upstream parenchymal atrophy (20.0%), bile duct thickening (66.0%), and bile duct stricture (40.0%). Based on the diagnostic criteria for AIP, surgery could have been avoided in two cases. Conclusions IgG4 measurement and integrated use of major diagnostic criteria should be emphasized in every patient eligible for pancreaticoduodenectomies.
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Affiliation(s)
- Lei Gong
- Department of Surgery, Beijing Shijitan Hospital, Capital Medical University, Beijing 100038, China
| | - Bin Shu
- Center of Hepatopancreatobiliary Diseases, Beijing Tsinghua Changgung Hospital, School of Clinical Medicine, Tsinghua University, Beijing 102218, China
| | - Fei Yu
- Center of Hepatopancreatobiliary Diseases, Beijing Tsinghua Changgung Hospital, School of Clinical Medicine, Tsinghua University, Beijing 102218, China
| | - Xinjing Zhang
- Department of Surgery, Beijing Shijitan Hospital, Capital Medical University, Beijing 100038, China
| | - Jianfei Chen
- Department of Surgery, Beijing Shijitan Hospital, Capital Medical University, Beijing 100038, China
| | - Jirun Peng
- Department of Surgery, Beijing Shijitan Hospital, Capital Medical University, Beijing 100038, China
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Ogunlaja T, Oni E, Ibeawuchi M, Sattar L, Eshete FD, Agyebinti FB. Obstructive Jaundice Mimicking Pancreatic Cancer: An Unusual Presentation of Autoimmune Pancreatitis. Cureus 2023; 15:e45970. [PMID: 37900366 PMCID: PMC10600591 DOI: 10.7759/cureus.45970] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 09/24/2023] [Indexed: 10/31/2023] Open
Abstract
Autoimmune pancreatitis (AIP) is an uncommon variant of chronic pancreatitis characterized by inflammatory changes within the pancreatic tissue triggered by autoimmune mechanisms. It is known to mimic pancreatic cancer due to its similar clinical and radiological presentations. We underline a case of a 55-year-old male who presented with weight loss, jaundice, and pruritus. Radiological imaging suggested a pancreatic mass, raising suspicion of malignancy. However, subsequent evaluation, absence of parenchymal tissue and lymphoplasmacytic cells on endoscopic ultrasound-guided biopsy, and elevated serum immunoglobulin G4 level resulted in the diagnosis of AIP. Our case emphasizes that AIP should be included in the differential diagnosis of obstructive jaundice, especially when clinical and radiological findings are inconclusive for pancreatic cancer.
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Affiliation(s)
- Tiwalade Ogunlaja
- General Surgery, Peoples Friendship University of Russia, Moscow, RUS
| | - Efe Oni
- Internal Medicine, American University of Antigua, Osbourn, ATG
| | | | - Lubna Sattar
- Medicine, Shadan Institute of Medical Sciences, Hyderabad, IND
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Kamisawa T. Epidemiology of Autoimmune Pancreatitis. THE PANCREAS 2023:533-539. [DOI: 10.1002/9781119876007.ch68] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 01/02/2025]
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Shimosegawa T. Clinical Manifestation of Type 1 Autoimmune Pancreatitis. THE PANCREAS 2023:546-553. [DOI: 10.1002/9781119876007.ch70] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 01/02/2025]
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Seki M, Ninomiya E, Saiura A, Takahashi Y, Inoue Y, Katori M, Yamamoto N, Takamatsu M, Kato Y, Yamada K, Matsueda K, Ohkura Y. Clinicopathological study of surgically treated non-neoplastic diseases of the pancreas with special reference to autoimmune pancreatitis. Langenbecks Arch Surg 2023; 408:223. [PMID: 37270454 DOI: 10.1007/s00423-023-02944-y] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/14/2022] [Accepted: 05/14/2023] [Indexed: 06/05/2023]
Abstract
PURPOSE After the popularization of serum immunoglobulin G4 (IgG4) measurement and endoscopic ultrasound-guided fine-needle aspiration (EUS-FNA) in our institute, surgical resection for non-neoplastic diseases of the pancreas became less common. Although the incidence of such false-positive cases was clarified in the 10-year period after the introduction of these measures (2009-2018), these data were not compared with the 30 years before 2009 (1979-2008). This study was performed to determine the percentage of autoimmune pancreatitis (AIP) that was included during the latter period and how the numbers of false-positive cases differed between the two periods. METHODS From 1979 to 2008, 51 patients had clinical suspicion of pancreatic carcinoma (false-positive disease). Among these 51 patients, 32 non-alcoholic patients who had tumor-forming chronic pancreatitis (TFCP) were clinically, histologically, and immunohistochemically compared with 11 patients who had TFCP during the latter 10-year period. RESULTS Retrospective IgG4 immunostaining of false-positive TFCP revealed 14 (35.0%) cases of AIP in the former 30 years versus 5 (45.5%) in the latter 10 years. There were 40 (5.9%) cases of TFCP among 675 patients in the former 30 years and 11 (0.9%) among 1289 patients in the latter 10 years. CONCLUSIONS When the TFCP ratio of pancreatic resections and the AIP ratio of false-positive TFCPs were compared between the two periods, the TFCP ratio was 5.9% versus 0.9% and the AIP ratio was 35.0% versus 45.5%, respectively. It can thus be speculated that IgG4 measurement and EUS-FNA are absolutely imperative for the diagnosis of TFCP.
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Affiliation(s)
- Makoto Seki
- Departments of Hepato-Biliary-Pancreatic Surgery, Cancer Institute of the Japanese Foundation for Cancer Research, 3-10-6 Ariake, Koto-Ku, Tokyo, 135-8558, Japan.
- Department of Surgery, Mitaka Central Hospital, 5-23-10, Kami-Renjaku, Mitaka City, Tokyo, 181-0012, Japan.
| | - Eiji Ninomiya
- Department of Endoscopy, Kasumigaseki Building Clinic, 3-5-2-2F, Kasumigaseki, Chiyoda-Ku, Tokyo, 100-6012, Japan
| | - Akio Saiura
- Departments of Hepato-Biliary-Pancreatic Surgery, Cancer Institute of the Japanese Foundation for Cancer Research, 3-10-6 Ariake, Koto-Ku, Tokyo, 135-8558, Japan
- Department of Hepato-Biliary-Pancreatic Surgery, Juntendo University Hospital, 2-1-1, Hongo, Bunkyo-Ku, Tokyo, 113-0033, Japan
| | - Yu Takahashi
- Departments of Hepato-Biliary-Pancreatic Surgery, Cancer Institute of the Japanese Foundation for Cancer Research, 3-10-6 Ariake, Koto-Ku, Tokyo, 135-8558, Japan
| | - Yosuke Inoue
- Departments of Hepato-Biliary-Pancreatic Surgery, Cancer Institute of the Japanese Foundation for Cancer Research, 3-10-6 Ariake, Koto-Ku, Tokyo, 135-8558, Japan
| | - Masamichi Katori
- Departments of Pathology, Cancer Institute of the Japanese Foundation for Cancer Research, 3-10-6 Ariake, Koto-Ku, Tokyo, 135-8558, Japan
| | - Noriko Yamamoto
- Departments of Pathology, Cancer Institute of the Japanese Foundation for Cancer Research, 3-10-6 Ariake, Koto-Ku, Tokyo, 135-8558, Japan
| | - Manabu Takamatsu
- Departments of Pathology, Cancer Institute of the Japanese Foundation for Cancer Research, 3-10-6 Ariake, Koto-Ku, Tokyo, 135-8558, Japan
| | - Yo Kato
- Departments of Pathology, Cancer Institute of the Japanese Foundation for Cancer Research, 3-10-6 Ariake, Koto-Ku, Tokyo, 135-8558, Japan
| | - Keiko Yamada
- Departments of Diagnostic Radiology, Cancer Institute of the Japanese Foundation for Cancer Research, 3-10-6 Ariake, Koto-Ku, Tokyo, 135-8558, Japan
| | - Kiyoshi Matsueda
- Departments of Diagnostic Radiology, Cancer Institute of the Japanese Foundation for Cancer Research, 3-10-6 Ariake, Koto-Ku, Tokyo, 135-8558, Japan
| | - Yasuo Ohkura
- Department of Pathology, Kyorin University School of Medicine, 6-20-2 Shinkawa, Mitaka City, Tokyo, 181-8618, Japan
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Shiraishi M, Igarashi T, Hiroaki F, Oe R, Ohki K, Ojiri H. Radiomics based on diffusion-weighted imaging for differentiation between focal-type autoimmune pancreatitis and pancreatic carcinoma. Br J Radiol 2022; 95:20210456. [PMID: 35946923 PMCID: PMC9733621 DOI: 10.1259/bjr.20210456] [Citation(s) in RCA: 4] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/10/2021] [Revised: 07/31/2022] [Accepted: 08/04/2022] [Indexed: 11/05/2022] Open
Abstract
OBJECTIVE To evaluate the parameters of support vector machine (SVM) using imaging data generated from the apparent diffusion coefficient (ADC) to differentiate between focal-type autoimmune pancreatitis (f-AIP) and pancreatic ductal adenocarcinoma (PDAC) when using SVM based on diffusion-weighted imaging. METHODS The 2D-ADCmean and texture parameters (16 texture features × [non-filter+17 filters]) were retrospectively segmented by 2 readers in 28 patients with f-AIP and 77 patients with pathologically proven PDAC. The diagnostic accuracy of the SVM model was evaluated by receiver operating characteristic curve analysis and calculation of the area under the curve (AUC). Interreader reliability was assessed by intraclass correlation coefficient (ICC). RESULTS The 2D-ADCmean and 3D-ADCmean were significantly lower in cases of f-AIP (1.10-1.15 × 10-3 mm2/s and 1.21-1.23× 10-3 mm2/s, respectively) vs PDAC (1.29-1.33 × 10-3 mm2/s and 1.41-1.43 × 10-3 mm2/s, respectively), with excellent and good interreader reliability, respectively (ICC = 0.909 and 0.891, respectively). Among the texture parameters, energy with exponential filtering yielded the highest AUC (Reader 1: 74.7%, Reader 2: 81.5%), with fair interreader reliability (ICC = 0.707). The non-linear SVM, a combination of 2D-ADCmean, object volume and exponential-energy showed an AUC value of 96.2% in the testing cohorts. CONCLUSION Our results suggest that non-linear SVM using a combination of 2D-ADCmean, object volume, and exponential-energy may assist in differentiating f-AIP from PDAC. ADVANCES IN KNOWLEDGE The radiomics based on an apparent diffusion coefficient value may assist in differentiating f-AIP from PDAC.
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Affiliation(s)
- Megumi Shiraishi
- Department of Radiology, The Jikei University School of Medicine, Tokyo, Japan
| | - Takao Igarashi
- Department of Radiology, The Jikei University School of Medicine, Tokyo, Japan
| | - Fujioka Hiroaki
- Department of Radiology, The Jikei University School of Medicine, Tokyo, Japan
| | - Rika Oe
- Department of Radiology, The Jikei University School of Medicine, Tokyo, Japan
| | - Kazuyoshi Ohki
- Department of Radiology, The Jikei University School of Medicine, Tokyo, Japan
| | - Hiroya Ojiri
- Department of Radiology, The Jikei University School of Medicine, Tokyo, Japan
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Abstract
In 1995, Yoshida et al. proposed first the concept of "autoimmune pancreatitis" (AIP). Since then, AIP has been accepted as a new pancreatic inflammatory disease and is now divided two subtypes. Type 1 AIP affected immunoglobulin G4 (IgG4) and implicates the pancreatic manifestation of IgG4-related disease, while type 2 is characterized by neutrophil infiltration and granulocytic epithelial lesions (GEL). Recent research has clarified the clinical and pathophysiological aspects of type 1 AIP, which is more than type 2 among the Japanese population. However, many details remain unclear about the pathogenesis and progression of this disease. In this review, we discuss the current knowledge and recent advances relating to type 1 AIP.
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Affiliation(s)
- Kazushige Uchida
- Department of Gastroenterology and Hepatology, Kochi Medical School, Kochi University, Okocho-Kohasu, Nankoku, Kochi, 783-8505, Japan.
| | - Kazuichi Okazaki
- Kansai Medical University Kouri Hospital, 8-45 Kourihondori, Neyagawa, Osaka, 572-8551, Japan
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Lee SC, Yang CH, Chang CT, Yu KH. Diagnostic Utility of Serum IgG4 in Autoimmune Pancreatitis: An Updated Comprehensive Systematic Review and Meta-analysis. J Clin Gastroenterol 2022; 56:810-817. [PMID: 34516462 DOI: 10.1097/mcg.0000000000001612] [Citation(s) in RCA: 3] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/12/2021] [Accepted: 07/30/2021] [Indexed: 01/10/2023]
Abstract
OBJECTIVES Despite many studies suggesting an association between serum immunoglobulin G4 (sIgG4) and autoimmune pancreatitis (AIP), the evidence of utility in differentiation between AIP and pancreatic cancer (PC) remain uncertain. METHODS The analysis based on published studies. Data were pooled by means of a random-effects model, and sensitivity, specificity, diagnostic odds ratios (DOR), areas under summary receiver operating characteristic curves were calculated. RESULTS In the included thirteen studies, sIgG4 were measured in 594 patients with AIP and 958 patients with PC. The pooled sensitivity, specificity, DOR, and area under the curve were 0.72 [95% confidence interval (CI): 0.68-0.75], 0.93 (95% CI: 0.92-0.95), 51.37 (95% CI: 23.20-113.74), and 0.91 (95% CI: 0.87-0.95). Subgroup analyses of the DORs for region and year: Asia, (112.10; 95% CI: 27.72-453.32), non-Asia (26.01; 95% CI: 12.38-54.65), and year before 2011 (107.61; 95% CI: 39.30-294.68), year after 2011 (26.96; 95% CI: 9.78-74.32). Overall, sIgG4 was associated with AIP, the result revealed a moderate sensitivity 0.72 and high specificity 0.93. In the meta-analysis, the pooled DOR of sIgG4 levels of 2-fold upper limit 50.44 was similar with the DOR 51.37 when 1-fold cut-off value, but the summary receiver operating characteristic was 0.755 and 0.91. The higher specificity (from 93% to 98%) derived from the cut-off value (from 130-140 to 260-280 mg/dL) for sIgG4 occurred at a significant reduction in sensitivity (from 72% to 43%). CONCLUSIONS The study revealed sIgG4 is a good marker of AIP. Screening of sIgG4 may help clinicians differentiate between AIP and PC, and the best cut-off value should be 140 rather than 280 mg/dL.
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Affiliation(s)
- Shih-Ching Lee
- Division of Rheumatology, Allergy and Immunology, Department of Internal Medicine, Chang Gung Memorial Hospital, Chang Gung University, Tao-Yuan, Taiwan
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Li M, Bai X, Xu K, Wu X, Guo T, Jiang Q, Wang Q, Zhang S, Yang Y, Feng Y, Yang A. Peripancreatic vascular involvement in patients with type 1 autoimmune pancreatitis. Hepatobiliary Surg Nutr 2022; 11:355-362. [PMID: 35693390 PMCID: PMC9186208 DOI: 10.21037/hbsn-21-82] [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: 02/24/2021] [Accepted: 05/13/2021] [Indexed: 08/30/2023]
Abstract
BACKGROUND Type 1 autoimmune pancreatitis (AIP) is the pancreatic manifestation of IgG4-related disease. However, this benign disease can result in the peripancreatic vascular involvement (PVI) on occasion, which increases the difficulty of diagnosis and treatment of this clinical entity as well as for differentiating it from pancreatic malignancies. METHODS We retrospectively reviewed the information on demographics, clinical presentation, laboratory, imaging and endoscopic findings of 101 hospitalized patients with type 1 AIP treated in our department. All the patients were divided into non-PVI and PVI groups according to the first hospitalized medical data. Univariate and multivariate analyses were performed to analyse the potential predictive parameter(s) of PVI in AIP patients. RESULTS Among the 101 type 1 AIP patients, 52 (51.5%) exhibited PVI, with a male/female ratio 5.5:1. Their average age was 58.37±8.68 years old. Univariate analysis revealed that the location of pancreatitis lesions, including the pancreatic tail (P=0.010), the presence of splenomegaly (P=0.001) and the white blood cell (WBC) number in peripheral blood (P=0.020), were significantly associated with PVI. The location of pancreatitis lesions, including the pancreatic tail (P=0.023), and the presence of splenomegaly (P=0.010) were found to be independent predictors of the development of PVI by a multivariable regression analysis. A total of 18 out of 25 patients in PVI group who underwent corticosteroid treatment and no less than 6 months radiological follow-up showed improvement in vascular lesions, and no case exhibited exacerbation of PVI lesions during follow-up. Of 36 patients in non-PVI group who were followed up for no less than 6 months, only one case exhibited PVI. CONCLUSIONS This retrospective study demonstrated that type 1 AIP was associated with a high proportion of PVI. Pancreatic tail involvement and splenomegaly may predict the PVI in type 1 AIP. PVI lesions are reversible in a subset of patients.
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Affiliation(s)
- Meizi Li
- Department of Gastroenterology, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, Beijing, China
| | - Xiaoyin Bai
- Department of Gastroenterology, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, Beijing, China
| | - Kai Xu
- Department of Radiology, Peking Union Medical College Hospital, Beijing, China
| | - Xi Wu
- Department of Gastroenterology, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, Beijing, China
| | - Tao Guo
- Department of Gastroenterology, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, Beijing, China
| | - Qingwei Jiang
- Department of Gastroenterology, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, Beijing, China
| | - Qiang Wang
- Department of Gastroenterology, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, Beijing, China
| | - Shengyu Zhang
- Department of Gastroenterology, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, Beijing, China
| | - Yingyun Yang
- Department of Gastroenterology, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, Beijing, China
| | - Yunlu Feng
- Department of Gastroenterology, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, Beijing, China
| | - Aiming Yang
- Department of Gastroenterology, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, Beijing, China
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Tacelli M, Zaccari P, Petrone MC, Della Torre E, Lanzillotta M, Falconi M, Doglioni C, Capurso G, Arcidiacono PG. Differential EUS findings in focal type 1 autoimmune pancreatitis and pancreatic cancer: A proof-of-concept study. Endosc Ultrasound 2022; 11:216-222. [PMID: 35142701 PMCID: PMC9258021 DOI: 10.4103/eus-d-21-00111] [Citation(s) in RCA: 6] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/17/2021] [Accepted: 10/20/2021] [Indexed: 11/23/2022] Open
Abstract
BACKGROUND AND OBJECTIVES Autoimmune pancreatitis (AIP) often mimics pancreatic cancer (PC), particularly if presenting as a focal lesion. EUS may orient the differential diagnosis between them. This study aims to identify EUS findings that might be useful to differentiate type 1 focal autoimmune pancreatitis (f-AIP1) and PC. MATERIALS AND METHODS F-AIP1 and PC patients were retrospectively collected, matched, and compared. EUS findings considered were: focal mass echogenicity, loss of lobularity, distal atrophy, peripancreatic hypoechoic margins (PHM), pancreatic duct dilation, duct-penetrating sign (DPS), pancreatic/common bile duct thickened walls (PD/CBD-TW), and vessel infiltration (VI). Elastography findings were also recorded. Variables with a P < 0.05 at univariate analysis were included in logistic multiple regression. RESULTS Fifteen patients with f-AIP and 60 with PC were studied. FE was hypoechoic in all patients from both groups. PHM was observed in 40% of f-AIP1 cases but not in PC ones (P < 0.001). DPS was found in 10/15 (66.7%) f-AIP1 and in 7/60 (11.7%) PC patients (P < 0.001). PD-TW and CBD-TW were observed in 66.7%/60% f-AIP1 cases and in 6.7%/13.6% PC patients, respectively (P < 0.001 for both comparisons). Pancreatic masses were significantly different at EUS elastography (elastic respectively in 71.4% f-AIP1 and 3.8% PC, P < 0.001). VI was suspected in 20% of f-AIPs and 85% of PCs (P < 0.001). At multiple regression, PD-TW, CBD-TW, elastic pattern, and the absence of VI independently supported a diagnosis of f-AIP1. CONCLUSIONS Our results suggest that EUS findings deserve consideration in the diagnostic workup of AIP to improve the differential diagnosis with PC.
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Affiliation(s)
- Matteo Tacelli
- Pancreato-Biliary Endoscopy and EUS Division, Pancreas Translational and Clinical Research Center, San Raffaele Scientific Institute IRCCS, Milan, Italy
- Pancreas Translational and Clinical Research Center, IRCCS San Raffaele Scientific Institute, Università Vita-Salute San Raffaele, Milan, Italy
| | - Piera Zaccari
- Pancreato-Biliary Endoscopy and EUS Division, Pancreas Translational and Clinical Research Center, San Raffaele Scientific Institute IRCCS, Milan, Italy
- Department of Translational and Precision Medicine, Sapienza University, Rome, Italy
| | - Maria Chiara Petrone
- Pancreato-Biliary Endoscopy and EUS Division, Pancreas Translational and Clinical Research Center, San Raffaele Scientific Institute IRCCS, Milan, Italy
| | - Emanuel Della Torre
- Pancreas Translational and Clinical Research Center, IRCCS San Raffaele Scientific Institute, Università Vita-Salute San Raffaele, Milan, Italy
- Unit of Immunology, Rheumatology, Allergy and Rare Diseases (Unirar), San Raffaele Scientific Institute IRCCS, Milan, Italy
| | - Marco Lanzillotta
- Pancreas Translational and Clinical Research Center, IRCCS San Raffaele Scientific Institute, Università Vita-Salute San Raffaele, Milan, Italy
- Unit of Immunology, Rheumatology, Allergy and Rare Diseases (Unirar), San Raffaele Scientific Institute IRCCS, Milan, Italy
| | - Massimo Falconi
- Division of Pancreatic Surgery, Pancreas Translational and Clinical Research Center, San Raffaele Scientific Institute IRCCS, Milan, Italy
| | - Claudio Doglioni
- Department of Pathology, Pancreas Translational and Clinical Research Center, San Raffaele Scientific Institute IRCCS, Milan, Italy
| | - Gabriele Capurso
- Pancreato-Biliary Endoscopy and EUS Division, Pancreas Translational and Clinical Research Center, San Raffaele Scientific Institute IRCCS, Milan, Italy
| | - Paolo Giorgio Arcidiacono
- Pancreato-Biliary Endoscopy and EUS Division, Pancreas Translational and Clinical Research Center, San Raffaele Scientific Institute IRCCS, Milan, Italy
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Löhr JM, Vujasinovic M, Rosendahl J, Stone JH, Beuers U. IgG4-related diseases of the digestive tract. Nat Rev Gastroenterol Hepatol 2022; 19:185-197. [PMID: 34750548 DOI: 10.1038/s41575-021-00529-y] [Citation(s) in RCA: 55] [Impact Index Per Article: 18.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Accepted: 09/22/2021] [Indexed: 12/15/2022]
Abstract
IgG4-related conditions affecting the digestive tract are part of a multi-organ fibro-inflammatory disorder termed IgG4-related disease (IgG4-RD), with autoimmune pancreatitis and IgG4-related cholangitis being the most prominent manifestations. Gastrointestinal symptoms include jaundice, weight loss, abdominal pain, biliary strictures, and pancreatic and hepatic masses that mimic malignant diseases. IgG4-RD manifestations occur less frequently elsewhere in the digestive tract, namely in the oesophagus, retroperitoneum or intestine. Evidence-based European guidelines frame the current state-of-the-art in the diagnosis and management of IgG4-related digestive tract disease. Diagnosis is based on histology (if available), imaging, serology, other organ involvement and response to therapy (HISORt criteria). Few biomarkers beyond serum IgG4 concentrations are reliable. The first-line therapy (glucocorticoids) is swiftly effective but disease flares are common at low doses or after tapering. Second-line therapy might consist of other immunosuppressive drugs such as thiopurines or rituximab. Further trials, for example, of anti-CD19 drugs, are ongoing. Although an association between IgG4-RD and the development of malignancies has been postulated, the true nature of this relationship remains uncertain at this time.
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Affiliation(s)
- J-Matthias Löhr
- Department for Upper Digestive Diseases, Karolinska University Hospital and Karolinska Institutet, Stockholm, Sweden.
| | - Miroslav Vujasinovic
- Department for Upper Digestive Diseases, Karolinska University Hospital and Karolinska Institutet, Stockholm, Sweden
| | - Jonas Rosendahl
- Department of Internal Medicine I, Martin Luther University, Halle, Germany
| | - John H Stone
- Division of Rheumatology, Massachusetts General Hospital and Harvard Medical School, Boston, MA, USA
| | - Ulrich Beuers
- Department of Gastroenterology & Hepatology, Amsterdam University Medical Centers, Location AMC, Amsterdam, Netherlands
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Agarwal KK, Jassal R, Browne A, Hossain M, Akhtar R. Autoimmune Pancreatitis Masquerading as Pancreatic Cancer: A Case Report and Literature Review. Cureus 2022; 14:e21900. [PMID: 35265424 PMCID: PMC8898479 DOI: 10.7759/cureus.21900] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 02/03/2022] [Indexed: 11/05/2022] Open
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20
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Yu T, Wu Y, Liu J, Zhuang Y, Jin X, Wang L. The risk of malignancy in patients with IgG4-related disease: a systematic review and meta-analysis. Arthritis Res Ther 2022; 24:14. [PMID: 34986892 PMCID: PMC8728936 DOI: 10.1186/s13075-021-02652-2] [Citation(s) in RCA: 46] [Impact Index Per Article: 15.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/02/2021] [Accepted: 10/12/2021] [Indexed: 12/23/2022] Open
Abstract
BACKGROUND The relationship between IgG4-related disease (IgG4-RD) and the risk of malignancy is still controversial. This article focused on assessing the risk of cancer in patients with IgG4-RD by meta-analysis. METHODS We conducted a systematic review of the literature and meta-analysis characterizing the associated risk of overall malignancy and four site-specific malignancies (pancreas, lung, gastric and lymphoma) in patients with IgG4-RD. A search from 2003 to 2020 was performed using specified terms from PubMed, Embase, Web of Science and SinoMed. Random-effects model analysis was used to pool standardized incidence ratios (SIRs) and 95% confidence intervals (CIs). Subgroup and sensitivity analyses were conducted to clarify the heterogeneity of the included studies. Begg's funnel plot and Egger's linear regression test were used to evaluate the bias of the meta-analysis. A P value < 0.05 indicated the existence of publication bias. RESULTS A total of 10 studies were included in the article. The overall SIR estimates suggested an increased risk of overall cancer in IgG4-RD patients (SIR 2.57 95% CI 1.72-3.84) compared with the general population. The specific SIRs for pancreas and lymphoma were higher than those of the general population in IgG4-RD patients (SIR 4.07 95% CI 1.04-15.92, SIR 69.17 95% CI 3.91-1223.04, respectively). No significant associations were revealed in respiratory and gastric cancer (SIR 2.14 95% CI 0.97-4.75, SIR 0.95 95% CI 0.24-3.95, respectively). Four studies were found to be the major sources of heterogeneity by sensitivity analysis. There was no evidence of publication bias via Egger's test. CONCLUSION Compared with the general population, patients with IgG4-RD appear to have a higher risk of overall cancer, especially pancreatic and lymphoma. The risk of lung and gastric cancer was not different between IgG4-RD patients and the general population.
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Affiliation(s)
- Tingfeng Yu
- Department of Gastroenterology, Sun Yat-sen Memorial Hospital, Guangzhou, China
| | - Yaxian Wu
- Department of Gastroenterology, Sun Yat-sen Memorial Hospital, Guangzhou, China
| | - Jia Liu
- Department of Gastroenterology, Sun Yat-sen Memorial Hospital, Guangzhou, China
| | - Yanyan Zhuang
- Department of Gastroenterology, Sun Yat-sen Memorial Hospital, Guangzhou, China
| | - Xiaoyan Jin
- Department of General Practice, Sun Yat-sen Memorial Hospital, Guangzhou, China.
| | - Lingyun Wang
- Department of Gastroenterology, Sun Yat-sen Memorial Hospital, Guangzhou, China.
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Lu S, Liang J, Liao S, Wu D, Wu F, Li H. Use of MRI signal intensity ratio to differentiate between autoimmune pancreatitis and pancreatic ductal adenocarcinoma. Clin Radiol 2021; 77:e84-e91. [PMID: 34756699 DOI: 10.1016/j.crad.2021.10.003] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/14/2021] [Accepted: 10/01/2021] [Indexed: 11/03/2022]
Abstract
AIM To evaluate the accuracy of the lesion-to-erector spinae signal intensity ratio (SIR) on magnetic resonance imaging (MRI) for distinguishing autoimmune pancreatitis (AIP) from pancreatic ductal adenocarcinoma (PDA). MATERIALS AND METHODS The MRI data of 21 patients with AIP and 27 patients with PDA were analysed retrospectively, and the signal intensity in pancreatic lesions and erector spinae muscles at the same level on T2-weighted imaging (T2WI), arterial phase (AP) imaging, and delayed phase (DP) imaging was measured for calculation of SIRs. RESULTS The mean SIRs of the pancreatic lesions and erector spinae from T2WI, AP, and DP images of AIP patients were 0.96, 1.27, and 1.42, respectively, while those of PDA patients were 1.35, 0.80, and 0.91, respectively. The differences in the SIRs between the AIP and PDA groups were statistically significant (p<0.001), with corresponding area under curve (AUC) values of 0.925, 0.906, and 0.961, respectively. The optimal cut-off values for the SIRs on T2WI, AP and DP images were 1.21, 1.01, and 1.08, respectively. SIR values < 1.21 on T2WI, >1.01 on AP imaging, and >1.08 on DP imaging identified AIP with sensitivities of 85.7%, 90.5%, and 90.5%, respectively, and specificities of 81.5%, 74.6%, and 81.5%, respectively. The AUC values for SIRs did not differ significantly between T2WI and DP imaging or AP and DP imaging (Z = 0.778, p=0.436; Z = 1.279, p=0.201). CONCLUSION The SIRs of pancreatic lesions and erector spinae on T2WI, AP, and DP images can be used to differentiate AIP from PDA.
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Affiliation(s)
- S Lu
- Department of Radiology, The First Affiliated Hospital of Jinan University, Guangzhou, 510630, China; Department of Radiology, The First Affiliated Hospital of Gannan Medical University, Ganzhou, 341000, China
| | - J Liang
- Department of Radiology, Shenzhen Baoan Hospital, Southern Medical University, Shenzhen, 518101, China
| | - S Liao
- Department of Radiology, The First Affiliated Hospital of Gannan Medical University, Ganzhou, 341000, China
| | - D Wu
- Department of Radiology, The First Affiliated Hospital of Gannan Medical University, Ganzhou, 341000, China
| | - F Wu
- Department of Radiology, The First Affiliated Hospital of Gannan Medical University, Ganzhou, 341000, China
| | - H Li
- Department of Radiology, The First Affiliated Hospital of Jinan University, Guangzhou, 510630, China.
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Yoon SB, Moon SH, Song TJ, Kim JH, Kim MH. Endoscopic ultrasound-guided fine needle aspiration versus biopsy for diagnosis of autoimmune pancreatitis: Systematic review and comparative meta-analysis. Dig Endosc 2021; 33:1024-1033. [PMID: 33030283 DOI: 10.1111/den.13866] [Citation(s) in RCA: 26] [Impact Index Per Article: 6.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/22/2020] [Accepted: 10/01/2020] [Indexed: 12/17/2022]
Abstract
BACKGROUND Endoscopic ultrasound (EUS) is recommended for guiding the acquisition of pancreatic tissue in patients with suspected autoimmune pancreatitis (AIP). Data comparing EUS-guided fine needle aspiration (FNA) and fine needle biopsy (FNB) sampling in the diagnosis of AIP are limited. METHODS A comprehensive literature search of the PubMed, EMBASE, and Ovid MEDLINE databases was conducted until April 2020. The pooled rates of diagnostic yield for the histologic criteria of AIP, histologic tissue procurement, and adverse events were compared between FNA and FNB. Diagnostic yields were also compared between 19 gauge (G) and 22G needles. RESULTS This meta-analysis included nine studies comprising 309 patients with AIP who underwent FNA and seven studies comprising 131 patients who underwent FNB. The pooled diagnostic yields for level 1 or 2 histology criteria of AIP were 55.8% (95% confidence interval (CI) 37.0-73.9%, I2 = 91.1) for FNA and 87.2% (95% CI 68.8-98.1%, I2 = 69.4) for FNB (P = 0.030). The pooled histologic procurement rates for FNA and FNB were 91.3% (95% CI, 84.9-97.6%, I2 = 82.9) and 87.0% (95% CI, 77.8-96.1%, I2 = 40.0), respectively (P = 0.501). Adverse events were comparable between two groups. When analyzed by needle size, the diagnostic yield was better with a 19G needle than with a 22G needle (88.9% vs. 60.6%, P = 0.023). CONCLUSIONS The diagnostic yield may be better with FNB needles than with FNA needles for the diagnosis of AIP, despite the similar rate of histologic tissue procurement. A quantitative definition for the histologic sample adequacy for AIP may be warranted.
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Affiliation(s)
- Seung Bae Yoon
- Department of Internal Medicine, College of Medicine, The Catholic University of Korea, Seoul, South Korea
| | - Sung-Hoon Moon
- Department of Internal Medicine, Hallym University Sacred Heart Hospital, Hallym University College of Medicine, Anyang, South Korea
- Institute for Liver and Digestive Diseases, Hallym University, Chuncheon, South Korea
| | - Tae Jun Song
- Department of Internal Medicine, Asan Medical Center, University of Ulsan College of Medicine, Seoul, South Korea
| | - Jong Hyeok Kim
- Department of Internal Medicine, Hallym University Sacred Heart Hospital, Hallym University College of Medicine, Anyang, South Korea
- Institute for Liver and Digestive Diseases, Hallym University, Chuncheon, South Korea
| | - Myung-Hwan Kim
- Department of Internal Medicine, Asan Medical Center, University of Ulsan College of Medicine, Seoul, South Korea
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Masaki Y, Nakase H, Tsuji Y, Nojima M, Shimizu K, Mizuno N, Ikeura T, Uchida K, Ido A, Kodama Y, Seno H, Okazaki K, Nakamura S, Masamune A. The clinical efficacy of azathioprine as maintenance treatment for autoimmune pancreatitis: a systematic review and meta-analysis. J Gastroenterol 2021; 56:869-880. [PMID: 34426870 PMCID: PMC8382580 DOI: 10.1007/s00535-021-01817-9] [Citation(s) in RCA: 8] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/11/2021] [Accepted: 08/04/2021] [Indexed: 02/04/2023]
Abstract
The effectiveness of azathioprine (AZA) in preventing relapse and maintaining autoimmune pancreatitis (AIP) remission has been reported; however, most of these studies are case series with no randomized control trials available in the literature. Therefore, this study performed a systematic review and meta-analysis of the existing literature on this subject to determine the clinical efficacy of AZA as maintenance therapy for AIP patients. A systematic search was performed to identify studies on the clinical efficacy of AZA as maintenance therapy in AIP patients. The crude multiple relapse rate was estimated to assess the ability of AZA to control relapses in AIP. Pooled estimates were obtained using a random-effects model with the DerSimonian-Laird method. We identified AIP patients who did not respond to initial steroid treatment, experienced steroid weaning failure, or those who relapsed during remission as refractory cases. After reviewing the studies, ten articles fulfilled the inclusion criteria and were selected for meta-analysis. Of all 4504 patients, 3534 patients were treated with steroids, and 346 patients were treated with AZA for relapsed AIP. In this meta-analysis, 14/73 (19.2%) patients receiving AZA for refractory AIP relapsed. Meanwhile, 14/47 (29.8%) patients without AZA experienced relapse. The integrated odds ratio for relapse risk in patients receiving AZA was estimated to be 0.52 (p = 0.15). This systematic review and meta-analysis demonstrated the efficacy of AZA in preventing relapse of AIP, which supports the use of AZA as a maintenance treatment in patients with AIP who relapse upon withdrawal of steroid therapy.
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Affiliation(s)
- Yoshiharu Masaki
- Department of Gastroenterology and Hepatology, Sapporo Medical University School of Medicine, S-1, W-16, Chuo-ku, Sapporo, Hokkaido, 060-8543, Japan
| | - Hiroshi Nakase
- Department of Gastroenterology and Hepatology, Sapporo Medical University School of Medicine, S-1, W-16, Chuo-ku, Sapporo, Hokkaido, 060-8543, Japan.
| | - Yoshihisa Tsuji
- Department of General Medicine, Sapporo Medical University School of Medicine, Sapporo, Japan
| | - Masanori Nojima
- Center for Translational Research, The Institute of Medical Science, The University of Tokyo, Tokyo, Japan
| | - Kyoko Shimizu
- Institute of Gastroenterology, Tokyo Women's Medical University, Tokyo, Japan
| | - Nobumasa Mizuno
- Department of Gastroenterology, Aichi Cancer Center Hospital, Nagoya, Japan
| | - Tsukasa Ikeura
- The Third Department of Internal Medicine, Kansai Medical University, Hirakata, Japan
| | - Kazushige Uchida
- Department of Gastroenterology and Hepatology, Kochi Medical School, Kochi University, Nankoku, Japan
| | - Akio Ido
- Digestive and Lifestyle Diseases, Kagoshima University Graduate School of Medical and Dental Sciences, Kagoshima, Japan
| | - Yuzo Kodama
- Department of Gastroenterology, Kobe University, Kobe, Japan
| | - Hiroshi Seno
- Department of Gastroenterology and Hepatology, Kyoto University Graduate School of Medicine, Kyoto, Japan
| | | | - Seiji Nakamura
- Section of Oral and Maxillofacial Oncology, Division of Maxillofacial Diagnostic and Surgical Sciences, Faculty of Dental Science, Kyushu University, Fukuoka, Japan
| | - Atsushi Masamune
- Division of Gastroenterology, Tohoku University Graduate School of Medicine, Sendai, Japan
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Lanzillotta M, Della-Torre E, Wallace ZS, Stone JH, Karadag O, Fernández-Codina A, Arcidiacono PG, Falconi M, Dagna L, Capurso G. Efficacy and safety of rituximab for IgG4-related pancreato-biliary disease: A systematic review and meta-analysis. Pancreatology 2021; 21:1395-1401. [PMID: 34244040 DOI: 10.1016/j.pan.2021.06.009] [Citation(s) in RCA: 24] [Impact Index Per Article: 6.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/11/2021] [Revised: 06/22/2021] [Accepted: 06/29/2021] [Indexed: 12/11/2022]
Abstract
BACKGROUND Type I autoimmune pancreatitis (AIP) and IgG4-related sclerosing cholangitis (IgG4-SC) belong to the IgG4-related disease (IgG4-RD) spectrum. Both entities respond to glucocorticoids, but iatrogenic toxicity associated with prolonged steroid therapy and relapse represent relevant clinical concerns in the long-term. Rituximab is increasingly used as an effective alternative strategy to induce remission but data regarding the safety and efficacy of B-cell depletion therapy for pancreato-biliary involvement of IgG4-RD are limited. We performed a systematic review and meta-analysis to estimate the rate of remission, flare, and adverse events (AEs) occurring in pancreato-biliary IgG4-RD following rituximab treatment. METHODS The MEDLINE, SCOPUS, and EMBASE databases were searched from inception to December 2020 to identify studies reporting the outcomes of IgG4-related pancreato-biliary disease after treatment with rituximab. Studies involving ≥2 patients were selected. In case of duplicated studies, the most recent or the one with the biggest N were chosen. The study was conducted in accordance with Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines. Pooled effects were calculated using a random-effect model and expressed in terms of pooled remission, relapse, and AEs rates. RESULTS Seven cohort studies met inclusion criteria and 101 patients were included. Reasons for rituximab administration were new disease onset (18.5%), disease flare after glucocorticoids (63.5%), and glucocorticoids intolerance (17.9%). The median follow-up time was 19 months. The pooled rate of complete response at 6 months was 88.9% (95%CI 80.5-93.9) with no heterogeneity (I2 = 0%). The pooled estimate of relapse rate was 21% (95%CI 10.5-40.3) with moderate heterogeneity (I2 = 51%). A higher rate of relapse (35.9%, 95%CI 17.3-60.1) was reported in studies including patients with multiorgan involvement (OOI). The median time to relapse was 10 months. The pooled estimate of rituximab-related AEs was 25% (95%CI 8.8-53) with substantial heterogeneity (I2 = 73.6%). No publication bias was observed. CONCLUSION Treatment of IgG4-related pancreato-biliary disease with rituximab is associated with high remission rate, a higher relapse rate in the presence of OOI, and limited AEs. Randomized controlled trials with adequate power are needed to confirm these findings.
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Affiliation(s)
- Marco Lanzillotta
- Università Vita-Salute San Raffaele, IRCCS San Raffaele Scientific Institute, Milan, Italy; Unit of Immunology, Rheumatology, Allergy and Rare Diseases, IRCCS San Raffaele Scientific Institute, Milan, Italy.
| | - Emanuel Della-Torre
- Università Vita-Salute San Raffaele, IRCCS San Raffaele Scientific Institute, Milan, Italy; Unit of Immunology, Rheumatology, Allergy and Rare Diseases, IRCCS San Raffaele Scientific Institute, Milan, Italy
| | | | - John H Stone
- Rheumatology Unit, Massachusetts General Hospital, Boston, MA, USA
| | - Omer Karadag
- Faculty of Medicine, Department of Internal Medicine, Division of Rheumatology, Hacettepe University, Sihhiye-Ankara, Turkey
| | - Andreu Fernández-Codina
- Rheumatology Division and General Internal Medicine division-Windsor Campus, Western University, 268 Grosvenor St, D2-191, Rheumatology Centre, St. Joseph's Health Care, London, Ontario, Canada
| | - Paolo Giorgio Arcidiacono
- Division of Pancreatic Surgery and Endosonography Division, Pancreas Translational and Clinical Research Center, IRCCS San Raffaele Scientific Institute, Milan, Italy
| | - Massimo Falconi
- Pancreato-Biliary Endoscopy and Endosonography Division, Pancreas Translational and Clinical Research Center, IRCCS San Raffaele Scientific Institute, Milan, Italy
| | - Lorenzo Dagna
- Università Vita-Salute San Raffaele, IRCCS San Raffaele Scientific Institute, Milan, Italy; Unit of Immunology, Rheumatology, Allergy and Rare Diseases, IRCCS San Raffaele Scientific Institute, Milan, Italy
| | - Gabriele Capurso
- Division of Pancreatic Surgery and Endosonography Division, Pancreas Translational and Clinical Research Center, IRCCS San Raffaele Scientific Institute, Milan, Italy
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25
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Marya NB, Powers PD, Chari ST, Gleeson FC, Leggett CL, Abu Dayyeh BK, Chandrasekhara V, Iyer PG, Majumder S, Pearson RK, Petersen BT, Rajan E, Sawas T, Storm AC, Vege SS, Chen S, Long Z, Hough DM, Mara K, Levy MJ. Utilisation of artificial intelligence for the development of an EUS-convolutional neural network model trained to enhance the diagnosis of autoimmune pancreatitis. Gut 2021; 70:1335-1344. [PMID: 33028668 DOI: 10.1136/gutjnl-2020-322821] [Citation(s) in RCA: 85] [Impact Index Per Article: 21.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/13/2020] [Revised: 09/10/2020] [Accepted: 09/15/2020] [Indexed: 02/06/2023]
Abstract
OBJECTIVE The diagnosis of autoimmune pancreatitis (AIP) is challenging. Sonographic and cross-sectional imaging findings of AIP closely mimic pancreatic ductal adenocarcinoma (PDAC) and techniques for tissue sampling of AIP are suboptimal. These limitations often result in delayed or failed diagnosis, which negatively impact patient management and outcomes. This study aimed to create an endoscopic ultrasound (EUS)-based convolutional neural network (CNN) model trained to differentiate AIP from PDAC, chronic pancreatitis (CP) and normal pancreas (NP), with sufficient performance to analyse EUS video in real time. DESIGN A database of still image and video data obtained from EUS examinations of cases of AIP, PDAC, CP and NP was used to develop a CNN. Occlusion heatmap analysis was used to identify sonographic features the CNN valued when differentiating AIP from PDAC. RESULTS From 583 patients (146 AIP, 292 PDAC, 72 CP and 73 NP), a total of 1 174 461 unique EUS images were extracted. For video data, the CNN processed 955 EUS frames per second and was: 99% sensitive, 98% specific for distinguishing AIP from NP; 94% sensitive, 71% specific for distinguishing AIP from CP; 90% sensitive, 93% specific for distinguishing AIP from PDAC; and 90% sensitive, 85% specific for distinguishing AIP from all studied conditions (ie, PDAC, CP and NP). CONCLUSION The developed EUS-CNN model accurately differentiated AIP from PDAC and benign pancreatic conditions, thereby offering the capability of earlier and more accurate diagnosis. Use of this model offers the potential for more timely and appropriate patient care and improved outcome.
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Affiliation(s)
- Neil B Marya
- Gastroenterology and Hepatology, Mayo Clinic, Rochester, Minnesota, USA
| | | | - Suresh T Chari
- Gastroenterology, Hepatology and Nutrition, University of Texas MD Anderson Cancer Center, Houston, Texas, USA
| | - Ferga C Gleeson
- Gastroenterology and Hepatology, Mayo Clinic, Rochester, Minnesota, USA
| | - Cadman L Leggett
- Gastroenterology and Hepatology, Mayo Clinic, Rochester, Minnesota, USA
| | | | | | - Prasad G Iyer
- Gastroenterology and Hepatology, Mayo Clinic, Rochester, Minnesota, USA
| | - Shounak Majumder
- Gastroenterology and Hepatology, Mayo Clinic, Rochester, Minnesota, USA
| | - Randall K Pearson
- Gastroenterology and Hepatology, Mayo Clinic, Rochester, Minnesota, USA
| | - Bret T Petersen
- Gastroenterology and Hepatology, Mayo Clinic, Rochester, Minnesota, USA
| | - Elizabeth Rajan
- Gastroenterology and Hepatology, Mayo Clinic, Rochester, Minnesota, USA
| | - Tarek Sawas
- Gastroenterology and Hepatology, Mayo Clinic, Rochester, Minnesota, USA
| | - Andrew C Storm
- Gastroenterology and Hepatology, Mayo Clinic, Rochester, Minnesota, USA
| | - Santhi S Vege
- Gastroenterology and Hepatology, Mayo Clinic, Rochester, Minnesota, USA
| | - Shigao Chen
- Diagnostic Radiology, Mayo Clinic, Rochester, Minnesota, USA
| | - Zaiyang Long
- Diagnostic Radiology, Mayo Clinic, Rochester, Minnesota, USA
| | - David M Hough
- Diagnostic Radiology, Mayo Clinic, Rochester, Minnesota, USA
| | - Kristin Mara
- Biomedical Statistics and Informatics, Mayo Clinic Rochester, Rochester, Minnesota, USA
| | - Michael J Levy
- Gastroenterology and Hepatology, Mayo Clinic, Rochester, Minnesota, USA
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26
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Cler SJ, Sharifai N, Baker B, Dowling JL, Pipkorn P, Yaeger L, Clifford DB, Dahiya S, Chicoine MR. IgG4-Related Disease of the Skull and Skull Base-A Systematic Review and Report of Two Cases. World Neurosurg 2021; 150:179-196.e1. [PMID: 33746107 DOI: 10.1016/j.wneu.2021.03.054] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/07/2021] [Revised: 03/10/2021] [Accepted: 03/11/2021] [Indexed: 12/15/2022]
Abstract
OBJECTIVE IgG4-related disease (IgG4-RD) is an inflammatory process that uncommonly can present in the skull base and calvarium and mimic a tumor but the nature of this condition is not well summarized in the neurosurgical literature. METHODS A review was performed of 2 cases of IgG4-RD in the skull base highlighting the diagnostic challenges with assessment of these skull base lesions, and a systematic review of relevant literature was carried out. RESULTS A systematic review of the literature conducted in accordance with PRISMA guidelines identified 113 articles, with 184 cases of IgG4-RD in the skull base or calvarium. The most commonly affected locations include the meninges, cavernous sinus, base of the posterior fossa, clivus, and mastoid bone. Headache, visual and auditory disturbances, cranial nerve dysfunction, and seizures were the most common presenting symptoms. Medical treatment was highly successful and most commonly consisted of corticosteroids coadministered with immunosuppressive agents such as rituximab. Prevalence seemed to be equal between sexes, and serum IgG4 levels were increased in 61% of patients. Delayed diagnosis and a need for multiple biopsies were reported in numerous cases. Two cases of skull base IgG4-RD from the authors' institution show the variable presentations of this disease. More invasive surgical biopsies were required in both cases, and corticosteroid treatment led to significant clinical improvement. CONCLUSIONS IgG4-RD is an uncommon condition with an increasing body of reported cases that can affect the skull base and calvarium and should be in the differential diagnosis, because delay in diagnosis and treatment may be common.
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Affiliation(s)
- Samuel J Cler
- Department of Neurosurgery, Washington University School of Medicine, Washington, D.C., USA.
| | - Nima Sharifai
- Department of Pathology and Immunology, Washington University School of Medicine, Washington, D.C., USA
| | - Brandi Baker
- Department of Neurology, Washington University School of Medicine, Washington, D.C., USA
| | - Joshua L Dowling
- Department of Neurosurgery, Washington University School of Medicine, Washington, D.C., USA
| | - Patrik Pipkorn
- Department of Otolaryngology, Washington University School of Medicine, Washington, D.C., USA
| | - Lauren Yaeger
- Bernard Becker Medical Library, Washington University School of Medicine, Washington, D.C., USA
| | - David B Clifford
- Department of Neurology, Washington University School of Medicine, Washington, D.C., USA; Department of Infectious Disease, Washington University School of Medicine, Washington, D.C., USA
| | - Sonika Dahiya
- Department of Pathology and Immunology, Washington University School of Medicine, Washington, D.C., USA
| | - Michael R Chicoine
- Department of Neurosurgery, Washington University School of Medicine, Washington, D.C., USA
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27
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Focal Autoimmune Pancreatitis: A Simple Flow Chart for a Challenging Diagnosis. Ultrasound Int Open 2021; 6:E67-E75. [PMID: 33490857 PMCID: PMC7815440 DOI: 10.1055/a-1323-4906] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/15/2020] [Accepted: 11/25/2020] [Indexed: 12/11/2022] Open
Abstract
Autoimmune pancreatitis is a chronic fibroinflammatory autoimmune mediated
disease of the pancreas. Clinically, obstructive painless jaundice and upper
abdominal pain are the main symptoms. Focal AIP is characterized by
segmental involvement of pancreatic parenchyma and it is often
radiologically represented by a pancreatic mass. In these cases, the
diagnosis can be very challenging, since it may be easily confused with
pancreatic cancer. Therefore, we suggest a combined approach of imaging
tests as the diagnostic workup. EUS study combined with CEUS and
elastography, if available, increases the accuracy of the method to rule out
cancer. Moreover, the lesion should always be sampled under EUS guidance to
obtain a cyto/histological diagnosis. The diagnostic workup should
also include the use of diagnostic clinical criteria (extrapancreatic
lesions, steroid response) and laboratory findings (CA 19.9 and IgG4
evaluations).
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28
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Mačinga P, Jarošová J, Špičák J, Hucl T. [Immunoglobulin G4-related disease in gastroenterology]. VNITRNI LEKARSTVI 2021; 67:76-83. [PMID: 34074105 DOI: 10.36290/vnl.2021.018] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 01/02/2025]
Abstract
IgG4-related disease is a recently defined clinical entity that can manifest itself in any organ. The most common gastrointestinal manifestations are diseases of the pancreas (autoimmune pancreatitis type 1) and biliary tree (IgG4-associated cholangitis); involvement of liver parenchyma is uncommon and the affection of tubular organs is very rare. IgG4-related pancreatitis and cholangitis can mimic malignancies in their clinical presentation. Diagnosis is often difficult and requires careful evaluation of the combination of symptoms, serology and imaging findings, while adhering to the established diagnostic criteria. The first line of treatment is the administration of corticoids and the remission is achieved in the vast majority of patients. In case of contraindication, intolerance or failure of corticotherapy, patients should receive B cell depletion therapy (rituximab). Based on the available knowledge, monotherapy with other immunosuppressants is not considered to be sufficiently effective. Some patients may benefit from maintenance treatment to prevent relapse, which is otherwise common in both IgG4-related pancreatitis and cholangitis. Recognized IgG4-related disease has a good prognosis, but some patients develop irreversible fibrotic changes in the affected organ with consequent dysfunction; the possible association of the disease with a higher risk of malignancy has not yet been reliably elucidated.
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29
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Pattabathula K, Waters PS, Hwang J, Bettington M, Singh M, Bryant RD, Cavallucci DJ, O'Rourke N. Diagnostic and therapeutic considerations in biopsy-proven type 2 autoimmune pancreatitis: comparative analysis with biopsy-proven type 1 autoimmune pancreatitis. ANZ J Surg 2020; 91:907-914. [PMID: 33369858 DOI: 10.1111/ans.16445] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/05/2020] [Revised: 10/26/2020] [Accepted: 11/03/2020] [Indexed: 12/16/2022]
Abstract
BACKGROUND Autoimmune processes are now an increasingly recognized cause of acute and chronic pancreatitis. Autoimmune pancreatitis is a rare, benign pathology with two distinct clinicopathologic subtypes. The aim of this study was to compare the presentation, diagnostic considerations and outcomes of patients with biopsy-proven type 1 and 2 autoimmune pancreatitis (AIP). METHODS A retrospective review of the Queensland Health pathology database of histologically proven AIP was conducted. Parameters compared included demographics, diagnostic criterion and post-treatment outcomes. RESULTS Twenty-three patients had a confirmed histological diagnosis of AIP (type 1 = 13, type 2 = 10). Patients with type 2 AIP were younger (median age 49 versus 59 years, P < 0.05). There was no significant difference in gender distribution of disease at presentation. Type 2 AIP presented with significant increased focal pancreatic changes on cross-sectional imaging (80% versus 54%, P < 0.05). Serum IgG4 levels were raised (>1.40 g/L) in 69% of patients with type 1 AIP and not detected in type 2 (P < 0.01). Concurrent underlying inflammatory bowel disease was present in a higher proportion of type 2 AIP (40% versus 15%, P < 0.05). A significantly increased proportion of patients with type 2 AIP underwent surgical resection (70% versus 30%, P < 0.05). Conservative management was utilized in more patients with type 1 disease (54% versus 30%). On follow-up, two patients have experienced symptomatic relapse at 6-18 months. CONCLUSIONS Diagnostic challenges do exist and clinicians must suspect 2 type AIP in young, serum IgG4-negative inflammatory bowel disease patients with recurrent pancreatitis.
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Affiliation(s)
- Krishna Pattabathula
- Department of Surgery, Royal Brisbane and Women's Hospital, Brisbane, Queensland, Australia
| | - Peadar S Waters
- Department of Surgery, Royal Brisbane and Women's Hospital, Brisbane, Queensland, Australia
| | - Jason Hwang
- Department of Gastroenterology, Royal Brisbane and Women's Hospital, Brisbane, Queensland, Australia
| | - Mark Bettington
- Department of Histopathology, Envoi Pathology, Brisbane, Queensland, Australia
| | - Mahendra Singh
- Pathology Queensland, Royal Brisbane and Women's Hospital, Brisbane, Queensland, Australia
| | - Richard D Bryant
- Department of Surgery, Royal Brisbane and Women's Hospital, Brisbane, Queensland, Australia.,Faculty of Medicine, The University of Queensland, Brisbane, Queensland, Australia
| | - David J Cavallucci
- Department of Surgery, Royal Brisbane and Women's Hospital, Brisbane, Queensland, Australia.,Faculty of Medicine, The University of Queensland, Brisbane, Queensland, Australia.,Department of Surgery, Wesley Hospital, Brisbane, Queensland, Australia
| | - Nicholas O'Rourke
- Department of Surgery, Royal Brisbane and Women's Hospital, Brisbane, Queensland, Australia.,Faculty of Medicine, The University of Queensland, Brisbane, Queensland, Australia.,Department of Surgery, Wesley Hospital, Brisbane, Queensland, Australia
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30
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Yoon SB, Moon SH, Kim JH, Song TJ, Kim MH. The use of immunohistochemistry for IgG4 in the diagnosis of autoimmune pancreatitis: A systematic review and meta-analysis. Pancreatology 2020; 20:1611-1619. [PMID: 33060017 DOI: 10.1016/j.pan.2020.10.028] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/22/2020] [Revised: 08/01/2020] [Accepted: 10/08/2020] [Indexed: 02/07/2023]
Abstract
BACKGROUND The diagnosis of autoimmune pancreatitis (AIP) remains challenging, especially when serum IgG4 is normal or imaging features are indeterminate. We performed a systematic review and meta-analysis to evaluate the performance of IgG4 immunostaining of pancreatic, biliary, and ampullary tissues as a diagnostic aid for AIP. METHODS A comprehensive literature search of the PubMed, EMBASE, and Ovid MEDLINE databases was conducted until February 2020. The methodological quality of each study was assessed according to the Quality Assessment of Diagnostic Accuracy Studies checklist. A random-effects model was used to summarize the diagnostic odds ratio and other measures of accuracy. RESULTS The meta-analysis included 20 studies comprising 346 patients with AIP and 590 patients with other pancreatobiliary diseases, including 371 pancreatobiliary malignancies. The summary estimates for tissue IgG4 in discriminating AIP and controls were as follows: diagnostic odds ratio 38.86 (95% confidence interval (CI), 18.70-80.75); sensitivity 0.64 (95% CI, 0.59-0.69); specificity 0.93 (95% CI, 0.91-0.95). The area under the curve was 0.939 for tissue IgG4 in discriminating AIP and controls. Subgroup analysis revealed no significant difference in diagnostic accuracy according to control groups (pancreatobiliary cancer versus other chronic pancreatitis) and sampling site (pancreas versus bile duct/ampulla). CONCLUSIONS Current data demonstrate that IgG4 immunostaining of pancreatic, biliary, and ampullary tissue has a high specificity but moderate sensitivity for diagnosing AIP. IgG4 immunostaining may be useful in supporting a diagnosis of AIP when AIP is clinically suspected, but a combination of imaging and serology does not provide a conclusive diagnosis.
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Affiliation(s)
- Seung Bae Yoon
- Department of Internal Medicine, College of Medicine, The Catholic University of Korea, Seoul, South Korea
| | - Sung-Hoon Moon
- Department of Internal Medicine, Hallym University Sacred Heart Hospital, Hallym University College of Medicine, Anyang, South Korea; Institute for Liver and Digestive Diseases, Hallym University, Chuncheon, South Korea.
| | - Jong Hyeok Kim
- Department of Internal Medicine, Hallym University Sacred Heart Hospital, Hallym University College of Medicine, Anyang, South Korea; Institute for Liver and Digestive Diseases, Hallym University, Chuncheon, South Korea
| | - Tae Jun Song
- Department of Internal Medicine, University of Ulsan, College of Medicine, Asan Medical Center, Seoul, South Korea
| | - Myung-Hwan Kim
- Department of Internal Medicine, University of Ulsan, College of Medicine, Asan Medical Center, Seoul, South Korea
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31
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[IgG4-related disease: Diagnostic criteria evolution toward the 2019 ACR/EULAR classification criteria]. Rev Med Interne 2020; 41:814-821. [PMID: 32732082 DOI: 10.1016/j.revmed.2020.06.015] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/20/2020] [Revised: 05/28/2020] [Accepted: 06/20/2020] [Indexed: 02/07/2023]
Abstract
The concept of IgG4-related disease (IgG4-RD) has recently been individualized in the early 2000s, but most of the organ involvements are known since more than 100 years. IgG4-RD is a non-malignant fibroinflammatory disorder, characterized by peculiar immunological and pathological abnormalities, which can affect virtually all organs or tissues. Diagnostic criteria have been proposed and have evolved rapidly, with general or organ specific criteria. An international and multidisciplinary group assembled by the American College of Rheumatology (ACR) and the European League Against Rheumatism (EULAR) has recently developed and validated a set of classification criteria called 2019 ACR/EULAR classification criteria for IgG4-related disease. The objective of this review is to discuss the evolution from organ specific and general diagnostic criteria toward the 2019 ACR/EULAR classification criteria, as well as respective benefits and limits of these criteria. The use of the 2019 ACR/EULAR classification criteria will help to better define homogeneous group of IgG4-RD patients in future clinical, epidemiological and basic science research studies on the disease.
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32
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Zhang R, Gao J, Zhao T, Zhang B, Wang C, Wang C, Cui L, Chen J, Fang S. A Case With IgG4-Related Spinal Pachymeningitis Causing Spinal Cord Compression. Front Neurol 2020; 11:500. [PMID: 32760335 PMCID: PMC7371922 DOI: 10.3389/fneur.2020.00500] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/14/2019] [Accepted: 05/07/2020] [Indexed: 11/13/2022] Open
Abstract
Immunoglobulin G4 (IgG4)-related disease is a systemic disease characterized by sclerosing lesions and an increased serum IgG4 level. This condition can involve any organ, but IgG4-related spinal pachymeningitis is relatively rare. In the current study, we report a case of spinal cord compression caused by IgG4-related spinal pachymeningitis. A 39-year-old man presented to us with a 15-day history of back pain and a 3-day history of dysuresia, exacerbated by weakness in the lower extremities for 2 days. Cervical magnetic resonance imaging (MRI) showed strip-shaped abnormal signals along the anterior and posterior borders of the spinal cord at the C5–T4 levels. The IgG level in cerebrospinal fluid was 718.0 mg/L. Thoracic MRI revealed strip-shaped abnormal signals with remarkable enhancement along the anterior and posterior borders of the dural sac at the T1–T6 levels. Histopathological examination confirmed IgG4-related spinal pachymeningitis. The symptoms worsened rapidly, and surgical resection of the space-occupying lesion in the vertebral canal was performed for spinal decompression. Corticosteroid therapy was administered, and the patient's motor functions were mildly improved. IgG4-related disease can manifest as spinal pachymeningitis and cause spinal cord compression. Clinicians should be aware of this rare condition, and early diagnosis, timely surgical decompression, and appropriate corticosteroid therapy should be highlighted.
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33
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Barresi L, Tacelli M, Crinò SF, Attili F, Petrone MC, De Nucci G, Carrara S, Manfredi G, Capurso G, De Angelis CG, Crocellà L, Fantin A, Dore MF, Garribba AT, Tarantino I, De Pretis N, Pagliari D, Rossi G, Manes G, Preatoni P, Barbuscio I, Tuzzolino F, Traina M, Frulloni L, Costamagna G, Arcidiacono PG, Buscarini E, Pezzilli R, Italian Association of Hospital Gastroenterologists and Endoscopists (AIGO), Italian Association for the Study of the Pancreas (AISP). Multicentric Italian survey on daily practice for autoimmune pancreatitis: Clinical data, diagnosis, treatment, and evolution toward pancreatic insufficiency. United European Gastroenterol J 2020; 8:705-715. [PMID: 32397913 PMCID: PMC7437084 DOI: 10.1177/2050640620924302] [Citation(s) in RCA: 26] [Impact Index Per Article: 5.2] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/07/2020] [Accepted: 04/03/2020] [Indexed: 12/22/2022] Open
Abstract
BACKGROUND Autoimmune pancreatitis (AIP) is a rare, and relatively new, form of chronic pancreatitis. The management of AIP can vary considerably among different centres in daily clinical practice. OBJECTIVES The aim of this study is to present a picture of epidemiological, clinical characteristics, outcomes, and the real-life practice in terms of management in several academic and non-academic centres in Italy. METHODS Data on the clinical presentation, diagnostic work-up, treatments, frequency of relapses, and long-term outcomes were retrospectively collected in a cohort of AIP patients diagnosed at 14 centres in Italy. RESULTS One hundred and six patients were classified as type 1 AIP, 48 as type 2 AIP, and 19 as not otherwise specified. Epidemiological, clinical, radiological, and serological characteristics, and relapses were similar to those previously reported for different types of AIP. Endoscopic cytohistology was available in 46.2% of cases, and diagnostic for AIP in only 35.2%. Steroid trial to aid diagnosis was administered in 43.3% cases, and effective in 93.3%. Steroid therapy was used in 70.5% of cases, and effective in 92.6% of patients. Maintenance therapy with low dose of steroid (MST) was prescribed in 25.4% of cases at a mean dose of 5 (±1.4) mg/die, and median time of MST was 60 days. Immunosuppressive drugs were rarely used (10.9%), and rituximab in 1.7%. Faecal elastase-1 was evaluated in only 31.2% of patients, and was pathological in 59.2%. CONCLUSIONS In this cohort of AIP patients, diagnosis and classification for subtype was frequently possible, confirming the different characteristics of AIP1 and AIP2 previously reported. Nevertheless, we observed a low use of histology and steroid trial for a diagnosis of AIP. Steroid treatment was the most used therapy in our cohort. Immunosuppressants and rituximab were rarely used. The evaluation of exocrine pancreatic insufficiency is underemployed considering its high prevalence.
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Affiliation(s)
- Luca Barresi
- Endoscopy Service, Department of Diagnostic and Therapeutic Services, IRCCS-ISMETT (Mediterranean Institute for Transplantation and Highly Specialized Therapies), Palermo, Italy
| | - Matteo Tacelli
- Endoscopy Service, Department of Diagnostic and Therapeutic Services, IRCCS-ISMETT (Mediterranean Institute for Transplantation and Highly Specialized Therapies), Palermo, Italy
- Section of Gastroenterology, Biomedical Department of Internal and Specialized Medicine (DI.BI.M.I.S.), University of Palermo, Palermo, Italy
| | - Stefano Francesco Crinò
- Gastroenterology and Digestive Endoscopy Unit, The Pancreas Institute, University Hospital of Verona, Verona, Italy
| | - Fabia Attili
- Digestive Endoscopy Unit, Fondazione Policlinico Universitario Agostino Gemelli IRCCS, Roma, Italy
| | - Maria Chiara Petrone
- Pancreato-Biliary Endoscopy and Endosonography Division, Pancreas Translational and Clinical Research Center, San Raffaele Scientific Institute IRCCS, Vita-Salute San Raffaele University, Milano, Italy
| | - Germana De Nucci
- Gastroenterology and Digestive Endoscopy Unit, ASST Rhodense, Garbagnate Milanese Hospitals, Milano, Italy
| | - Silvia Carrara
- Digestive Endoscopy Unit, Division of Gastroenterology, Humanitas Clinical and Research Center-IRCCS Rozzano (MI), Italy
| | - Guido Manfredi
- Gastroenterology and Digestive Endoscopy Department, Maggiore Hospital, ASST Crema, Crema, Italy
| | - Gabriele Capurso
- Pancreato-Biliary Endoscopy and Endosonography Division, Pancreas Translational and Clinical Research Center, San Raffaele Scientific Institute IRCCS, Vita-Salute San Raffaele University, Milano, Italy
- Digestive and Liver Disease Unit, S. Andrea Hospital, Roma, Italy
| | | | - Lucia Crocellà
- Gastroenterology Unit, Mauriziano Umberto I Hospital, Torino, Italy
| | - Alberto Fantin
- Department of Surgical, Oncological and Gastroenterological Sciences, Gastroenterology Division, Azienda Ospedaliera di Padova, University of Padova, Padua, Italy
| | | | | | - Ilaria Tarantino
- Endoscopy Service, Department of Diagnostic and Therapeutic Services, IRCCS-ISMETT (Mediterranean Institute for Transplantation and Highly Specialized Therapies), Palermo, Italy
| | - Nicolò De Pretis
- Gastroenterology unit, Pancreas center, University of Verona, Verona, Italy
| | - Danilo Pagliari
- Division of Internal Medicine and Gastroenterology & Pancreatic Unit, Fondazione Policlinico Universitario A. Gemelli-IRCCS, Universita’ del Sacro Cuore, Roma, Italy
| | - Gemma Rossi
- Pancreato-Biliary Endoscopy and Endosonography Division, Pancreas Translational and Clinical Research Center, San Raffaele Scientific Institute IRCCS, Vita-Salute San Raffaele University, Milano, Italy
| | - Gianpiero Manes
- Gastroenterology and Digestive Endoscopy Unit, ASST Rhodense, Garbagnate Milanese Hospitals, Milano, Italy
| | - Paoletta Preatoni
- Digestive Endoscopy Unit, Division of Gastroenterology, Humanitas Clinical and Research Center-IRCCS Rozzano (MI), Italy
| | - Ilenia Barbuscio
- Endoscopy Service, Department of Diagnostic and Therapeutic Services, IRCCS-ISMETT (Mediterranean Institute for Transplantation and Highly Specialized Therapies), Palermo, Italy
- Department of Surgical, Oncological and Gastroenterological Sciences, Gastroenterology Division, Azienda Ospedaliera di Padova, University of Padova, Padua, Italy
| | - Fabio Tuzzolino
- Research Office, IRCCS ISMETT (Mediterranean Institute for Transplantation and Advanced Specialized Therapies), Palermo, Italy
| | - Mario Traina
- Endoscopy Service, Department of Diagnostic and Therapeutic Services, IRCCS-ISMETT (Mediterranean Institute for Transplantation and Highly Specialized Therapies), Palermo, Italy
| | - Luca Frulloni
- Gastroenterology unit, Pancreas center, University of Verona, Verona, Italy
| | - Guido Costamagna
- Digestive Endoscopy Unit, Fondazione Policlinico Universitario Agostino Gemelli IRCCS, Roma, Italy
- Centre for Endoscopic Research Therapeutics and Training-CERTT, Università del SacroCuore, Roma, Italy
| | - Paolo Giorgio Arcidiacono
- Pancreato-Biliary Endoscopy and Endosonography Division, Pancreas Translational and Clinical Research Center, San Raffaele Scientific Institute IRCCS, Vita-Salute San Raffaele University, Milano, Italy
| | - Elisabetta Buscarini
- Gastroenterology and Digestive Endoscopy Department, Maggiore Hospital, ASST Crema, Crema, Italy
| | - Raffaele Pezzilli
- Pancreas Unit, Department of Gastroenterology, Sant’Orsola Polyclinic, Bologna, Italy
| | - Italian Association of Hospital Gastroenterologists and Endoscopists (AIGO), Italian Association for the Study of the Pancreas (AISP)
- Endoscopy Service, Department of Diagnostic and Therapeutic Services, IRCCS-ISMETT (Mediterranean Institute for Transplantation and Highly Specialized Therapies), Palermo, Italy
- Section of Gastroenterology, Biomedical Department of Internal and Specialized Medicine (DI.BI.M.I.S.), University of Palermo, Palermo, Italy
- Gastroenterology and Digestive Endoscopy Unit, The Pancreas Institute, University Hospital of Verona, Verona, Italy
- Digestive Endoscopy Unit, Fondazione Policlinico Universitario Agostino Gemelli IRCCS, Roma, Italy
- Pancreato-Biliary Endoscopy and Endosonography Division, Pancreas Translational and Clinical Research Center, San Raffaele Scientific Institute IRCCS, Vita-Salute San Raffaele University, Milano, Italy
- Gastroenterology and Digestive Endoscopy Unit, ASST Rhodense, Garbagnate Milanese Hospitals, Milano, Italy
- Digestive Endoscopy Unit, Division of Gastroenterology, Humanitas Clinical and Research Center-IRCCS Rozzano (MI), Italy
- Gastroenterology and Digestive Endoscopy Department, Maggiore Hospital, ASST Crema, Crema, Italy
- Digestive and Liver Disease Unit, S. Andrea Hospital, Roma, Italy
- Department of Medical Sciences, Division of Gastroenterology, University of Torino, Torino, Italy
- Gastroenterology Unit, Mauriziano Umberto I Hospital, Torino, Italy
- Department of Surgical, Oncological and Gastroenterological Sciences, Gastroenterology Division, Azienda Ospedaliera di Padova, University of Padova, Padua, Italy
- Gastroenterology Unit, Brotzu Hospital, Cagliari, Italy
- Gastroenterology Unit, AUSL Romagna, Ravenna, Italy
- Gastroenterology unit, Pancreas center, University of Verona, Verona, Italy
- Division of Internal Medicine and Gastroenterology & Pancreatic Unit, Fondazione Policlinico Universitario A. Gemelli-IRCCS, Universita’ del Sacro Cuore, Roma, Italy
- Research Office, IRCCS ISMETT (Mediterranean Institute for Transplantation and Advanced Specialized Therapies), Palermo, Italy
- Centre for Endoscopic Research Therapeutics and Training-CERTT, Università del SacroCuore, Roma, Italy
- Pancreas Unit, Department of Gastroenterology, Sant’Orsola Polyclinic, Bologna, Italy
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Wallace ZS, Perugino C, Matza M, Deshpande V, Sharma A, Stone JH. Immunoglobulin G4-related Disease. Clin Chest Med 2020; 40:583-597. [PMID: 31376893 DOI: 10.1016/j.ccm.2019.05.005] [Citation(s) in RCA: 35] [Impact Index Per Article: 7.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/14/2022]
Abstract
Immunoglobulin G4 (IgG4)-Related Disease (IgG4-RD) can cause fibroinflammatory lesions in nearly any organ and lead to organ dysfunction and irreversible damage. In addition to frequent involvement of the salivary glands, lacrimal glands, and/or pancreas, IgG4-RD often affects the chest. Thoracic manifestations include lung nodules and consolidations, pleural thickening, aortitis, and lymphadenopathy. The diagnosis is made after careful clinicopathologic correlation because there is no single diagnostic test with excellent sensitivity or specificity. Biopsy of pulmonary lesions can be useful for distinguishing IgG4-RD from common mimickers. Immunosuppressive regimens, such as glucocorticoids and/or glucocorticoid-sparing agents, form the cornerstone of treatment.
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Affiliation(s)
- Zachary S Wallace
- Clinical Epidemiology Program, Massachusetts General Hospital, Harvard Medical School, 55 Fruit Street, Boston, MA 02114, USA; Rheumatology Unit, Division of Rheumatology, Allergy, and Immunology, Massachusetts General Hospital, Harvard Medical School, 55 Fruit Street, Boston, MA 02114, USA.
| | - Cory Perugino
- Rheumatology Unit, Division of Rheumatology, Allergy, and Immunology, Massachusetts General Hospital, Harvard Medical School, 55 Fruit Street, Boston, MA 02114, USA
| | - Mark Matza
- Rheumatology Unit, Division of Rheumatology, Allergy, and Immunology, Massachusetts General Hospital, 55 Fruit Street, Boston, MA 02114, USA
| | - Vikram Deshpande
- Department of Pathology, Massachusetts General Hospital, Harvard Medical School, 55 Fruit Street, Boston, MA 02114, USA
| | - Amita Sharma
- Department of Radiology, Massachusetts General Hospital, Harvard Medical School, 55 Fruit Street, Boston, MA 02114, USA
| | - John H Stone
- Rheumatology Unit, Division of Rheumatology, Allergy, and Immunology, Massachusetts General Hospital, Harvard Medical School, 55 Fruit Street, Boston, MA 02114, USA
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Oh D, Song TJ, Moon SH, Kim JH, Lee NJ, Hong SM, Lee JS, Jo SJ, Cho DH, Park DH, Lee SS, Seo DW, Lee SK, Kim MH. Type 2 Autoimmune Pancreatitis (Idiopathic Duct-Centric Pancreatitis) Highlighting Patients Presenting as Clinical Acute Pancreatitis: A Single-Center Experience. Gut Liver 2020; 13:461-470. [PMID: 30970429 PMCID: PMC6622566 DOI: 10.5009/gnl18429] [Citation(s) in RCA: 20] [Impact Index Per Article: 4.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/20/2018] [Revised: 11/04/2018] [Accepted: 11/10/2018] [Indexed: 12/15/2022] Open
Abstract
Background/Aims Type 2 autoimmune pancreatitis (AIP) has been considered extremely rare in East Asia. This study aimed to clarify the prevalence, clinical characteristics and radiological findings of type 2 AIP highlighting patients presenting as acute pancreatitis in a single center. Methods Type 2 AIP patients were classified according to International Consensus Diagnostic Criteria. Radiological findings were compared between type 2 AIP presenting as acute pancreatitis and gallstone pancreatitis. Results Among 244 patients with AIP, 27 (11.1%) had type 2 AIP (definite, 15 [55.5%] and probable 12 [44.5%]). The median age of patients with type 2 AIP was 29 years (interquartile range, 20 to 39 years). Acute pancreatitis was the most common initial presentation (n=17, 63%) while obstructive jaundice was present in only one patient. Ulcerative colitis (UC) was associated with type 2 AIP in 44.4% (12/27) of patients. Radiological pancreatic imaging such as delayed enhancement of diffusely enlarged pancreas, homogeneous enhancement of focal enlargement/mass, absent/minimal peripancreatic fat infiltration or fluid collection, and multifocal main pancreatic duct narrowings were helpful for differentiating type 2 AIP from gallstone pancreatitis. During follow-up (median, 32.3 months), two patients (2/25, 8%) experienced relapse. Conclusions In South Korea, type 2 AIP is not as rare as previously thought. Overall, the clinical profile of type 2 AIP was similar to that of Western countries. Type 2 AIP should be considered in young UC patients with acute pancreatitis of uncertain etiology.
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Affiliation(s)
- Dongwook Oh
- Department of Gastroenterology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Tae Jun Song
- Department of Gastroenterology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Sung-Hoon Moon
- Department of Internal Medicine, Hallym University Sacred Heart Hospital, Hallym University College of Medicine, Anyang, Korea
| | - Jin Hee Kim
- Departments of Radiology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Nam Joo Lee
- Departments of Radiology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Seung-Mo Hong
- Departments of Pathology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Joune Seup Lee
- Department of Gastroenterology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Seok Jung Jo
- Department of Gastroenterology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Dong Hui Cho
- Department of Gastroenterology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Do Hyun Park
- Department of Gastroenterology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Sang Soo Lee
- Department of Gastroenterology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Dong-Wan Seo
- Department of Gastroenterology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Sung Koo Lee
- Department of Gastroenterology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Myung-Hwan Kim
- Department of Gastroenterology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
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Noguchi K, Nakai Y, Mizuno S, Isayama H, Hirano K, Kanai S, Nakamura T, Uchino R, Takahara N, Kogure H, Tada M, Koike K. Insulin secretion improvement during steroid therapy for autoimmune pancreatitis according to the onset of diabetes mellitus. J Gastroenterol 2020; 55:198-204. [PMID: 31471733 DOI: 10.1007/s00535-019-01615-4] [Citation(s) in RCA: 13] [Impact Index Per Article: 2.6] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/08/2019] [Accepted: 08/12/2019] [Indexed: 02/04/2023]
Abstract
BACKGROUND Autoimmune pancreatitis (AIP) is frequently complicated by diabetes mellitus (DM), but DM associated with AIP is reported to improve after steroid therapy. The aim of this study is to investigate glucose intolerance during steroid therapy according to the onset of DM. METHODS Sixty-one patients who underwent steroid therapy for AIP were included into this study. We evaluated C peptide index (CPI), homeostasis model assessment for insulin resistance (HOMA-R), and the pancreatic diameter at AIP diagnosis and after 4 weeks, 1 year, and 2 years of steroid therapy. Patients were categorized into three groups according to DM onset: Pre-existing DM (pDM), concurrent DM (cDM), and non-DM (nDM). RESULTS Forty-three patients (71%) had DM: 15 pDM and 28 cDM. At AIP diagnosis, CPI was lower in patients with pDM (0.7, P = 0.007) and cDM (0.9, P = 0.018) than nDM (1.3). After 4 weeks of steroid therapy, CPI improved in cDM (P < 0.001) and in nDM (P = 0.021). After 2 years of steroid therapy, HOMA-R increased (2.1-3.0, P = 0.007) but CPI gradually improved (1.0-2.1, P = 0.004). DM improved in 23% of cDM, and 55% of insulin users in cDM discontinued using insulin. Pancreatic atrophy was seen in 30%, and was associated with DM. CONCLUSION DM in patients with AIP was associated with impaired insulin secretion rather than insulin resistance. Insulin secretion improved during steroid therapy for AIP in patients with concurrent DM. Thus, glucose intolerance can be an indication for AIP treatment.
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Affiliation(s)
- Kensaku Noguchi
- Department of Gastroenterology, Graduate School of Medicine, The University of Tokyo, Tokyo, Japan
| | - Yousuke Nakai
- Department of Gastroenterology, Graduate School of Medicine, The University of Tokyo, Tokyo, Japan.
| | - Suguru Mizuno
- Department of Gastroenterology, Graduate School of Medicine, The University of Tokyo, Tokyo, Japan
| | - Hiroyuki Isayama
- Department of Gastroenterology, Graduate School of Medicine, Juntendo University, Tokyo, Japan
| | - Kenji Hirano
- Department of Gastroenterology, Graduate School of Medicine, The University of Tokyo, Tokyo, Japan
| | - Sachiko Kanai
- Department of Gastroenterology, Graduate School of Medicine, The University of Tokyo, Tokyo, Japan
| | - Tomoka Nakamura
- Department of Gastroenterology, Graduate School of Medicine, The University of Tokyo, Tokyo, Japan
| | - Rie Uchino
- Department of Gastroenterology, Graduate School of Medicine, The University of Tokyo, Tokyo, Japan
| | - Naminatsu Takahara
- Department of Gastroenterology, Graduate School of Medicine, The University of Tokyo, Tokyo, Japan
| | - Hirofumi Kogure
- Department of Gastroenterology, Graduate School of Medicine, The University of Tokyo, Tokyo, Japan
| | - Minoru Tada
- Department of Gastroenterology, Graduate School of Medicine, The University of Tokyo, Tokyo, Japan
| | - Kazuhiko Koike
- Department of Gastroenterology, Graduate School of Medicine, The University of Tokyo, Tokyo, Japan
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Sanogo SD, Dicko MY, Soumaré G, Doumbia K, Sow H, Tounkara MS, Kassambara Y, Guindo H, Maiga A, Kondé A, Diarra A, Guindo I, Katilé D, Mallé O, Konate A, Diarra MT, Sanogo B, Maiga MY. Case Report: Autoimmune Pancreatitis: About a Case in Hepato-Gastroenterology Department of Mother-Child Hospital in Bamako/Mali. Health (London) 2020. [DOI: 10.4236/health.2020.1210099] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/20/2022]
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38
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Japanese Clinical Diagnostic Criteria for Autoimmune Pancreatitis, 2018: Revision of Japanese Clinical Diagnostic Criteria for Autoimmune Pancreatitis, 2011. Pancreas 2020; 49:e13-e14. [PMID: 31856100 PMCID: PMC6946098 DOI: 10.1097/mpa.0000000000001443] [Citation(s) in RCA: 77] [Impact Index Per Article: 15.4] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/12/2022]
Abstract
Supplemental digital content is available in the text.
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Li G, Liu T, Zheng J, Kang W, Xu J, Gao Z, Ma J. Untypical autoimmune pancreatitis and pancreatic cancer: differential diagnosis experiences extracted from misdiagnose of two cases. Orphanet J Rare Dis 2019; 14:245. [PMID: 31699117 PMCID: PMC6839088 DOI: 10.1186/s13023-019-1217-z] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/03/2019] [Accepted: 10/03/2019] [Indexed: 01/12/2023] Open
Abstract
BACKGROUND Differentiation between pancreatic cancer (PC) and focal form of autoimmune pancreatitis (AIP) is very challenging, with similar clinical presentations, laboratory results and morphologic imagings of US, CT, EUS, MRI, ERCP, PET-CT. Even serum IgG4 and biopsy sometimes cannot give clear-cut differential accurate diagnostis. Considering the totally different management strategy of the two diseases, accurate diagnostic value is urgently needed to remind the clinicians of the rare diagnosis of untypical AIP among frequent PC-suspected patients. RESULTS We present 2 laparotomy cases of AIP that had a high similar characteristic to PC and retrospectively extracted the warning signs that may help select untypical AIP in PC-suspected patients. CONCLUSIONS We find that mild fluctuating jaundice with abdominal pain, young age, tumor marker of TPS, TPA and diverse results between variable radiological tests can help to differentiate AIP mass from PC, through retrospectively analyzing work-up process of AIP in two patients who underwent laparotomy for suspected PC.
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Affiliation(s)
- Gaopeng Li
- Department of general surgery, Shanxi Cancer Hospital, Shanxi Medical University, Taiyuan, Shanxi Province China
| | - Ting Liu
- Department of general surgery, Shanxi Cancer Hospital, Shanxi Medical University, Taiyuan, Shanxi Province China
| | - Jian Zheng
- Department of critical care medicine, The first hospital of Shanxi medical University, The Hospital of Shanxi University of Traditional Chinese Medicine, Taiyuan, Shanxi Province China
| | - Wenqin Kang
- Department of general surgery, The Hospital of Shanxi University of Traditional Chinese Medicine, Taiyuan, Shanxi Province China
| | - Jun Xu
- Department of general surgery, Shanxi Dayi Hospital, Shanxi academy of Medical science, Taiyuan, Shanxi Province China
| | - Zefeng Gao
- Department of general surgery, Shanxi Cancer Hospital, Shanxi Medical University, Taiyuan, Shanxi Province China
| | - Jinfeng Ma
- Department of general surgery, Shanxi Cancer Hospital, Shanxi Medical University, Taiyuan, Shanxi Province China
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Abstract
Autoimmune pancreatitis (AIP) is an entity that has been recognized since 1961. Prior to the discovery of elevated serum IgG4 as a useful biomarker for its diagnosis, Dr. Yoshida in 1995 first described the entity of AIP, which in retrospect closely resembles the current concept of type 1 AIP. Since the discovery of IgG4 as a biomarker (the IgG4-era), a novel concept of IgG4-related disease (IgG4-RD) has been accepted as being comprised of two subtypes of AIP: type 1 defined as the pancreatic manifestation of IgG4-RD, and type 2 characterized by granulocytic epithelial lesions. The characteristic features of type 1 AIP are increased serum IgG4 levels, lymphoplasmacytic sclerosing pancreatitis (abundant infiltration of IgG4+ plasmocytes and lymphocytes, storiform fibrosis, and obliterative phlebitis), extrapancreatic manifestations of IgG4-RD (e.g., sclerosing cholangitis, sclerosing sialadenitis, retroperitoneal fibrosis), and steroid responsiveness. These entities can be differentiated from mimickers by a combination of serum IgG4 level, imaging features, and histopathological findings. The current first-line therapy is corticosteroids, or rituximab in high-risk patients with steroid intolerance. Although relapse rates are high, treatment of relapsed disease remains experimental.
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Affiliation(s)
- Kazuichi Okazaki
- Division of Gastroenterology and Hepatology, The Third Department of Internal Medicine, Kansai Medical University, Shinmachi, Hirakata, Osaka, 573-1197, Japan.
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Hegade VS, Sheridan MB, Huggett MT. Diagnosis and management of IgG4-related disease. Frontline Gastroenterol 2019; 10:275-283. [PMID: 31288262 PMCID: PMC6583577 DOI: 10.1136/flgastro-2018-101001] [Citation(s) in RCA: 22] [Impact Index Per Article: 3.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/08/2018] [Revised: 09/09/2018] [Accepted: 09/29/2018] [Indexed: 02/04/2023] Open
Abstract
IgG subclass 4-related disease (IgG4-RD) is a rare but increasingly recognised fibroinflammatory condition known to affect multiple organs. IgG4-RD is characterised by unique histological features of lymphoplasmacytic infiltration, storiform fibrosis and obliterative phlebitis. In this review we describe the pancreaticobiliary manifestations of IgG4-RD, with particular emphasis on type 1 autoimmune pancreatitis (AIP) and IgG4-related sclerosing cholangitis (IgG4-SC). AIP and IgG4-SC can pose diagnostic challenges to the clinician as they may mimic pancreatic cancer and primary sclerosing cholangitis, respectively. We discuss current knowledge, clinical diagnostic criteria and recent advances and summarise the evidence base for current therapeutic approaches for AIP and IgG4-SC.
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Affiliation(s)
- Vinod S Hegade
- Department of Gastroenterology, St James University Hospital, Leeds, UK
| | - Maria B Sheridan
- Department of Radiology, St James University Hospital, Leeds, UK
| | - Matthew T Huggett
- Department of Gastroenterology, St James University Hospital, Leeds, UK
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Tacelli M, Celsa C, Magro B, Barresi L, Guastella S, Capurso G, Frulloni L, Cabibbo G, Cammà C. Risk Factors for Rate of Relapse and Effects of Steroid Maintenance Therapy in Patients With Autoimmune Pancreatitis: Systematic Review and Meta-analysis. Clin Gastroenterol Hepatol 2019; 17:1061-1072.e8. [PMID: 30312787 DOI: 10.1016/j.cgh.2018.09.051] [Citation(s) in RCA: 36] [Impact Index Per Article: 6.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/22/2018] [Revised: 09/20/2018] [Accepted: 09/22/2018] [Indexed: 02/07/2023]
Abstract
BACKGROUND & AIMS Risk for relapse after induction of remission with steroid therapy has been studied extensively in patients with autoimmune pancreatitis (AIP), but findings have been equivocal. We performed a systematic review and meta-analysis to estimate the relapse rate of AIP after initial remission after steroid treatment and to identify factors associated with relapse. METHODS Three reviewers searched MEDLINE, SCOPUS, and EMBASE until July 2018 to identify studies on rate of relapse of AIP after induction of remission with steroid therapy. A pooled estimate was calculated using the DerSimonian and Laird method for a random-effects model. This study was conducted in accordance with Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines. RESULTS Thirty-six studies met the inclusion criteria for meta-analysis. The median follow-up time was 40.8 months. Fifty-two percent of patients were classified as having type 1 AIP. The pooled estimate of relapse rate was 33% (95% CI, 30%-37%). A higher proportion of patients with type 1 AIP had a relapse compared with patients with type 2 AIP (37.5% vs 15.9%; P < .001). We found significant heterogeneity among studies (P < .01). Long-term maintenance therapy with steroids and study quality were associated independently with AIP relapse, after we adjusted for year of publication by multivariate meta-regression. CONCLUSIONS In a systematic review and meta-analysis, we found that a large proportion of patients with AIP treated successfully with steroid induction therapy had a relapse (33%)-particularly patients with type 1 AIP (37%). Maintenance steroid therapy lasting longer than 1 year could reduce risk of relapse. However, the data characterizing relapse rates are of limited quality, indicating the need for randomized controlled trials and new immunosuppressive drugs.
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Affiliation(s)
- Matteo Tacelli
- Section of Gastroenterology, Biomedical Department of Internal and Specialized Medicine, University of Palermo, Palermo, Italy
| | - Ciro Celsa
- Section of Gastroenterology, Biomedical Department of Internal and Specialized Medicine, University of Palermo, Palermo, Italy
| | - Bianca Magro
- Section of Gastroenterology, Biomedical Department of Internal and Specialized Medicine, University of Palermo, Palermo, Italy
| | - Luca Barresi
- Endoscopy Service, Department of Diagnostic and Therapeutic Services, Istituto di Ricovero e Cura a Carattere Scientifico-Istituto Mediterraneo per i Trapianti e Terapie ad alta Specializzazione (Mediterranean Institute for Transplantation and Highly Specialized Therapies), Palermo, Italy
| | - Salvatore Guastella
- Section of Gastroenterology, Biomedical Department of Internal and Specialized Medicine, University of Palermo, Palermo, Italy
| | - Gabriele Capurso
- PancreatoBiliary Endoscopy and Endoscopic Ultrasonography Division, Pancreas Translational and Clinical Research Center, San Raffaele Scientific Institute (Istituto di Ricovero e Cura a Carattere Scientifico), Vita Salute San Raffaele University, Milan, Italy
| | - Luca Frulloni
- Department of Medicine, Pancreas Institute, University of Verona, Verona, Italy
| | - Giuseppe Cabibbo
- Section of Gastroenterology, Biomedical Department of Internal and Specialized Medicine, University of Palermo, Palermo, Italy
| | - Calogero Cammà
- Section of Gastroenterology, Biomedical Department of Internal and Specialized Medicine, University of Palermo, Palermo, Italy.
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Kwon JH, Kim JH, Kim SY, Byun JH, Kim HJ, Lee MG, Lee SS. Differentiating focal autoimmune pancreatitis and pancreatic ductal adenocarcinoma: contrast-enhanced MRI with special emphasis on the arterial phase. Eur Radiol 2019; 29:5763-5771. [PMID: 31028441 DOI: 10.1007/s00330-019-06200-0] [Citation(s) in RCA: 25] [Impact Index Per Article: 4.2] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/20/2018] [Revised: 03/21/2019] [Accepted: 03/25/2019] [Indexed: 12/18/2022]
Abstract
OBJECTIVES To compare focal-type autoimmune pancreatitis (AIP) and pancreatic ductal adenocarcinoma (PDA) using contrast-enhanced MR imaging (CE-MRI), and to assess diagnostic performance of the lesion contrast at arterial phase (AP) (ContrastAP) for differentiating between the two diseases. METHODS Thirty-six patients with focal-type AIP and 72 patients with PDA were included. All included patients underwent CE-MRI with triple phases. The signal intensity (SI) of the mass and normal pancreas was measured at each phase, and the lesion contrast (SIpancreas/SImass) was compared between AIP and PDA groups. The sensitivity and specificity of ContrastAP using an optimal cutoff point were compared with those of key imaging features specific to AIP and PDA. RESULTS The lesion contrast differed significantly between AIP and PDA groups at all phases of CE-MRI; the maximum difference was observed at AP. For AIP, the sensitivity (94.4%) and specificity (87.5%) of ContrastAP (cutoff ≤ 1.41) were comparable or significantly higher than those of all key imaging features (sensitivity, 38.9-88.9%; specificity, 48.6-95.8%), except for the halo sign. For PDA, the sensitivity (87.5%) and specificity (94.4%) of ContrastAP (cutoff > 1.41) were comparable or significantly higher than those of all key imaging features (sensitivity, 40.3-68.1%; specificity, 72.2-94.4%), except for the discrete mass. CONCLUSIONS Quantitative analysis of the lesion contrast using CE-MRI, particularly at AP, was helpful to differentiate focal-type AIP from PDA. The diagnostic performance of ContrastAP was mostly comparable or higher than those of the key imaging features. KEY POINTS • Diagnosis of focal-type AIP vs. PDA using imaging techniques is extremely challenging. • Lesion contrast in the arterial-phase MRI differs significantly between focal-type AIP and PDA. • Quantitative analysis of lesion contrast using CE-MRI, particularly at the arterial phase, is helpful to differentiate focal-type AIP from PDA.
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Affiliation(s)
- Ji Hye Kwon
- Department of Radiology and Research Institute of Radiology, University of Ulsan College of Medicine, Asan Medical Center, 88 Olympic-ro 43-gil, Songpa-Gu, Seoul, 05505, South Korea
| | - Jin Hee Kim
- Department of Radiology and Research Institute of Radiology, University of Ulsan College of Medicine, Asan Medical Center, 88 Olympic-ro 43-gil, Songpa-Gu, Seoul, 05505, South Korea.
| | - So Yeon Kim
- Department of Radiology and Research Institute of Radiology, University of Ulsan College of Medicine, Asan Medical Center, 88 Olympic-ro 43-gil, Songpa-Gu, Seoul, 05505, South Korea
| | - Jae Ho Byun
- Department of Radiology and Research Institute of Radiology, University of Ulsan College of Medicine, Asan Medical Center, 88 Olympic-ro 43-gil, Songpa-Gu, Seoul, 05505, South Korea
| | - Hyoung Jung Kim
- Department of Radiology and Research Institute of Radiology, University of Ulsan College of Medicine, Asan Medical Center, 88 Olympic-ro 43-gil, Songpa-Gu, Seoul, 05505, South Korea
| | - Moon-Gyu Lee
- Department of Radiology and Research Institute of Radiology, University of Ulsan College of Medicine, Asan Medical Center, 88 Olympic-ro 43-gil, Songpa-Gu, Seoul, 05505, South Korea
| | - Seung Soo Lee
- Department of Radiology and Research Institute of Radiology, University of Ulsan College of Medicine, Asan Medical Center, 88 Olympic-ro 43-gil, Songpa-Gu, Seoul, 05505, South Korea
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Zeng XP, Liu TT, Hao L, Xin L, Wang T, He L, Pan J, Wang D, Bi YW, Ji JT, Liao Z, Du TT, Lin JH, Zhang D, Guo HL, Chen H, Zou WB, Li BR, Cong ZJ, Wang LS, Xu ZL, Xie T, Liu MH, Deng AM, Li ZS, Hu LH. Autoantibody detection is not recommended for chronic pancreatitis: a cross-sectional Study of 557 patients. BMC Gastroenterol 2019; 19:31. [PMID: 30764766 PMCID: PMC6376735 DOI: 10.1186/s12876-019-0947-7] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/19/2018] [Accepted: 01/30/2019] [Indexed: 12/15/2022] Open
Abstract
BACKGROUND Autoimmune factor was regarded as one of the risk factors in the pathogenesis of chronic pancreatitis (CP), especially for autoimmune pancreatitis (AIP). However, whether autoimmune factor plays a role in non-AIP CP or not was unknown. METHODS Hospitalized patients with non-AIP CP from January 2010 to October 2016 were detected for 22 autoantibodies at the time of hospital admission. Autoantibodies with frequency > 0.5% were enrolled to calculate the frequency in historial healthy controls through literature search in PubMed. Differentially expressed autoantibodies were determined between patients and historial healthy controls, and related factors were identified by multivariate logistic regression analysis. RESULTS In a total of 557 patients, 113 cases were detected with 19 kinds of positive autoantibodies, among them anti-β2-glycoprotein I (β2-GPI) antibody was most frequent (9.16%). Compared with historial healthy controls, the frequencies of serum β2-GPI and anti SS-B antibody in patients were significantly higher, while frequencies of anti-smooth muscle antibody and anticardiolipin antibody were significantly lower (all P < 0.05). Multivariate logistic regression analysis result showed that diabetes mellitus (OR = 2.515) and common bile duct stricture (OR = 2.844) were the risk factors of positive β2-GPI antibody in patients while diabetes mellitus in first-/second-/third-degree relatives (OR = 0.266) was the protective factor. There were no related factors for other three differentially expressed autoantibodies. CONCLUSIONS Four autoantibodies were expressed differentially between patients with non-AIP CP and historial healthy controls. Due to limited significance for diagnosis and treatment of chronic pancreatitis, autoantibodies detection is not recommended conventionally unless suspected of AIP.
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Affiliation(s)
- Xiang-Peng Zeng
- Department of Gastroenterology, Gongli Hospital, The Second Military Medical University, Shanghai, China
- Department of Gastroenterology, Changhai Hospital, The Second Military Medical University, Shanghai, China
| | - Ting-Ting Liu
- Department of Laboratory Medicine, Changhai Hospital, The Second Military Medical University, Shanghai, China
| | - Lu Hao
- Department of Gastroenterology, Hainan Branch of Chinese PLA General Hospital, Sanya, China
- Digestive Endoscopy Center, Changhai Hospital, The Second Military Medical University, Shanghai, China
| | - Lei Xin
- Department of Gastroenterology, Gongli Hospital, The Second Military Medical University, Shanghai, China
- Department of Gastroenterology, Changhai Hospital, The Second Military Medical University, Shanghai, China
- Digestive Endoscopy Center, Changhai Hospital, The Second Military Medical University, Shanghai, China
| | - Teng Wang
- Department of Gastroenterology, Changhai Hospital, The Second Military Medical University, Shanghai, China
- Digestive Endoscopy Center, Changhai Hospital, The Second Military Medical University, Shanghai, China
| | - Lin He
- Department of Gastroenterology, Changhai Hospital, The Second Military Medical University, Shanghai, China
| | - Jun Pan
- Department of Gastroenterology, Changhai Hospital, The Second Military Medical University, Shanghai, China
- Digestive Endoscopy Center, Changhai Hospital, The Second Military Medical University, Shanghai, China
| | - Dan Wang
- Department of Gastroenterology, Changhai Hospital, The Second Military Medical University, Shanghai, China
| | - Ya-Wei Bi
- Department of Gastroenterology, Changhai Hospital, The Second Military Medical University, Shanghai, China
| | - Jun-Tao Ji
- Department of Gastroenterology, Changhai Hospital, The Second Military Medical University, Shanghai, China
| | - Zhuan Liao
- Department of Gastroenterology, Gongli Hospital, The Second Military Medical University, Shanghai, China
- Department of Gastroenterology, Changhai Hospital, The Second Military Medical University, Shanghai, China
- Digestive Endoscopy Center, Changhai Hospital, The Second Military Medical University, Shanghai, China
| | - Ting-Ting Du
- Department of Gastroenterology, Changhai Hospital, The Second Military Medical University, Shanghai, China
| | - Jin-Huan Lin
- Department of Gastroenterology, Changhai Hospital, The Second Military Medical University, Shanghai, China
| | - Di Zhang
- Department of Gastroenterology, Changhai Hospital, The Second Military Medical University, Shanghai, China
- Digestive Endoscopy Center, Changhai Hospital, The Second Military Medical University, Shanghai, China
| | - Hong-Lei Guo
- Department of Gastroenterology, Changhai Hospital, The Second Military Medical University, Shanghai, China
| | - Hui Chen
- Department of Gastroenterology, Gongli Hospital, The Second Military Medical University, Shanghai, China
- Department of Gastroenterology, Changhai Hospital, The Second Military Medical University, Shanghai, China
- Digestive Endoscopy Center, Changhai Hospital, The Second Military Medical University, Shanghai, China
| | - Wen-Bin Zou
- Department of Gastroenterology, Changhai Hospital, The Second Military Medical University, Shanghai, China
- Digestive Endoscopy Center, Changhai Hospital, The Second Military Medical University, Shanghai, China
| | - Bai-Rong Li
- Department of Gastroenterology, Air Force General Hospital, Beijing, China
| | - Zhi-Jie Cong
- Department of General Surgery, Renji Hospital, Shanghai Jiaotong University, Shanghai, China
| | - Li-Sheng Wang
- Department of Gastroenterology, Shenzhen People’s Hospital, Shenzhen, China
| | - Zheng-Lei Xu
- Department of Gastroenterology, Shenzhen People’s Hospital, Shenzhen, China
| | - Ting Xie
- Department of Gastroenterology, Zhongda Hospital, Southeast University, Nanjing, China
| | - Ming-Hao Liu
- Department of Gastroenterology, PLA Rocket Force Characteristic Medical Center, Beijing, China
| | - An-Mei Deng
- Department of Laboratory Medicine, Changhai Hospital, The Second Military Medical University, Shanghai, China
| | - Zhao-Shen Li
- Department of Gastroenterology, Gongli Hospital, The Second Military Medical University, Shanghai, China
- Department of Gastroenterology, Changhai Hospital, The Second Military Medical University, Shanghai, China
- Digestive Endoscopy Center, Changhai Hospital, The Second Military Medical University, Shanghai, China
| | - Liang-Hao Hu
- Department of Gastroenterology, Gongli Hospital, The Second Military Medical University, Shanghai, China
- Department of Gastroenterology, Changhai Hospital, The Second Military Medical University, Shanghai, China
- Digestive Endoscopy Center, Changhai Hospital, The Second Military Medical University, Shanghai, China
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Cao L, Wang Y, Wang J, Guo Q, Chen Q, Wu X, Tang SJ, Cheng B. The role of EUS-guided fine needle aspiration in autoimmune pancreatitis: a single center prospective study. Scand J Gastroenterol 2018; 53:1604-1610. [PMID: 30422724 DOI: 10.1080/00365521.2018.1534137] [Citation(s) in RCA: 18] [Impact Index Per Article: 2.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/04/2023]
Abstract
OBJECTIVES Histopathological examination is pivotal in diagnosing autoimmune pancreatitis (AIP). The usefulness of EUS-guided fine needle aspiration (EUS-FNA) in diagnosing AIP remains controversial worldwide. The authors conducted this study to evaluate the efficacy of EUS-FNA for AIP diagnosis using a 22-gauge needle. METHODS Between January 2013 and May 2017, 37 patients had imaging studies suggestive of AIP at Tongji Hospital, and 27 patients of them were enrolled in this study. Tissue specimens acquired through EUS-FNA were analyzed for periductal lymphoplasmacytic infiltrate (LPI), storiform fibrosis (SF), obliterative phlebitis (OP) and immunoglobulin G4 (IgG4)-positive plasma cell counts. Clinical Trials.gov no: TJ-C20121220. RESULTS LPI and SF were present in 18 (66.67%) and 18 (66.67%) of 27 patients, respectively. Abundant IgG4-positive plasmacyte infiltration >10/high-power field (HPF) was detected in 8 of 27 patients (29.63%). OP and the characteristic findings of idiopathic duct-centric chronic pancreatitis (IDCP) and granulocytic epithelial lesion (GEL) were not detected in this study. According to the International Consensus Diagnostic Criteria (ICDC) for AIP, 5 and 12 of 27 patients were assessed as having level 1 and level 2 histological findings, respectively, suggesting that 17 of 27 patients (62.96%) had lymphoplasmacytic sclerosing pancreatitis (LPSP) based on the ICDC. CONCLUSIONS In 92.6% of patients, pancreatic tissues with >5 HPFs were obtained by EUS-FNA using a 22-G needle. In 63% of patients, histology was evaluated to be ≥ level 2 according to the ICDC. The study indicates that EUS-FNA with a 22-G needle is valuable in the histopathological diagnosis of AIP.
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Affiliation(s)
- Li Cao
- a Department of Gastroenterology and Hepatology, Tongji Hospital, Tongji Medical College , Huazhong University of Science and Technology , Wuhan , China
| | - Yun Wang
- a Department of Gastroenterology and Hepatology, Tongji Hospital, Tongji Medical College , Huazhong University of Science and Technology , Wuhan , China
| | - Jinlin Wang
- a Department of Gastroenterology and Hepatology, Tongji Hospital, Tongji Medical College , Huazhong University of Science and Technology , Wuhan , China
| | - Qiaozhen Guo
- a Department of Gastroenterology and Hepatology, Tongji Hospital, Tongji Medical College , Huazhong University of Science and Technology , Wuhan , China
| | - Qian Chen
- a Department of Gastroenterology and Hepatology, Tongji Hospital, Tongji Medical College , Huazhong University of Science and Technology , Wuhan , China
| | - Xiaoli Wu
- a Department of Gastroenterology and Hepatology, Tongji Hospital, Tongji Medical College , Huazhong University of Science and Technology , Wuhan , China
| | - Shou-Jiang Tang
- b Division of Digestive Diseases, Department of Medicine , University of Mississippi Medical Center , Jackson , MS , USA
| | - Bin Cheng
- a Department of Gastroenterology and Hepatology, Tongji Hospital, Tongji Medical College , Huazhong University of Science and Technology , Wuhan , China
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Vujasinovic M, Valente R, Maier P, von Beckerath V, Haas SL, Arnelo U, Del Chiaro M, Kartalis N, Pozzi-Mucelli RM, Fernandez-Moro C, Verbeke CS, Yu J, Ye W, Löhr JM. Diagnosis, treatment and long-term outcome of autoimmune pancreatitis in Sweden. Pancreatology 2018; 18:900-904. [PMID: 30236651 DOI: 10.1016/j.pan.2018.09.003] [Citation(s) in RCA: 42] [Impact Index Per Article: 6.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/16/2018] [Revised: 09/05/2018] [Accepted: 09/12/2018] [Indexed: 02/07/2023]
Abstract
INTRODUCTION Autoimmune pancreatitis (AIP) is a pancreatic inflammatory process characterized by a strong inflammatory cell infiltration and two histopathologically distinct subtypes: type 1 and type 2. Diagnosis is often challenging and requires a combination of clinical, laboratory and imaging data. AIP can mimic pancreatic tumours leading to unnecessary resections if not correctly diagnosed. Short- and long-term outcomes of AIP have been poorly investigated so far and no large series have been previously reported from Sweden. METHODS A single-centre, retrospective, cohort study of patients with histologically confirmed or highly probable diagnosis of AIP according to ICDC criteria. Demographic, clinical and radiological characteristics, type of treatment and its outcomes were collected and analysed. RESULTS Seventy-one patients with AIP (87% with type 1), were evaluated at Karolinska University Hospital between 2004 and 2018; 49% males, mean age 49 years (range 44-53). Among them, 28% were histologically confirmed, 35% presented with jaundice, 22% with acute pancreatitis, 39% had non-specific symptoms such as weight loss or abdominal pain, 84% showed other organ involvement (OOI). Radiologically, 76% showed a focal pancreatic enlargement, 27% diffuse enlargement, 27% signs of acute pancreatitis and 10% of chronic pancreatitis. Overall, 58 patients (81%) underwent treatment with different medications: 46 (79%) cortisone, 7 (12%) azathioprine, 5 (8%) other immunosuppressive drugs. Twenty-six (36%) underwent biliary stenting and 12 (16%) were given surgery. In total, 47% of patients developed pancreatic exocrine insufficiency (PEI), of whom 76% had a severe form (faecal elastase-1 < 100 μg/g) and 21% of patients developed diabetes mellitus (pancreatic endocrine insufficiency), of whom 73% required insulin. CONCLUSIONS AIP is a challenging disease for diagnosis and treatment. Cortisone treatment is generally successful and provides clinical remission in the large majority of patients (>90%). In the further course of the disease, a considerable number of patients develop PEI and diabetes. Only one-quarter of patients exhibit on imaging the characteristic "sausage-like" pancreas (diffuse enlargement), approximately three-quarters had a focal mass that could be misdiagnosed as pancreatic malignancy.
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Affiliation(s)
- Miroslav Vujasinovic
- Department for Digestive Diseases, Karolinska University Hospital, Stockholm, Sweden
| | - Roberto Valente
- Department for Digestive Diseases, Karolinska University Hospital, Stockholm, Sweden; Department for Digestive Diseases, Sapienza University of Rome, Rome, Italy
| | - Pia Maier
- Department for Digestive Diseases, Karolinska University Hospital, Stockholm, Sweden
| | | | - Stephan L Haas
- Department for Digestive Diseases, Karolinska University Hospital, Stockholm, Sweden
| | - Urban Arnelo
- Department for Digestive Diseases, Karolinska University Hospital, Stockholm, Sweden
| | - Marco Del Chiaro
- Department for Digestive Diseases, Karolinska University Hospital, Stockholm, Sweden
| | - Nikolaos Kartalis
- Department of Abdominal Radiology, Karolinska University Hospital, Stockholm, Sweden; Department of Clinical Science, Intervention, and Technology (CLINTEC), Karolinska Institute, Stockholm, Sweden
| | - Raffaella Maria Pozzi-Mucelli
- Department of Abdominal Radiology, Karolinska University Hospital, Stockholm, Sweden; Department of Clinical Science, Intervention, and Technology (CLINTEC), Karolinska Institute, Stockholm, Sweden
| | | | - Caroline Sophie Verbeke
- Department of Pathology, Karolinska University Hospital, Stockholm, Sweden; Department of Pathology, University of Oslo, Oslo, Norway
| | - Jingru Yu
- Department of Medical Epidemiology and Biostatistics, Karolinska Institute, Stockholm, Sweden
| | - Weimin Ye
- Department of Medical Epidemiology and Biostatistics, Karolinska Institute, Stockholm, Sweden
| | - J Matthias Löhr
- Department for Digestive Diseases, Karolinska University Hospital, Stockholm, Sweden; Department of Clinical Science, Intervention, and Technology (CLINTEC), Karolinska Institute, Stockholm, Sweden.
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Shi Y, Cang L, Zhang X, Cai X, Wang X, Ji R, Wang M, Hong Y. The use of magnetic resonance elastography in differentiating autoimmune pancreatitis from pancreatic ductal adenocarcinoma: A preliminary study. Eur J Radiol 2018; 108:13-20. [PMID: 30396645 DOI: 10.1016/j.ejrad.2018.09.001] [Citation(s) in RCA: 24] [Impact Index Per Article: 3.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/26/2018] [Revised: 08/30/2018] [Accepted: 09/03/2018] [Indexed: 12/12/2022]
Abstract
PURPOSE To assess the value of magnetic resonance elastography (MRE) in patients with autoimmune pancreatitis (AIP) and in the differentiation of AIP from pancreatic ductal adenocarcinoma (PDAC). METHOD AND MATERIALS This prospective study included 14 AIP patients, 26 PDAC patients, and 14 healthy volunteers. All participants underwent pancreatic MRE (40-Hz; 3 T scanner) at enrollment, and 7 AIP patients underwent a second MRE after initiation of steroid therapy. Pancreatic stiffness values were obtained by MRE and a new logistic regression model (the calculated Rad score) was used to combine pancreatic stiffness and the distribution and shape of high-stiffness areas for differentiation of AIP and PDAC. The area under the curve (AUC) was calculated for all parameters using receiver operating characteristic (ROC) analysis. RESULTS Pancreatic stiffness was significantly higher (2.67 kPa [interquartile range, 2.24-3.56 kPa]) in AIP than in healthy pancreas (1.24 kPa [1.18-1.24 kPa]) and significantly lower in AIP than in PDAC (3.78 kPa [3.22-5.11 kPa]; both P < 0.05). Diffuse (n = 4 vs 1; P = 0.043) and multiple (n = 3 vs 0; P = 0.037) lesions were more common in AIP, while solitary (n = 25 vs 7; P = 0.001) and nodular lesions (n = 18 vs 2; P = 0.002) were more frequent in PDAC. Rad scores outperformed individual imaging parameters in distinguishing AIP from PDAC (AUC, 0.948 vs 0.607 to 0.782; all P < 0.05), with 84.6% specificity and 92.9% sensitivity. Pancreatic stiffness in AIP decreased significantly, from 2.66 kPa [2.29 to 3.05 kPa] to 1.55 kPa [1.43 to 1.67 kPa] (P = 0.016), during treatment. CONCLUSIONS MRE shows promise as a quantitative imaging method for differentiating AIP from PDAC and for monitoring the treatment response in AIP.
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Affiliation(s)
- Yu Shi
- Department of Radiology, Shengjing Hospital of China Medical University, Shenyang, PR China
| | - Lizhuo Cang
- Department of Radiology, Shengjing Hospital of China Medical University, Shenyang, PR China
| | - Xianyi Zhang
- Department of Radiology, Shengjing Hospital of China Medical University, Shenyang, PR China
| | - Xiaoli Cai
- Department of Radiology, Shengjing Hospital of China Medical University, Shenyang, PR China
| | | | - Ruoyun Ji
- Department of Radiology, Shengjing Hospital of China Medical University, Shenyang, PR China
| | - Min Wang
- Department of Radiology, Shengjing Hospital of China Medical University, Shenyang, PR China
| | - Yang Hong
- Department of Neurosurgery, Shengjing Hospital, China Medical University, Shenyang, PR China.
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Suk Lee Y, Kim NH, Hyuk Son J, Wook Kim J, Ki Bae W, Kim KA, Sung Lee J. Type 2 Autoimmune Pancreatitis with Crohn's Disease. Intern Med 2018; 57:2957-2962. [PMID: 29526939 PMCID: PMC6232013 DOI: 10.2169/internalmedicine.0213-17] [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] [Received: 09/13/2017] [Accepted: 01/05/2018] [Indexed: 12/11/2022] Open
Abstract
Autoimmune pancreatitis (AIP) is a distinct subtype of pancreatitis, which is classified into type 1 and 2 based on the clinicopathological features. According to the international consensus diagnostic criteria, pancreas resection or core biopsy specimens are recommended to make an accurate histological evaluation. However, the usefulness of endoscopic ultrasonography (EUS) guided fine needle aspiration (FNA) for histological evaluation has also been reported. Furthermore, the simultaneous presentation of type 2 AIP and Crohn's disease (CD) is very rare, especially in the Asian population. Therefore, we herein report a case of type 2 AIP with CD, which was diagnosed using EUS guided FNA with a 22-gauge needle.
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Affiliation(s)
- Yoon Suk Lee
- Department of Internal Medicine, Inje University College of Medicine, Ilsan Paik Hospital, Korea
| | - Nam-Hoon Kim
- Department of Internal Medicine, Inje University College of Medicine, Ilsan Paik Hospital, Korea
| | - Jun Hyuk Son
- Department of Internal Medicine, Inje University College of Medicine, Ilsan Paik Hospital, Korea
| | - Jung Wook Kim
- Department of Internal Medicine, Inje University College of Medicine, Ilsan Paik Hospital, Korea
| | - Won Ki Bae
- Department of Internal Medicine, Inje University College of Medicine, Ilsan Paik Hospital, Korea
| | - Kyung-Ah Kim
- Department of Internal Medicine, Inje University College of Medicine, Ilsan Paik Hospital, Korea
| | - June Sung Lee
- Department of Internal Medicine, Inje University College of Medicine, Ilsan Paik Hospital, Korea
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Abstract
Type 1 autoimmune pancreatitis (AIP) is an IgG-4-related systemic disease that can manifest as a pancreatic disorder or another disorder of presumed autoimmune origin. Type 2 disease is typically characterized by absent IgG-4-positive cells. As patients often present with acute pancreatitis, obstructive jaundice, or pancreatic mass, it is imperative to exclude malignancy, a more common diagnosis. AIP may respond to corticosteroids, and has a strong association with other immune-mediated diseases. Recent literature suggests the benefit of immune-modulating therapy, including rituximab, although no consensus exists. This review covers the essentials of diagnosis, but focuses primarily on management of AIP.
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Affiliation(s)
- Kamraan Madhani
- Department of Medicine, Yale School of Medicine, 333 Cedar Street, New Haven, CT 06510, USA; Department of Medicine, Waterbury Internal Medicine Residency Program, Waterbury Hospital, Yale New Haven Hospital, Main 3, 64 Robbins Street, Waterbury, CT 06708, USA
| | - James J Farrell
- Section of Digestive Diseases, Yale University School of Medicine, Yale Center for Pancreatic Disease, Yale University, LMP 1080, 15 York Street, New Haven, CT 06510, USA.
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Xin L, Meng QQ, Hu LH, Lin H, Pan J, Hao L, Liu PP, Qian W, Wang LW, Li ZS. Prediction and Management for Relapse of Type 1 Autoimmune Pancreatitis After Initial Steroid Treatment: A Long-Term Follow-up From China. Pancreas 2018; 47:1110-1114. [PMID: 30142120 DOI: 10.1097/mpa.0000000000001147] [Citation(s) in RCA: 9] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/11/2022]
Abstract
OBJECTIVES This study aimed to investigate the frequency, predictors, and management of relapse of type 1 autoimmune pancreatitis (AIP) after steroid treatment. METHODS One hundred one patients with type 1 AIP receiving steroid as initial treatment were enrolled. Predictors were identified by multivariate analysis using Cox proportional hazards model. Hazard ratios (HRs) and 95% confidence intervals (95% CIs) were calculated. RESULTS Thirty-nine (41.1%) patients experienced relapse after steroid treatment during a median follow-up of 40 months. The cumulative rate of relapse reached a plateau at 57.6% (95% CI, 50.3%-64.9%) at 59 months. Without maintenance therapy (HR, 4.53; 95% CI, 1.35-15.17) and proximal bile duct involvement (HR, 2.10; 95% CI, 1.07-4.14) were identified as risk factors. Repeated steroid treatment with the same initial dose but longer induction and tapering duration was given to 87.2% patients with relapse and obtained response (97.1%) during a short-term follow-up (median, 19 months). CONCLUSIONS Relapses are common in type 1 AIP after steroid treatment. Without maintenance therapy and proximal bile duct involvement were risk factors. Repeated steroid treatment with longer duration is effective for relapse.
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Affiliation(s)
- Lei Xin
- From the Digestive Endoscopy Center, Department of Gastroenterology, Changhai Hospital, Second Military Medical University, Shanghai, China
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