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Coburn S, Trojanowski PJ, Vagadori J, Hinds P, Germone M, Liu E, Streisand R, Bost J. Development and Validation of a Pediatric Celiac Disease-Specific Quality of Life Measure. Am J Gastroenterol 2025; 120:1116-1126. [PMID: 39401060 PMCID: PMC11994830 DOI: 10.14309/ajg.0000000000003132] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/17/2024] [Accepted: 10/03/2024] [Indexed: 10/15/2024]
Abstract
INTRODUCTION Quality of life (QOL) is critical in screening and management of chronic medical conditions, including celiac disease (CD). The aim of this project was to develop a CD-specific pediatric QOL measure (Celiac Disease Life Inventory of Family Experiences [CDLIFE]) with parallel self-report and parent-report forms by generating items through concept elicitation interviews, iterative refinement using cognitive debriefing interviews, and evaluating its psychometric properties and validity. METHODS Concept elicitation interviews were conducted to develop items (9 youth ages 8-19 years with CD; 10 parents of youth with CD), followed by cognitive interviews with additional stakeholders (3 youth with CD, 3 parents, and 8 clinicians) and item administration (parent/youth reports: n = 103/102). Analyses included response frequencies, internal consistency reliability, exploratory factor analyses, and correlations with related measures (Patient-Reported Outcomes Measurement Information System, Pediatric Quality of Life Inventory, and Gluten-Free Diet-Visual Analog Scale). RESULTS From concept elicitation interviews, 42 youth and 45 parent items were developed. Cognitive debriefing interviews yielded 36 refined items. Psychometric analyses identified 15 items to remove due to ceiling/floor effects, poor item-to-test correlations, and youth-parent mismatch or conceptual mismatch. Total score internal consistency was high (αs = 0.89-0.90). A 4-factor model solution had the best fit (Social Impact, External Support, Adaptive Vigilance, Eating Behaviors/Adjustment) with a fifth single-item domain (Financial Resources). The final CDLIFE (21 items) total scores correlated with most related measures in expected directions for parent and youth forms. DISCUSSION The CDLIFE may facilitate insight into CD-specific QOL for youth ages 2-18 years, capturing important dimensions of physical and socioemotional health. Administering the CDLIFE will help identify and track families needing support.
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Affiliation(s)
- Shayna Coburn
- Center for Translational Research, Children’s National Hospital, 111 Michigan Ave NW, Washington, D.C., 20010
- The George Washington School of Medicine and Health Sciences, 2300 I St NW, Washington, DC 20052
| | - Paige J. Trojanowski
- Center for Translational Research, Children’s National Hospital, 111 Michigan Ave NW, Washington, D.C., 20010
| | - Jack Vagadori
- Center for Translational Research, Children’s National Hospital, 111 Michigan Ave NW, Washington, D.C., 20010
| | - Pamela Hinds
- Center for Translational Research, Children’s National Hospital, 111 Michigan Ave NW, Washington, D.C., 20010
- The George Washington School of Medicine and Health Sciences, 2300 I St NW, Washington, DC 20052
- Department of Nursing Science, Professional Practice & Quality, Children’s National Hospital, 111 Michigan Ave NW, Washington, D.C., 20010
| | - Monique Germone
- Digestive Health Institute, Children’s Hospital Colorado, 13123 E. 16 Avenue, Aurora, CO 80045/ University of Colorado School of Medicine, 13001 E 17th Pl, Aurora, CO 80045
| | - Edwin Liu
- Digestive Health Institute, Children’s Hospital Colorado, 13123 E. 16 Avenue, Aurora, CO 80045/ University of Colorado School of Medicine, 13001 E 17th Pl, Aurora, CO 80045
| | - Randi Streisand
- Center for Translational Research, Children’s National Hospital, 111 Michigan Ave NW, Washington, D.C., 20010
- The George Washington School of Medicine and Health Sciences, 2300 I St NW, Washington, DC 20052
| | - James Bost
- Center for Translational Research, Children’s National Hospital, 111 Michigan Ave NW, Washington, D.C., 20010
- The George Washington School of Medicine and Health Sciences, 2300 I St NW, Washington, DC 20052
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Vagadori J, Trojanowski PJ, Keenan C, Bui HNT, Raber C, Perez-Junkera G, Hinds P, Streisand R, Harlan M, Coburn S. Behavioral telehealth intervention development for adolescents with celiac disease and their caregivers: The Gluten-Free Resilience and Overall Wellness (GROW) project. Nutr Health 2025:2601060251330938. [PMID: 40304638 DOI: 10.1177/02601060251330938] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 05/02/2025]
Abstract
BackgroundCeliac disease (CD) is a chronic autoimmune condition that, when left untreated, increases the risk of significant health challenges. The only medical treatment for CD is a strict gluten-free diet (GFD), which is behaviorally dependent. Despite quality of life (QOL) and adherence being impacted by the complicated and burdensome nature of the GFD, there exists a paucity of established behavioral interventions aimed to improve adherence and QOL in adolescents with CD.AimsDevelop and refine the Gluten-Free Resilience and Overall Wellness (GROW) Project, the first family-centered, online behavioral intervention to improve QOL and GFD self-management in adolescents with CD and their parents.MethodsStudy staff adapted and refined an existing online behavioral intervention for adults with CD. Two rounds of interviews with patient, parent, and clinician stakeholders were conducted to collect feedback and inform the final intervention structure and content. Qualitative interview data were analyzed using inductive content analysis.ResultsStakeholder feedback supported a group-based, virtual format across both rounds of interviews. Participants proposed format changes to the intervention to increase participant engagement. Content suggestions included enhancing information about reliable digital resources, building resilience, GFD and alcohol, and the scientific development of the program.ConclusionThe GROW Project addresses a critical need for interventions that strengthen behavioral self-management strategies in adolescents with CD and their families by providing virtual skill-building and psychoeducation that may improve family's QOL while managing CD and the GFD.
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Affiliation(s)
- Jack Vagadori
- Center for Translational Research, Children's National Hospital, Washington, DC, USA
| | - Paige J Trojanowski
- Center for Translational Research, Children's National Hospital, Washington, DC, USA
| | - Charlotte Keenan
- Center for Translational Research, Children's National Hospital, Washington, DC, USA
| | - Hong N T Bui
- Center for Translational Research, Children's National Hospital, Washington, DC, USA
- Department of Psychology, The University of Maryland, College Park, MD, USA
| | - Catherine Raber
- Center for Translational Research, Children's National Hospital, Washington, DC, USA
- Division of Gastroenterology, Children's National Hospital, Washington, DC, USA
| | - Gesala Perez-Junkera
- Center for Translational Research, Children's National Hospital, Washington, DC, USA
- GLUTEN3S Research Group, Department of Nutrition and Food Science, University of the Basque Country, Vitoria-Gasteiz, Spain
| | - Pamela Hinds
- Center for Translational Research, Children's National Hospital, Washington, DC, USA
- Department of Nursing Science, Professional Practice, and Quality, Children's National Hospital, Washington, DC, USA
- Division of Pediatrics, The George Washington School of Medicine and Health Sciences, Washington DC, USA
| | - Randi Streisand
- Center for Translational Research, Children's National Hospital, Washington, DC, USA
- Division of Pediatrics, The George Washington School of Medicine and Health Sciences, Washington DC, USA
- Division of Psychiatry and Behavioral Sciences, The George Washington School of Medicine and Health Sciences, Washington DC, USA
| | - Michelle Harlan
- Center for Translational Research, Children's National Hospital, Washington, DC, USA
- Department of Psychology, George Mason University, Fairfax, VA, USA
| | - Shayna Coburn
- Center for Translational Research, Children's National Hospital, Washington, DC, USA
- Division of Pediatrics, The George Washington School of Medicine and Health Sciences, Washington DC, USA
- Division of Psychiatry and Behavioral Sciences, The George Washington School of Medicine and Health Sciences, Washington DC, USA
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Sweeny KF, Lee CK, Mitchell PD, Osiakwan S, Scott C, Jonas MM. A Cross-Sectional Retrospective Review of Neuropsychiatric Comorbidities in Children With Non-Alcoholic Fatty Liver Disease. Clin Pediatr (Phila) 2025:99228251330113. [PMID: 40146031 DOI: 10.1177/00099228251330113] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 03/28/2025]
Abstract
This single-center cross-sectional retrospective chart review sought to define the prevalence of neuropsychiatric (NP) disorders among children with non-alcoholic fatty liver disease (NAFLD) compared to the general population. The prevalence rate of NP disorders was determined at diagnosis among children evaluated from 2006 to 2016. The NP disorder prevalence rates were compared to those reported in the 2016 National Survey of Children's Health (NSCH). 24.3% of 254 children with NAFLD reported to have at least 1 NP disorder compared to 16.9% of those in NSCH (relative risk [RR] = 1.44; P < .0001). More children had intellectual disability (ID) (4.5% vs 0.8%; RR = 5.43, P < .0001), depression (3.9% vs 2.2%; RR = 1.82, P = .0009), or autism spectrum disorder (ASD) (3.3% vs 2.0%; RR = 1.67, P = .047) in the Boston Children's Hospital (BCH) NAFLD cohort. The prevalence of 1 NP disorder was 29.4% in our cohort compared to 22.3% in NSCH (RR = 1.32; P = .007). Children with NAFLD have a higher rate of NP disorders compared to the general population.
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Affiliation(s)
- Katherine F Sweeny
- Division of Gastroenterology, Hepatology and Nutrition, Department of Pediatrics, Boston Children's Hospital, Boston, MA, USA
| | - Christine K Lee
- Division of Gastroenterology, Hepatology and Nutrition, Department of Pediatrics, Boston Children's Hospital, Boston, MA, USA
| | - Paul D Mitchell
- Institutional Centers for Clinical and Translational Research, Boston Children's Hospital, Boston, MA, USA
| | - Shavonne Osiakwan
- Division of Gastroenterology, Hepatology and Nutrition, Department of Pediatrics, Boston Children's Hospital, Boston, MA, USA
| | - Cosette Scott
- Division of Gastroenterology, Hepatology and Nutrition, Department of Pediatrics, Boston Children's Hospital, Boston, MA, USA
| | - Maureen M Jonas
- Division of Gastroenterology, Hepatology and Nutrition, Department of Pediatrics, Boston Children's Hospital, Boston, MA, USA
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Kridin K, Papara C, Bieber K, De Luca DA, Klein JP, Ludwig MA, Curman P, Vorobyev A, Dempfle A, Ludwig RJ. Nicotine dependence is associated with an increased risk of developing chronic, non-communicable inflammatory disease: a large-scale retrospective cohort study. Front Psychiatry 2025; 16:1429297. [PMID: 40012715 PMCID: PMC11860976 DOI: 10.3389/fpsyt.2025.1429297] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/12/2024] [Accepted: 01/08/2025] [Indexed: 02/28/2025] Open
Abstract
Introduction Chronic, non-communicable inflammatory diseases (CIDs) affect a large portion of the population, imposing a significant morbidity, encompassing a substantial mortality. Thus, they are a major medical burden with a high unmet need. CIDs develop over the span of several years, and the risk of developing CIDs has been linked to genetic and environmental factors. Thus, modification of environmental factors is a promising approach for the prevention of CIDs. Among modifiable environmental factors that have been linked to the CID risk is nicotine dependence. However, for only few CIDs, compelling evidence suggests that nicotine dependence increases (e.g., rheumatoid arthritis and asthma) or decreases (e.g., pemphigus) the CID risk. For most CIDs, there are inconsistent, scant, or no reports on the risk of CID associated with nicotine dependence. Methods To address this gap, we leveraged TriNetX, analyzing data from over 120 million electronic health records (EHRs). Using propensity score matching (PSM) to control for age, sex, ethnicity, and other CID risk factors, we contrasted the risk of developing any or any of the 38 CIDs in 881,192 EHRs from individuals with nicotine dependence to PSM-matched unexposed counterparts. Results The analytical pipeline was validated by demonstrating an increased risk of individuals exposed to nicotine dependence for subsequent diagnosis of myocardial infarction, malignant neoplasm of the lung, and chronic obstructive pulmonary disease. Overall, 16.8% of individuals with nicotine dependence developed CIDs, compared to 9.6% of individuals not exposed to nicotine dependence (hazard ratio 2.12, confidence interval 2.10-2.14, p < 0.0001). Investigating single CIDs, nicotine dependence imposed increased risks for 23 of the 38 investigated diseases, i.e., dermatomyositis, granulomatosis with polyangiitis, pyoderma gangrenosum, and immune thrombocytopenic purpura. The sex-stratified analysis revealed few sex-specific differences in CID risk. Discussion Our study emphasizes the importance of preventive measures targeting nicotine addiction to reduce the global burden of CIDs.
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Affiliation(s)
- Khalaf Kridin
- Lübeck Institute of Experimental Dermatology, University of Lübeck, Lübeck, Germany
- Azrieli Faculty of Medicine, Bar-Ilan University, Safed, Israel
- Unit of Dermatology and Skin Research Laboratory, Galilee Medical Center, Nahariya, Israel
| | - Cristian Papara
- Institure and Comprehensive Centre for Inflammation Medicine, University Hospital Schleswig-Holstein (UKSH), Lübeck, Germany
| | - Katja Bieber
- Lübeck Institute of Experimental Dermatology, University of Lübeck, Lübeck, Germany
| | - David A. De Luca
- Institure and Comprehensive Centre for Inflammation Medicine, University Hospital Schleswig-Holstein (UKSH), Lübeck, Germany
| | - Jan Philipp Klein
- Department of Psychiatry, Psychosomatics and Psychotherapy, Lübeck University, Lübeck, Germany
| | | | - Philip Curman
- Lübeck Institute of Experimental Dermatology, University of Lübeck, Lübeck, Germany
- Dermato-Venereology Clinic, Karolinska University Hospital, Stockholm, Sweden
- Department of Medical Epidemiology and Biostatistics, Karolinska Institutet, Stockholm, Sweden
- Department of Medicine Solna, Karolinska Institutet, Stockholm, Sweden
| | - Artem Vorobyev
- Department of Dermatology, University Hospital Schleswig-Holstein (UKSH), Lübeck, Germany
| | - Astrid Dempfle
- Institute of Medical Informatics and Statistics, Kiel University, Kiel, Germany
| | - Ralf J. Ludwig
- Department of Dermatology, University Hospital Schleswig-Holstein (UKSH), Lübeck, Germany
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Gallegos D, McLaren-Kennedy A, Lang R, Thapar N, Mealing S. Living with Pediatric Coeliac Disease: Lessons for Health Service Delivery. Dig Dis Sci 2025; 70:516-525. [PMID: 39762594 PMCID: PMC11839893 DOI: 10.1007/s10620-024-08761-7] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/01/2024] [Accepted: 11/14/2024] [Indexed: 02/21/2025]
Abstract
BACKGROUND Coeliac Disease (CD) affects up to 1.4% of children worldwide, with a rising global incidence. A less typical clinical presentation and the need for a life-long gluten exclusion diet raise challenges for diagnosis, management, and healthcare delivery with considerable impacts for CD patients and families as well as clinical services. AIMS To explore the lived experiences of caregivers of children with CD to identify barriers and enablers to diagnosis, referral, and treatment to inform a more streamlined service delivery model. METHODS Semi-structured interviews with caregivers of children with CD diagnosed for at least one month with no significant co-morbidities. Interviews were thematically analyzed. RESULTS Sixteen participants representing 12 family units were interviewed. Children with CD ranged in age from 3 to 18 years. Time from symptom onset to diagnosis varied from one month to > 10 years and symptoms were mainly atypical and non-specific. Six key themes were identified: the iterative diagnosis journey, restricted choices, child responsibility and autonomy, mental load (encompassing foodwork, emotional rollercoaster, and lack of trust), Google™ reigns for information, and where to from here? CONCLUSION There is a need to decentralize CD diagnosis and management to meet the increasing demand created by rising incidence. Participants highlighted the need for a more streamlined diagnosis pathway, increased training of health professionals, and access to age-appropriate resources. Efforts need to be made to advocate for increased community awareness. These insights will be used to reimagine and co-design a decentralized model of care for pediatric CD diagnosis and management in Queensland, Australia.
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Affiliation(s)
- Danielle Gallegos
- Centre for Childhood Nutrition Research, Queensland University of Technology (QUT), Graham St, South Brisbane, 4101, Australia.
- School of Exercise and Nutrition Sciences, Queensland University of Technology (QUT), Victoria Park Rd, Kelvin Grove, 4059, Australia.
| | - Annette McLaren-Kennedy
- Department of Paediatric Gastroenterology, Hepatology and Liver Transplant, Queensland Children's Hospital, Graham St, South Brisbane, 4101, Australia
| | - Ray Lang
- Department of Paediatric Gastroenterology, Hepatology and Liver Transplant, Queensland Children's Hospital, Graham St, South Brisbane, 4101, Australia
| | - Nikhil Thapar
- Centre for Childhood Nutrition Research, Queensland University of Technology (QUT), Graham St, South Brisbane, 4101, Australia
- Department of Paediatric Gastroenterology, Hepatology and Liver Transplant, Queensland Children's Hospital, Graham St, South Brisbane, 4101, Australia
- School of Medicine, University of Queensland, Herston Rd, Herston, Brisbane, Australia
| | - Sasha Mealing
- Department of Paediatric Gastroenterology, Hepatology and Liver Transplant, Queensland Children's Hospital, Graham St, South Brisbane, 4101, Australia
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Cheung T, McDonald C, Setty M, Tsai P, Wadhwani SI. Social Adversities Associate with Worse Disease Control in Pediatric Celiac Disease. J Pediatr 2025; 276:114305. [PMID: 39293560 PMCID: PMC11812434 DOI: 10.1016/j.jpeds.2024.114305] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/07/2024] [Revised: 06/24/2024] [Accepted: 09/11/2024] [Indexed: 09/20/2024]
Abstract
OBJECTIVE To characterize how social adversities influence disease control in children with celiac disease (CeD). STUDY DESIGN We conducted a cross-sectional analysis of data from 325 eligible children ≤18 years old with CeD enrolled between 2015 through 2023 into iCureCeliac, a patient-centered US registry for CeD. We evaluated the associations between financial insecurity, social stigmatization, decreased health knowledge, and mental health comorbidity with 2 validated patient-reported outcomes on disease activity and gluten-free diet adherence: celiac symptom index and CeD adherence test, respectively. We used multivariable logistic and linear regression analysis to adjust for race, primary spoken language, and socioeconomic status. RESULTS Among 325 children with available financial insecurity data, the median age was 11 years (IQR 8, 15), 67% were female, and 88% were White. In multivariable logistic regression, the odds of elevated disease activity among children with financial insecurity, social stigmatization, decreased health knowledge, and mental health comorbidity were 2.6 (95% CI 0.9, 8.0; P = .09), 2.8 (95% CI 1.6, 5.1; P < .001), 4.8 (95% CI 2.4, 9.8; P < .001), and 1.9 (95% CI 1.1, 3.3; P = .03), respectively. For insufficient dietary adherence, the respective odds were 1.6 (95% CI 0.5, 4.7; P = .43), 3.3 (95% CI 1.7, 6.5; P < .001), 2.9 (95% CI 1.5, 5.7; P = .002), and 2.3 (95% CI 1.2, 4.2; P = .01). Statistically significant associations in logistic regression aligned with results of linear models. CONCLUSIONS Social stigmatization, decreased health knowledge, and mental health comorbidity were associated with worse disease control in pediatric CeD. Targeted interventions aimed at addressing these social adversities may improve disease activity and dietary adherence.
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Affiliation(s)
- Telly Cheung
- Department of Pediatrics, University of California, San Francisco, San Francisco, CA
| | - Christine McDonald
- Department of Pediatrics, University of California, San Francisco, San Francisco, CA
| | - Mala Setty
- Department of Pediatrics, University of California, San Francisco, San Francisco, CA
| | - Patrika Tsai
- Department of Pediatrics, University of California, San Francisco, San Francisco, CA
| | - Sharad I Wadhwani
- Department of Pediatrics, University of California, San Francisco, San Francisco, CA.
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Ciaccio EJ, Lee AR, Lebovits J, Wolf RL, Lewis SK. Physical and psychological symptoms and survey importance in celiac disease. World J Gastrointest Endosc 2024; 16:632-639. [PMID: 39735391 PMCID: PMC11669964 DOI: 10.4253/wjge.v16.i12.632] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/24/2024] [Revised: 09/20/2024] [Accepted: 10/24/2024] [Indexed: 12/12/2024] Open
Abstract
Celiac disease is an autoimmune condition that affects approximately 1% of the worldwide community. Originally thought to be confined mostly to the small intestine, resulting in villous atrophy and nutrient malabsorption, it has more recently been implicated in systemic manifestations as well, particularly when undiagnosed or left untreated. Herein, the physical and psychological symptoms of celiac disease are described and explored. An emphasis is placed on efforts to query prospective and confirmed celiac disease patients via the use of surveys. Suggestions are made regarding the development of efficacious surveys for the purpose of screening for celiac disease in undiagnosed persons, and monitoring efficacy of the gluten-free diet in persons diagnosed with celiac disease. There are broad categories of physical and psychological symptoms associated with celiac disease. There is also an essential interaction between such physical and the psychological symptoms. It is important to capture the association between symptoms, via queries directed toward suspected and confirmed persons with celiac disease. The use of anonymous online surveys can be helpful to determine the qualities and characteristics which may be associated with this condition. It is suggested that personal surveys should be given a greater role in screening and to lessen the time for diagnosis. Querying the subject directly via a survey can provide clues as to the types of symptoms being experienced by those with celiac disease currently, as well as to determine the salient aspects of the symptomatology, which will be useful for rapid screening and monitoring in future work.
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Affiliation(s)
- Edward J Ciaccio
- Celiac Disease Center at Columbia University Medical Center, Columbia University, New York, NY 10032, United States
| | - Anne R Lee
- Celiac Disease Center at Columbia University Medical Center, Columbia University, New York, NY 10032, United States
| | - Jessica Lebovits
- Celiac Disease Center at Columbia University Medical Center, Columbia University, New York, NY 10032, United States
| | - Randi L Wolf
- Department of Health Studies and Applied Educational Psychology, Columbia University, Teachers College, New York, NY 10027, United States
| | - Suzanne K Lewis
- Celiac Disease Center at Columbia University Medical Center, Columbia University, New York, NY 10032, United States
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Bozas A, Karakatsoulis G, Panagopoulou E, Xinias I, Fotoulaki M. Charting the Path: Psychological Factors and Diet Adherence in Adolescents With Celiac Disease. Cureus 2024; 16:e74103. [PMID: 39712698 PMCID: PMC11661695 DOI: 10.7759/cureus.74103] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 11/20/2024] [Indexed: 12/24/2024] Open
Abstract
Introduction Adolescence is a pivotal time for individuals with celiac disease (CD), presenting a host of psychosocial challenges. Managing a strict gluten-free diet (GFD) while forming self-identity, striving for autonomy, and navigating social relationships significantly impacts adolescents with CD. The present pilot study investigates the impact of psychological factors on behavioral and dietary responses in adolescents with CD, utilizing repeated measures over time. Methods Thirty-one adolescents aged 11-18 from a pediatric outpatient CD department were recruited. Participants completed the Resilience Youth Development Module (RYDM), the Child and Adolescent Mindfulness Measure (CAMM), the Strengths and Difficulties Questionnaire (SDQ), and Celiac Dietary Adherence Test (CDAT) over four phases spanning four months. Twenty-seven parents also agreed to participate in completing the SDQ parents' version. Results The results revealed moderate levels of mindfulness and resilience, accompanied by inadequate adherence to a GFD. Baseline assessments revealed difficulties in peer relationships and psychosocial functioning, which parents corroborated. Over time, reductions in mindfulness and resilience were observed, along with modest improvements in dietary adherence and decreases in psychosocial difficulties. Discussion This study underscores the importance of psychological traits, suggesting that enhancing mindfulness and resilience may improve both dietary adherence and overall well-being. However, given the dynamic nature of adolescents' coping strategies through developmental changes and social environments, tailored interventions are essential for effective disease management and social integration.
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Affiliation(s)
- Antonios Bozas
- 4th Department of Pediatrics, Aristotle University of Thessaloniki, Thessaloniki, GRC
| | - Georgios Karakatsoulis
- Institute of Applied Biosciences, Center for Research and Technology Hellas (CERTH), Thessaloniki, GRC
| | - Efharis Panagopoulou
- Laboratory of Primary Health Care, Aristotle University of Thessaloniki, Thessaloniki, GRC
| | - Ioannis Xinias
- 3rd Department of Pediatrics, Aristotle University of Thessaloniki, Thessaloniki, GRC
| | - Maria Fotoulaki
- 4th Department of Pediatrics, Aristotle University of Thessaloniki, Thessaloniki, GRC
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Sahın ÖO, Tasdelen Y, Kutahyalıoglu NS, Guney EU, Ozer K. School Experiences in the Disease Management of Children With Celiac Disease: A Qualitative Study. Child Care Health Dev 2024; 50:e70010. [PMID: 39555629 DOI: 10.1111/cch.70010] [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: 08/12/2024] [Revised: 10/30/2024] [Accepted: 11/03/2024] [Indexed: 11/19/2024]
Abstract
BACKGROUND This study was conducted to examine in depth the disease management experiences of school-age children with celiac disease and the effects of family, friends, and teachers on disease management at school. METHODS The study employed a qualitative research method using the phenomenology design. Face-to-face, semi-structured, in-depth interviews were conducted with children (n = 14) with celiac disease. The transcripts were analysed using a phenomenological approach and an inductive process. RESULTS Three main themes were identified: Individual, Institutional, and Environmental. It was observed that children adapted better to the celiac diet as they got older, but they had difficulty adjusting to the diet in the school environment. In addition, it was found that school-age children had challenges in diagnosing the disease and finding products suitable for the diet in a rural city with limited facilities. It was determined that the approaches of peers and teachers throughout the child's school life affected the disease management levels of children. CONCLUSION The study reveals the emotional, cognitive, and behavioural difficulties that children face while adapting to celiac disease and diet, especially in the school environment. The results emphasize that school-age children with celiac disease should not be ignored and that the school nurse plays an essential role in this process.
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Affiliation(s)
- Özlem Ozturk Sahın
- Faculty of Health Sciences, Pediatric Nursing Department, Karabük University, Karabük, Turkiye
| | - Yeliz Tasdelen
- Faculty of Health Sciences, Pediatric Nursing Department, Karabük University, Karabük, Turkiye
| | - Nesibe S Kutahyalıoglu
- Faculty of Health Sciences, Pediatric Nursing Department, Karabük University, Karabük, Turkiye
| | - Emine Uzuntarla Guney
- Faculty of Health Sciences, Pediatric Nursing Department, Karabük University, Karabük, Turkiye
| | - Kadriye Ozer
- Zonguldak Provincial Directorate of National Education, Zonguldak, Turkiye
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10
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Fucà E, Costanzo F, Galassi P, Celestini L, Valentini D, Vicari S. Celiac disease in children and adolescents with Down syndrome: behavioural, adaptive and sleep profiles. JOURNAL OF INTELLECTUAL DISABILITY RESEARCH : JIDR 2024; 68:932-940. [PMID: 38509762 DOI: 10.1111/jir.13135] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Grants] [Track Full Text] [Subscribe] [Scholar Register] [Received: 07/03/2023] [Revised: 03/01/2024] [Accepted: 03/01/2024] [Indexed: 03/22/2024]
Abstract
BACKGROUND Individuals with Down syndrome (DS) exhibit higher risk for celiac disease (CD) than general population. Although literature suggests CD could be associated with behavioural problems in both paediatric and adult age, such association has been poorly explored in children and adolescents DS. Therefore, the current study aimed to investigate differences in emotional/behavioural difficulties, adaptive skills and sleep problems between children with DS with and without CD. METHODS Data were retrospectively collected from a database including data from 381 individuals with DS (3-18 years). The final sample included 65 participants, 27 with co-occurring CD and 38 age, IQ, sex and body mass index-matched controls without CD. Emotional/behavioural difficulties, adaptive skills and sleep problems were assessed through parent report questionnaires. RESULTS No group differences emerged in emotional/behavioural difficulties, whereas participants in the CD group showed better adaptive skills in the practical domain than control group. Weak differences emerged in sleep problems. CONCLUSIONS Youth with DS and co-occurring CD do not exhibit more emotional and behavioural problems than youth with DS without co-occurring CD but exhibit better adaptive skills in the practical domain.
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Affiliation(s)
- E Fucà
- Child and Adolescent Neuropsychiatry Unit, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy
| | - F Costanzo
- Child and Adolescent Neuropsychiatry Unit, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy
| | - P Galassi
- Child and Adolescent Neuropsychiatry Unit, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy
| | - L Celestini
- Pediatric Unit, Pediatric Emergency Department, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy
| | - D Valentini
- Pediatric Unit, Pediatric Emergency Department, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy
| | - S Vicari
- Child and Adolescent Neuropsychiatry Unit, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy
- Department of Life Science and Public Health, Catholic University of the Sacred Heart, Rome, Italy
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11
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Ciaccio EJ, Lee AR, Lebovits J, Wolf RL, Lewis SK, Ciacci C, Green PHR. Psychological, Psychiatric, and Organic Brain Manifestations of Celiac Disease. Dig Dis 2024; 42:419-444. [PMID: 38861947 DOI: 10.1159/000534219] [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: 04/19/2023] [Accepted: 09/07/2023] [Indexed: 06/13/2024]
Abstract
INTRODUCTION Celiac disease is an autoimmune condition that affects approximately 1% of the population worldwide. Although its main impact often concerns the small intestine, resulting in villous atrophy and nutrient malabsorption, it can also cause systemic manifestations, particularly when undiagnosed or left untreated. METHOD Attention is directed to the possible psychological, psychiatric, and organic brain manifestations of celiac disease. Specific topics related to the influence and risk of such manifestations with respect to celiac disease are defined and discussed. Overall, eighteen main topics are considered, sifted from over 500 references. RESULTS The most often studied topics were found to be the effect on quality of life, organic brain dysfunction and ataxia, epilepsy, Down syndrome, generalized psychological disorders, eating dysfunction, depression, and schizophrenia. For most every topic, although many studies report a connection to celiac disease, there are often one or more contrary studies and opinions. A bibliographic analysis of the cited articles was also done. There has been a sharp increase in interest in this research since 1990. Recently published articles tend to receive more referencing, up to as many as 15 citations per year, suggesting an increasing impact of the topics. The number of manuscript pages per article has also tended to increase, up to as many as 12 pages. The impact factor of the publishing journal has remained level over the years. CONCLUSION This compendium may be useful in developing a consensus regarding psychological, psychiatric, and organic brain manifestations that can occur in celiac disease and for determining the best direction for ongoing research focus.
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Affiliation(s)
- Edward J Ciaccio
- Department of Medicine - Celiac Disease Center, Columbia University Irving Medical Center, New York, New York, USA
| | - Anne R Lee
- Department of Medicine - Celiac Disease Center, Columbia University Irving Medical Center, New York, New York, USA
| | - Jessica Lebovits
- Department of Medicine - Celiac Disease Center, Columbia University Irving Medical Center, New York, New York, USA
| | - Randi L Wolf
- Teachers College, Columbia University, New York, New York, USA
| | - Suzanne K Lewis
- Department of Medicine - Celiac Disease Center, Columbia University Irving Medical Center, New York, New York, USA
| | - Carolina Ciacci
- Department of Medicine, Surgery, and Dentistry, Scuola Medica Salernitana, Università degli Studi di Salerno, Salerno, Italy
| | - Peter H R Green
- Department of Medicine - Celiac Disease Center, Columbia University Irving Medical Center, New York, New York, USA
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12
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Anderson RP, Verma R, Schumann M. A Look Into the Future: Are We Ready for an Approved Therapy in Celiac Disease? Gastroenterology 2024; 167:183-193. [PMID: 38355059 DOI: 10.1053/j.gastro.2024.02.005] [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: 11/29/2023] [Revised: 02/02/2024] [Accepted: 02/06/2024] [Indexed: 02/16/2024]
Abstract
As it appears that we are currently at the cusp of an era in which drugs that are new, re-purposed, or "supplements" will be introduced to the management of celiac disease, we need to reflect on whether the framework is set for celiac disease to be treated increasingly with pharmaceuticals as well as diet. This refers to reflecting on the rigor of current diagnostic practices; the limitations of the current standard of care, which is a gluten-free diet; and that we lack objective markers of disease severity. Investigating these issues will help us to identify gaps in technology and practices that could be critical for selecting patients with a well-defined need for an improved or alternative treatment. Both aspects, circumscribed limitations of the gluten-free diet and diagnostics helping to define celiac disease target groups, together with the guiding requirements by the responsible regulatory authorities, will contribute to defining the subgroups of patients with confirmed celiac disease eligible for distinct pharmacologic strategies. Because many patients with celiac disease are diagnosed in childhood, these aspects need to be differentially discussed for the pediatric setting. In this perspective, we aimed to describe these contextual issues and then looked ahead to the future. What might be the major challenges in celiac disease clinics in the coming years once drugs are an option alongside diet? And what will be the future objectives for researchers who further decipher the mucosal immunology of celiac disease? Speculating on the answers to these questions is as stimulating as it is fascinating to be part of this turning point.
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Affiliation(s)
- Robert P Anderson
- Gastroenterology Service, Mackay Base Hospital, West Mackay, Queensland, Australia
| | - Ritu Verma
- University of Chicago, Comer Children's Hospital, Chicago, Illinois
| | - Michael Schumann
- Department of Gastroenterology, Infectious Diseases and Rheumatology, Charité Universitätsmedizin Berlin, Berlin, Germany.
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13
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Mathialagan K, Rondla M. Celiac disease and mental health - A concealed association. Am J Med Sci 2023; 365:545-546. [PMID: 36925063 DOI: 10.1016/j.amjms.2023.03.008] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/17/2023] [Accepted: 03/09/2023] [Indexed: 03/16/2023]
Affiliation(s)
| | - Madhumitha Rondla
- Department of Internal Medicine, Narayana Medical College, Nellore, India
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14
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Coburn S, Germone M, McGarva J, Taft T. Psychological Considerations for Food Intolerances: Celiac Sprue, Eosinophilic Esophagitis, and Non-Celiac Gluten Sensitivity. Gastroenterol Clin North Am 2022; 51:753-764. [PMID: 36375994 PMCID: PMC10581356 DOI: 10.1016/j.gtc.2022.07.003] [Citation(s) in RCA: 7] [Impact Index Per Article: 2.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/04/2022]
Abstract
Several chronic digestive conditions are physiologically based on food intolerance, including celiac disease, nonceliac gluten sensitivity, and eosinophilic esophagitis. Patients are expected to follow medically prescribed diets to eliminate identified food triggers to control symptoms. However, the psychological impacts of these dietary approaches are largely unaddressed in clinical practice. Hypervigilance and anxiety regarding food and symptoms, and disordered eating, may emerge and negatively affect outcomes. Clinicians working with pediatric and adult populations with food intolerances should be aware of these psychological comorbidities, and equally emphasize effective ways to help patients manage the mental and physical aspects of their condition.
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Affiliation(s)
- Shayna Coburn
- Children's National Hospital 111 Michigan Avenue NW, Center for Translational Research, 6th Flr Main, Washington, DC 20010, USA
| | - Monique Germone
- Departments of Psychiatry and Pediatrics, University of Colorado Anschutz Medical Campus, Digestive Health Institute, Children's Hospital Colorado, 13123 E. 16th Ave., B130, Aurora, CO 80045, USA
| | - Josie McGarva
- Division of Gastroenterology and Hepatology, Northwestern University Feinberg School of Medicine, 676 North Saint Clair Street Suite 1400, Chicago, IL 60611, USA
| | - Tiffany Taft
- Division of Gastroenterology and Hepatology, Northwestern University Feinberg School of Medicine, 676 North Saint Clair Street Suite 1400, Chicago, IL 60611, USA.
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15
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Germone M, Phu T, Slosky C, Pan Z, Jones A, Stahl M, Mehta P, Shull M, Ariefdjohan M, Liu E. Anxiety and Depression in Pediatric Patients with Celiac Disease: A Large Cross-Sectional Study. J Pediatr Gastroenterol Nutr 2022; 75:181-185. [PMID: 35641896 DOI: 10.1097/mpg.0000000000003497] [Citation(s) in RCA: 11] [Impact Index Per Article: 3.7] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/10/2022]
Abstract
Mental health is a growing concern in pediatric celiac disease (CD). This study utilized the Revised Children's Anxiety and Depression Scale (RCADS) to investigate anxiety and depression symptom rates. Participants were children ages 8 to 17 years (M = 11.7, SD = 2.7; N = 175) with biopsy-proven CD (Median = 1.1 years post-diagnosis, IQR = 0-4) categorized into groups based on the child's age, caregiver or child respondent, presence or absence of comorbidities, and gluten-free diet duration. Self-reported RCADS scores showed 39% of children having clinically significant concerns for anxiety or depression ( P < 0.0001) but only 7% of caregiver-proxy RCADS scores indicated significant concerns for the child's anxiety and 14% for the child's depression. Rates of child-reported anxiety and depression symptoms were significantly higher for those without medical comorbidities than those with ( P = 0.04). Therefore, screening for mental health concerns, particularly anxiety and depression, should be routinely performed in pediatric patients with CD.
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Affiliation(s)
- Monique Germone
- From the Department of Psychiatry, University of Colorado Anschutz Medical Campus, Aurora, CO
- the Pediatric Mental Health Institute, Children's Hospital Colorado, Aurora, CO
- the Colorado Center for Celiac Disease, Digestive Health Institute, Children's Hospital Colorado, Aurora, CO
| | - Tiffany Phu
- the Pediatric Mental Health Institute, Children's Hospital Colorado, Aurora, CO
| | - Camryn Slosky
- From the Department of Psychiatry, University of Colorado Anschutz Medical Campus, Aurora, CO
| | - Zhaoxing Pan
- the Biostatistics Core of Children's Hospital Colorado Research Institute
| | - Anna Jones
- the Department of Psychology, St. Jude Children's Research Hospital, Memphis, TN
| | - Marisa Stahl
- the Colorado Center for Celiac Disease, Digestive Health Institute, Children's Hospital Colorado, Aurora, CO
- the Division of Gastroenterology, Hepatology, and Nutrition, Department of Pediatrics, University of Colorado Anschutz Medical Campus, Aurora, CO
| | - Pooja Mehta
- the Colorado Center for Celiac Disease, Digestive Health Institute, Children's Hospital Colorado, Aurora, CO
- the Division of Gastroenterology, Hepatology, and Nutrition, Department of Pediatrics, University of Colorado Anschutz Medical Campus, Aurora, CO
| | - Mary Shull
- the Colorado Center for Celiac Disease, Digestive Health Institute, Children's Hospital Colorado, Aurora, CO
- the Division of Gastroenterology, Hepatology, and Nutrition, Department of Pediatrics, University of Colorado Anschutz Medical Campus, Aurora, CO
| | - Merlin Ariefdjohan
- From the Department of Psychiatry, University of Colorado Anschutz Medical Campus, Aurora, CO
| | - Edwin Liu
- the Colorado Center for Celiac Disease, Digestive Health Institute, Children's Hospital Colorado, Aurora, CO
- the Division of Gastroenterology, Hepatology, and Nutrition, Department of Pediatrics, University of Colorado Anschutz Medical Campus, Aurora, CO
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16
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Cesa KT, Cunningham CA, Noll RB, Kim SC. Parental Distress in Pediatric Inflammatory Bowel Diseases: Associations With Time From Diagnosis, Disease Activity, and Demographic Factors. CROHN'S & COLITIS 360 2022; 4:otac019. [PMID: 35783219 PMCID: PMC9237933 DOI: 10.1093/crocol/otac019] [Citation(s) in RCA: 4] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/08/2022] [Indexed: 11/14/2022] Open
Abstract
Background There are limited studies examining caregiver distress when raising a child with inflammatory bowel disease (IBD). The aim of this study was to investigate the occurrence of symptoms of distress (anxiety, depression, and post-traumatic stress disorder [PTSD]) among parents with children with IBD and associations with disease severity, time from diagnosis, and demographic factors. Methods We conducted a cross-sectional study with parents of children (2-17 years) diagnosed with IBD. There were 2 cohorts: (1) recently diagnosed cohort (<6 months from diagnosis); (2) established diagnosis cohort (>1 year from diagnosis). Parents completed measures of anxiety, depression, and PTSD, while children completed surveys on the symptoms of their IBD. Results Fifty-two parents in the recently diagnosed cohort and 103 parents in the established diagnosis cohort completed surveys. For the entire cohort of parents, we found the mean scores on all measures of distress were within the normal ranges with 20%, 13%, and 8% of parents reporting moderate-to-severe symptoms of anxiety, depression, and PTSD, respectively. Symptoms of anxiety and depression were not significantly associated with time from diagnosis; symptoms of anxiety and PTSD were significantly associated with patients' IBD clinical activity. Conclusions Parents with children with IBD are remarkably resilient to distress even soon after their child's diagnosis. Despite considerable resilience, routine brief caregiver screening for symptoms of anxiety during annual visits seems reasonable and feasible.
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Affiliation(s)
- Kevin T Cesa
- Address correspondence to: Kevin T. Cesa, MD, Department of Pediatrics, University of Pittsburgh School of Medicine, 4401 Penn Avenue, Pittsburgh, PA 15224, USA ()
| | - Catherine A Cunningham
- Department of Pediatrics, University of Pittsburgh School of Medicine, Pittsburgh, Pennsylvania, USA
| | - Robert B Noll
- Department of Child Development, University of Pittsburgh School of Medicine, Pittsburgh, Pennsylvania, USA
| | - Sandra C Kim
- Division of Pediatric Gastroenterology, Hepatology, and Nutrition, University of Pittsburgh School of Medicine, Pittsburgh, Pennsylvania, USA
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17
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Gaur S. The Association between ADHD and Celiac Disease in Children. CHILDREN (BASEL, SWITZERLAND) 2022; 9:781. [PMID: 35740718 PMCID: PMC9221618 DOI: 10.3390/children9060781] [Citation(s) in RCA: 7] [Impact Index Per Article: 2.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Received: 02/28/2022] [Revised: 04/11/2022] [Accepted: 05/13/2022] [Indexed: 11/17/2022]
Abstract
UNLABELLED Controversy around the association between celiac disease (CeD) and attention deficit hyperactive disorder (ADHD) was addressed by a systematic review in 2015, ultimately showing no association. Since 2015, there have been several studies showing an association between celiac disease and attention deficit hyperactive disorder. This is an updated systematic review. BACKGROUND Most experts agree on the recommendation to not screen as part of the standard of care for ADHD in persons with CeD or vice versa. Simultaneously, they propose that untreated patients with CeD and neurological symptoms such as chronic fatigue, inattention, pain, and headache could be predisposed to ADHD-like behavior, namely inattention (which may be alleviated by following a gluten-free diet). The inattentive subtype of ADHD that encompasses the symptoms of inattention is phenotypically heterogeneous, as it includes the clinical construct of sluggish cognitive tempo (SCT). SCT symptoms overlap with the neurological manifestations of CeD. METHODS A systematic search (PRISMA) of PubMed, Google Scholar, EMBASE, Web of Science, Stanford Lane, SCOPUS, and Ovid was conducted for articles up to 21 February 2022. Of these, 23 studies met the criteria. RESULTS Out of the 23 studies, 13 showed a positive association between ADHD and CeD. Most studies that showed a positive association had been published in the last five years. Inconsistencies in the results remain due to the heterogeneous methodology used, specifically for ADHD and the outcome questionnaires, as well as a lack of reporting on ADHD subtypes. CONCLUSION There is an association between ADHD and celiac disease. The current methodological limitations will be lessened if we examine the subtypes of ADHD.
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Affiliation(s)
- Sonia Gaur
- Department of Psychiatry, Stanford School of Medicine, 401 Quarry Road, Stanford, CA 94305, USA
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18
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Honar N, Radanfar R, Ghanizadeh A, Barkhordarian M, Javaherizadeh H. Is attention-deficit hyperactivity disorder increased in patients with celiac disease? MIDDLE EAST CURRENT PSYCHIATRY 2022. [DOI: 10.1186/s43045-022-00201-4] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/10/2022] Open
Abstract
Abstract
Background
Some studies have shown that neurological and mental disorders in children with celiac disease are higher than in the general population. The aim of this study is to find frequency of ADHD in celiac patients.
Result
The findings of the study showed that the frequency of ADHD in males was 6 cases (22.2%) and 6 cases (18.2%) in females, and although the ADHD score was higher in males, there was no significant difference in sex (P = 0.697). The frequency of ADHD in terms of age group also showed that there were no hyperactive children in the age group under 5 years of age; in the age group of 5–9 years old, 8 cases (28.6%); in the age group of 10 to 14 years old, 4 cases (19%); and in the age group of 15 and older, no one was diagnosed with ADHD. The chi-square test showed that the frequency of ADHD in terms of age group was not significantly different (P = 0.26). It should be noted that according to Pearson correlation test, there was a reverse correlation between the age and ADHD, but it was not statistically significant (P = 0.33).
Conclusions
The frequency of ADHD among boys and girls with celiac disease was 22.2% and 18.2%, respectively.
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19
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Lerner A, Benzvi C. Checkpoint Inhibitors and Induction of Celiac Disease-like Condition. Biomedicines 2022; 10:609. [PMID: 35327411 PMCID: PMC8945786 DOI: 10.3390/biomedicines10030609] [Citation(s) in RCA: 3] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/06/2022] [Revised: 03/02/2022] [Accepted: 03/03/2022] [Indexed: 12/13/2022] Open
Abstract
Immune checkpoint inhibitors herald a new era in oncological therapy-resistant cancer, thus bringing hope for better outcomes and quality of life for patients. However, as with other medications, they are not without serious side effects over time. Despite this, their advantages outweigh their disadvantages. Understanding the adverse effects will help therapists locate, apprehend, treat, and perhaps diminish them. The major ones are termed immune-related adverse events (irAEs), representing their auto-immunogenic capacity. This narrative review concentrates on the immune checkpoint inhibitors induced celiac disease (CD), highlighting the importance of the costimulatory inhibitors in CD evolvement and suggesting several mechanisms for CD induction. Unraveling those cross-talks and pathways might reveal some new therapeutic strategies.
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Affiliation(s)
- Aaron Lerner
- Chaim Sheba Medical Center, The Zabludowicz Research Center for Autoimmune Diseases, Research Department, Ramat Gan 52621, Israel;
- Research Department, Ariel University, Ariel 40700, Israel
| | - Carina Benzvi
- Chaim Sheba Medical Center, The Zabludowicz Research Center for Autoimmune Diseases, Research Department, Ramat Gan 52621, Israel;
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20
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Wessels M, Dolinsek J, Castillejo G, Donat E, Riznik P, Roca M, Valitutti F, Veenvliet A, Mearin ML. Follow-up practices for children and adolescents with celiac disease: results of an international survey. Eur J Pediatr 2022; 181:1213-1220. [PMID: 34817672 DOI: 10.1007/s00431-021-04318-2] [Citation(s) in RCA: 16] [Impact Index Per Article: 5.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/04/2021] [Revised: 11/08/2021] [Accepted: 11/09/2021] [Indexed: 10/19/2022]
Abstract
Adequate follow-up in celiac disease is important to improve dietary compliance and treat disease-related symptoms and possible complications. However, data on the follow-up of celiac children is scarce. We aimed to assess current pediatric celiac follow-up practices across Europe. Pediatricians and pediatric gastroenterologists from 35 countries in Europe, Israel, Turkey, and Russia completed an anonymous survey which comprised a 52-item questionnaire developed by the ESPGHAN Special Interest Group on Celiac Disease. A total of 911 physicians, the majority of whom exclusively worked in pediatric care (83%) and academic institutions (60%), completed the questionnaire. Mean age and mean experience with celiac care were 48.7 years (± 10.6) and 15.7 years (± 9.9), respectively. The vast majority (≥ 92%) always assessed anthropometry, dietary adherence, and tissue-transglutaminase IgA-antibodies at every visit, with the first visit being between 3 and 6 months after diagnosis. Other parameters (% always tested) were as follows: complete blood count (60%), iron status (48%), liver enzymes (42%), thyroid function (38%), and vitamin D (26%). Quality of life was never assessed by 35% of the responding physicians. Transition to adult care was mostly completed via a written transition report (37%) or no formal transition at all (27%).Conclusions: Follow-up of celiac children and adolescents in Europe may be improved, especially regarding a more rational use of (laboratory) tests, dietary and QoL assessment, and transition to adult care. Evidence-based advice from international scientific societies is needed. What is Known: • Follow-up in celiac disease is important to treat disease-related symptoms, improve dietary compliance, and prevent possible complications. • There is a lack of consensus about the appropriate follow-up. What is New: • Almost all European physicians assess anthropometry, tissue-transglutaminase IgA-antibodies, and dietary adherence at every visit, but there are large variations in other follow-up aspects. • Follow-up could be improved by a more rational use of (laboratory) tests, increased intention to dietary compliance, and quality of life together with transition programs to adult care.
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Affiliation(s)
- Margreet Wessels
- Department of Pediatrics, Rijnstate Hospital Arnhem, Wagnerlaan 55, 6815 AD, Arnhem, The Netherlands.
| | - Jernej Dolinsek
- Department of Pediatrics, Gastroenterology, Hepatology and Nutrition Unit, University Medical Centre Maribor, Maribor, Slovenia.,Department of Pediatrics, Medical Faculty, University Medical Centre Maribor, Maribor, Slovenia
| | - Gemma Castillejo
- Pediatric Gastroenterology Unit, Hospital Universitario Sant Joan de Reus, Reus, Spain
| | - Ester Donat
- Pediatric Gastroenterology and Hepatology Unit, Hospital Universitari I Politècnic La Fe, Valencia, Spain
| | - Petra Riznik
- Department of Pediatrics, Gastroenterology, Hepatology and Nutrition Unit, University Medical Centre Maribor, Maribor, Slovenia
| | - Maria Roca
- Celiac Disease and Digestive Immunopathology Unit, Instituto de Investigación Sanitaria La Fe, Valencia, Spain
| | | | - Anne Veenvliet
- Department of Pediatrics, Rijnstate Hospital Arnhem, Wagnerlaan 55, 6815 AD, Arnhem, The Netherlands
| | - M Luisa Mearin
- Department of Pediatrics, Leiden University Medical Center, Leiden, The Netherlands
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21
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Affiliation(s)
- Tracy R Ediger
- Department of Clinical Pediatrics, The Ohio State College of Medicine, and Division of Gastroenterology, Hepatology, and Nutrition, Nationwide Children's Hospital, Columbus, OH
| | - Ivor D Hill
- Department of Clinical Pediatrics, The Ohio State College of Medicine, and Division of Gastroenterology, Hepatology, and Nutrition, Nationwide Children's Hospital, Columbus, OH
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22
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Tittel SR, Dunstheimer D, Hilgard D, Knauth B, Fröhlich-Reiterer E, Galler A, Wurm M, Holl RW, For the DPV Initiative. Coeliac disease is associated with depression in children and young adults with type 1 diabetes: results from a multicentre diabetes registry. Acta Diabetol 2021; 58:623-631. [PMID: 33483854 PMCID: PMC8076130 DOI: 10.1007/s00592-020-01649-8] [Citation(s) in RCA: 10] [Impact Index Per Article: 2.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/14/2020] [Accepted: 11/28/2020] [Indexed: 02/06/2023]
Abstract
AIMS To analyse the association between coeliac disease (CD) and depression in children, adolescents, and young adults with type 1 diabetes (T1D). METHODS We included 79,067 T1D patients aged 6-20 years, with at least six months of diabetes duration, and treatment data between 1995 and 2019 were documented in the diabetes patient follow-up registry. We categorized patients into four groups: T1D only (n = 73,699), T1 + CD (n = 3379), T1D + depression (n = 1877), or T1D + CD + depression (n = 112). RESULTS CD and depression were significantly associated (adjusted OR: 1.25 [1.03-1.53]). Females were more frequent in both the depression and the CD group compared with the T1D only group. Insulin pumps were used more frequently in T1D + CD and T1D + depression compared with T1D only (both p < .001). HbA1c was higher in T1D + depression (9.0% [8.9-9.0]), T1D + CD + depression (8.9% [8.6-9.2]), both compared with T1D only (8.2% [8.2-8.2], all p < .001). We found comorbid autism, attention deficit hyperactivity disorder, anxiety, schizophrenia, and eating disorders more frequently in the T1D + CD + depression group compared with T1D only (all p < .001). CONCLUSIONS CD and depression are associated in young T1D patients. The double load of T1D and CD may lead to an increased risk for depression. Depression was associated with additional psychological and neurological comorbidities. Aside from imperative CD screening after T1D diagnosis and regular intervals, depression screening might be helpful in routine care, especially in patients with diagnosed CD.
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Affiliation(s)
- Sascha René Tittel
- Institute for Epidemiology and Medical Biometry, ZIBMT, Ulm University, Albert-Einstein-Allee 41, 89081 Ulm, Germany
- German Center for Diabetes Research (DZD), Munich-Neuherberg, Germany
| | - Désirée Dunstheimer
- Paediatrics and Adolescent Medicine, Medical Faculty University of Augsburg, Augsburg, Germany
| | | | - Burkhild Knauth
- Department of Pediatrics and Adolescent Medicine, CJD Berchtesgaden, Berchtesgaden, Germany
| | - Elke Fröhlich-Reiterer
- Division of General Pediatrics, Department of Paediatrics and Adolescent Medicine, Medical University Graz, Graz, Austria
| | - Angela Galler
- Charité - Universitätsmedizin Berlin, corporate member of Freie Universität Berlin, Humboldt-Universität zu Berlin, and Berlin Institute of Health, Berlin, Germany
| | - Michael Wurm
- Clinic St. Hedwig, University Children’s Hospital Regensburg (KUNO Clinics), University of Regensburg, Regensburg, Germany
| | - Reinhard Walter Holl
- Institute for Epidemiology and Medical Biometry, ZIBMT, Ulm University, Albert-Einstein-Allee 41, 89081 Ulm, Germany
- German Center for Diabetes Research (DZD), Munich-Neuherberg, Germany
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23
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Shani M, Kraft L, Müller M, Boehnke K. The potential benefits of camps for children and adolescents with celiac disease on social support, illness acceptance, and health-related quality of life. J Health Psychol 2020; 27:1635-1645. [PMID: 33198516 DOI: 10.1177/1359105320968142] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/16/2022] Open
Abstract
Among N = 165 14-22-year-old bearers of celiac disease (CD), the German-based study examined if participation in camps for children with CD is related to higher CD-related quality of life (CD-QoL); N = 48 of the study participants attended at least one camp. Camp participation was found to be related to higher CD-QoL, an effect mediated by having more friends with CD and by perceiving higher social support. Camp participation was also associated with higher illness acceptance and lower anxiety. Results show the potential benefits of recreational activities in CD treatment, to be further examined in experimental research.
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Affiliation(s)
- Maor Shani
- Jacobs University Bremen, Germany.,Osnabrück University, Germany
| | | | | | - Klaus Boehnke
- Jacobs University Bremen, Germany.,National Research University Higher School of Economics, Russia
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24
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Abstract
PURPOSE OF REVIEW The purpose of this review is to describe current updates in celiac disease. RECENT FINDINGS Recent developments in the understanding of the pathogenesis of celiac disease continue to emerge that may implicate the role of gluten exposure. Several studies have shown that the amount of gluten consumed by the infant may affect the age of onset of celiac disease in genetically predisposed individuals. New guidelines from the European Society of Paediatric Gastroenterology, Hepatology and Nutrition allow serology-based celiac diagnosis, omitting endoscopic biopsies, in children. Recent data and updated guidelines in adults no longer support biopsies in all patients who are genetically susceptible with celiac disease who have been identified by serology with clinical signs and symptoms of celiac disease. A new assay was identified in the immune response to epitopes of the tissue transglutaminase-deamidated gliadin peptide complex. In addition, a recent study shows that serum IL-2 elevations correlate with timing and severity of symptoms after gluten ingested in celiac disease patients. Measuring gluten immunogenic peptides (GIPs) in the stool of celiac patients may help monitor adherence to a gluten-free diet (GFD). Of importance, we should be aware that the quality of life is affected in celiac disease patients. During adolescence, the education on the importance of long-term follow-up with an adult gastroenterologist is associated with more successful rates of medical care transition for young adults with celiac disease. Latiglutenase, an orally administered mixture of two gluten-specific recombinant proteases that degrades gluten proteins into small physiologically irrelevant fragments, is currently in a phase 2 trial. Latiglutenase has shown to be safe and effective in reducing symptoms of celiac disease patients upon a GFD with improvement of quality of life. Lastly, a recent study describes a mouse model that is characteristic of celiac disease. SUMMARY Our knowledge of celiac disease continues to grow with increasing evidence of contributory factors to its pathogenesis. There is some evidence that the quantity ingested of gluten by the infant effects the age of onset of celiac disease in genetically susceptible patients. Changes have been made to the guidelines in the diagnosis of celiac disease proposed by new studies. Recent studies have shown the significant effects on quality of life for celiac patients. As improved laboratory methods continue to be developed, these tests can have utility in both diagnosis of celiac disease and monitoring adherence to the GFD. Current therapeutic trials offer promising nondietary treatment for celiac patients. The development of an animal model can provide a better understanding of the pathogenesis of celiac disease.
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Zhang C, Shao H, Peng X, Liu T, Tan Z. Microbiotal characteristics colonized in intestinal mucosa of mice with diarrhoea and repeated stress. 3 Biotech 2020; 10:372. [PMID: 32832332 PMCID: PMC7399726 DOI: 10.1007/s13205-020-02368-1] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/23/2020] [Accepted: 07/27/2020] [Indexed: 12/27/2022] Open
Abstract
To understand the role of intestinal mucosal microbiota on mental stress-related diarrhoea, we collected the intestinal mucosa of mice treated with Folium senna extract gavage combined with restraint and tail pinch stress for 7 days; and intestinal mucosal microbiota characteristics were analyzed by 16S rRNA Pacbio SMRT gene full-length sequencing. The results showed that the diversity (i.e., alpha diversity including the Chao1, Simpson, ACE, and Shannon indices and beta diversity including the NMDS of weighted UniFrac distances) and composition of the microbial community in the intestinal mucosa of mice with diarrhoea and repeated stress changed significantly (P < 0.05). In the co-occurrence network, Staphylococcus sciuri and Escherichia fergusonii was identified as putative keystone species. Moreover, the characteristics of the intestinal microbial species was analyzed by LEfSe, Metastats, and group difference, and ten altered gut microbiota species can be used as characteristic microbes in the mice with diarrhoea and repeated stress: the abundances of Stigmatella aurantiaca, Candidatus arthromitus sp. SFB-mouse, Erythrobacter gaetbuli, Desulfitobacterium hafniense, Ochrobactrum pituitosum, and Candidatus arthromitus sp. SFB-mouse-NL in the model group were significantly lower than those in the control group (P < 0.05); whereas Microbacterium dextranolyticum, Klebsiella pneumoniae, Escherichia sp. BBDP27, and Streptococcus danieliae were enriched in the control group (P < 0.05). Collectively, mental stress-related diarrhoea increased the intestinal microbiota diversity. The species associated with mental stress-related diarrhoea including Microbacterium dextranolyticum, Klebsiella pneumoniae, Escherichia sp. BBDP27, and Streptococcus danieliae were significantly enriched; while the species which are beneficial to mental stress-related diarrhoea are Stigmatella aurantiaca, Candidatus arthromitus sp. SFB-mouse, Erythrobacter gaetbuli, Desulfitobacterium hafniense, Ochrobactrum pituitosum, and Candidatus arthromitus sp. SFB-mouse-NL for its significantly depleted.
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Affiliation(s)
- Chenyang Zhang
- Hunan University of Chinese Medicine, Xueshi Road 300, Yuelu District, Changsha, 410208 Hunan Province China
| | - Haoqing Shao
- Hunan University of Chinese Medicine, Xueshi Road 300, Yuelu District, Changsha, 410208 Hunan Province China
| | - Xinxin Peng
- The First Hospital of Hunan University of Chinese Medicine, Changsha, 410007 Hunan Province China
| | - Tianhao Liu
- College of Chinese Medicine, Jinan University, No. 601 Huangpu Avenue West, Tianhe District, Guangzhou, 510632 Guangdong China
| | - Zhoujin Tan
- Hunan University of Chinese Medicine, Xueshi Road 300, Yuelu District, Changsha, 410208 Hunan Province China
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Araya M, Bascuñán KA, Alarcón-Sajarópulos D, Cabrera-Chávez F, Oyarzún A, Fernández A, Ontiveros N. Living with Gluten and Other Food Intolerances: Self-Reported Diagnoses and Management. Nutrients 2020; 12:nu12061892. [PMID: 32604710 PMCID: PMC7353382 DOI: 10.3390/nu12061892] [Citation(s) in RCA: 8] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/03/2020] [Revised: 06/15/2020] [Accepted: 06/18/2020] [Indexed: 02/07/2023] Open
Abstract
People suffering from a food intolerance (FI) tend to initiate restrictive diets such as a gluten-free diet (GFD), to alleviate their symptoms. To learn about how people live with these problems in daily life (independent of their medical diagnoses), 1203 participants answered a previously validated questionnaire and were divided into: G1 (those self-reporting symptoms after gluten consumption) and G2 (those informing no discomfort after gluten consumption). Self-reported clinical characteristics, diagnoses and diets followed were registered. Twenty nine percent referred some FI (8.5% in G1). In G1, self-reported diagnoses were more frequent (p < 0.0001), including a high proportion of eating and mood disorders. Diagnoses were reported to be given by a physician, but GFD was indicated by professional and nonprofessional persons. In G2, despite declaring no symptoms after gluten consumption, 11.1% followed a GFD. The most frequent answer in both groups was that GFD was followed “to care for my health”, suggesting that some celiac patients do not acknowledge it as treatment. Conclusion: close to one third of the population report suffering from some FI. Those perceiving themselves as gluten intolerant report more diseases (p < 0.0001). A GFD is followed by ~11% of those declaring no symptoms after gluten ingestion. This diet is perceived as a healthy eating option.
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Affiliation(s)
- Magdalena Araya
- Institute of Nutrition and Food Technology (INTA), University of Chile, Santiago 7830490, Chile; (K.A.B.); (D.A.-S.); (A.O.)
- Correspondence: (M.A.); (N.O.); Tel.: +569-9539-5667 (M.A.); +52-642-425-9950 (N.O.)
| | - Karla A. Bascuñán
- Institute of Nutrition and Food Technology (INTA), University of Chile, Santiago 7830490, Chile; (K.A.B.); (D.A.-S.); (A.O.)
- Department of Nutrition, Faculty of Medicine, University of Chile, Santiago 8380453, Chile;
| | - Dana Alarcón-Sajarópulos
- Institute of Nutrition and Food Technology (INTA), University of Chile, Santiago 7830490, Chile; (K.A.B.); (D.A.-S.); (A.O.)
- Faculty of Nutrition and Gastronomy Sciences, Master of Science Graduate Program in Nutrition and Medicinal Foods, University of Sinaloa, Culiacán, Sinaloa 80019, Mexico;
| | - Francisco Cabrera-Chávez
- Faculty of Nutrition and Gastronomy Sciences, Master of Science Graduate Program in Nutrition and Medicinal Foods, University of Sinaloa, Culiacán, Sinaloa 80019, Mexico;
| | - Amaya Oyarzún
- Institute of Nutrition and Food Technology (INTA), University of Chile, Santiago 7830490, Chile; (K.A.B.); (D.A.-S.); (A.O.)
| | - Alan Fernández
- Department of Nutrition, Faculty of Medicine, University of Chile, Santiago 8380453, Chile;
| | - Noé Ontiveros
- Division of Sciences and Engineering, Department of Chemical, Biological and Agricultural Sciences, Clinical and Research Laboratory (LACIUS, URS), University of Sonora, Navojoa, Sonora 85880, Mexico
- Correspondence: (M.A.); (N.O.); Tel.: +569-9539-5667 (M.A.); +52-642-425-9950 (N.O.)
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