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      Celiac disease and Sjögren’s syndrome: A case report and review of literature

      case-report

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          Abstract

          BACKGROUND

          Celiac disease (CD) is a systemic, chronic immune-mediated disease triggered by gluten ingestion in genetically-susceptible individuals, with a prevalence of 1% worldwide. Sjogren's syndrome (SS) is also a systemic autoimmune disease, mainly characterized by ocular and oral sicca symptoms and signs. Sharing a common genetic background, CD and SS are known associated autoimmune diseases, but currently available guidelines are not reporting it.

          CASE SUMMARY

          We report the case of a 39-year-old woman, who was in the care of her rheumatologist for 2 years with SS. On routine follow-up she was found to have iron deficiency, without anemia. She had no gastrointestinal complaints and denied any obvious source of blood loss. IgA tissue transglutaminase antibodies were positive and endoscopy with duodenal biopsies revealed crypt hyperplasia and villous atrophy. A diagnosis of CD was set and gluten-free diet was recommended.

          CONCLUSION

          We present a review of existing data in the literature regarding the association of the two diseases, summarizing prevalence studies of CD in SS patients and the other way around. Screening recommendations and future research perspectives are also discussed, highlighting clinically relevant unanswered questions with respect to the association of CD with SS.

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          Prevalence of celiac disease in at-risk and not-at-risk groups in the United States: a large multicenter study.

          Alessio Fasano, Irene Berti, Tania Gerarduzzi … (2003)
          Celiac disease (CD) is an immune-mediated enteropathic condition triggered in genetically susceptible individuals by the ingestion of gluten. Although common in Europe, CD is thought to be rare in the United States, where there are no large epidemiologic studies of its prevalence. The aim of this study was to determine the prevalence of CD in at-risk and not-at-risk groups in the United States. Serum antigliadin antibodies and anti-endomysial antibodies (EMA) were measured. In EMA-positive subjects, human tissue transglutaminase IgA antibodies and CD-associated human leukocyte antigen DQ2/DQ8 haplotypes were determined. Intestinal biopsy was recommended and performed whenever possible for all EMA-positive subjects. A total of 13 145 subjects were screened: 4508 first-degree and 1275 second-degree relatives of patients with biopsy-proven CD, 3236 symptomatic patients (with either gastrointestinal symptoms or a disorder associated with CD), and 4126 not-at-risk individuals. In at-risk groups, the prevalence of CD was 1:22 in first-degree relatives, 1:39 in second-degree relatives, and 1:56 in symptomatic patients. The overall prevalence of CD in not-at-risk groups was 1:133. All the EMA-positive subjects who underwent intestinal biopsy had lesions consistent with CD. Our results suggest that CD occurs frequently not only in patients with gastrointestinal symptoms, but also in first- and second-degree relatives and patients with numerous common disorders even in the absence of gastrointestinal symptoms. The prevalence of CD in symptomatic patients and not-at-risk subjects was similar to that reported in Europe. Celiac disease appears to be a more common but neglected disorder than has generally been recognized in the United States.
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            Celiac disease: a comprehensive current review

            Giacomo Caio, Umberto Volta, Anna Sapone … (2019)
            Background Celiac disease remains a challenging condition because of a steady increase in knowledge tackling its pathophysiology, diagnosis, management, and possible therapeutic options. Main body A major milestone in the history of celiac disease was the identification of tissue transglutaminase as the autoantigen, thereby confirming the autoimmune nature of this disorder. A genetic background (HLA-DQ2/DQ8 positivity and non-HLA genes) is a mandatory determinant of the development of the disease, which occurs with the contribution of environmental factors (e.g., viral infections and dysbiosis of gut microbiota). Its prevalence in the general population is of approximately 1%, with female predominance. The disease can occur at any age, with a variety of symptoms/manifestations. This multifaceted clinical presentation leads to several phenotypes, i.e., gastrointestinal, extraintestinal, subclinical, potential, seronegative, non-responsive, and refractory. Although small intestinal biopsy remains the diagnostic ‘gold standard’, highly sensitive and specific serological tests, such as tissue transglutaminase, endomysial and deamidated gliadin peptide antibodies, have become gradually more important in the diagnostic work-up of celiac disease. Currently, the only treatment for celiac disease is a life-long, strict gluten-free diet leading to improvement in quality of life, ameliorating symptoms, and preventing the occurrence of refractory celiac disease, ulcerative jejunoileitis, and small intestinal adenocarcinoma and lymphoma. Conclusions The present review is timely and provides a thorough appraisal of various aspects characterizing celiac disease. Remaining challenges include obtaining a better understanding of still-unclear phenotypes such as slow-responsive, potential (minimal lesions) and seronegative celiac disease. The identification of alternative or complementary treatments to the gluten-free diet brings hope for patients unavoidably burdened by diet restrictions.
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              Update on serologic testing in celiac disease.

              D L Leffler, Detlef Schuppan (2010)
              Contemporary serologic testing has revolutionized the field of celiac disease (CD). Highly accurate serologic assays have shown the prevalence of CD to be nearly 1:100 in many populations. These mostly ELISA (enzyme-linked immunosorbent assay)-based tests allow noninvasive screening and detection. However, the growing number of available serologic tests necessitates reevaluation of their predictive power as a single test or in combination. We review the available tests for CD, including antibodies against gliadin, endomysium, tissue transglutaminase, and deamidated gliadin, and the evidence for preferential use of specific tests in different settings. Despite several novel developments, standardized ELISA-based assays for IgA autoantibodies against tissue transglutaminase remain the test of choice for most populations. We discuss the need to develop tests for CD activity in order to assess the efficacy of upcoming nondietary therapies.
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                Author and article information

                Contributors
                Daniel Vasile Balaban
                Ancuta Mihai
                Alina Dima
                Alina Popp
                Mariana Jinga
                Ciprian Jurcut
                Journal
                Journal ID (nlm-ta): World J Clin Cases
                Journal ID (publisher-id): WJCC
                Title: World Journal of Clinical Cases
                Publisher: Baishideng Publishing Group Inc
                ISSN (Electronic): 2307-8960
                Publication date (Print): 26 September 2020
                Publication date (Electronic): 26 September 2020
                Volume: 8
                Issue: 18
                Pages: 4151-4161
                Affiliations
                Internal Medicine and Gastroenterology, Carol Davila University of Medicine and Pharmacy, Dr. Carol Davila Central Military Emergency University Hospital, Bucharest 020021, Romania
                Internal Medicine, Dr. Carol Davila Central Military Emergency University Hospital, Bucharest 010825, Romania
                Internal Medicine and Gastroenterology, Carol Davila University of Medicine and Pharmacy, Dr. Carol Davila Central Military Emergency University Hospital, Bucharest 020021, Romania
                Pediatrics, Carol Davila University of Medicine and PharmacyCarol Davila University of Medicine and Pharmacy, Bucharest 020021, Romania
                Alfred Rusescu Institute for Mother and Child Care, Bucharest 020021, Romania
                Tampere Center for Child Health Research, University of Tampere and Tampere University Hospital, Bucharest 020021, Romania
                Internal Medicine and Gastroenterology, Carol Davila University of Medicine and Pharmacy, Dr. Carol Davila Central Military Emergency University Hospital, Bucharest 020021, Romania. mariana_jinga@ 123456yahoo.com
                Internal Medicine, Dr. Carol Davila Central Military Emergency University Hospital, Bucharest 010825, Romania
                Author notes

                Author contributions: Jurcut C proposed the research idea; Balaban DV, Jurcut C, Mihai A, Popp A, and Jinga M were involved in the case management; Balaban DV, Mihai A, Popp A and Dima A performed the literature search and selection; Balaban DV, Mihai A, Popp A and Dima A drafted the manuscript; Jurcut C and Jinga M critically reviewed the manuscript; All authors approved the final version of the manuscript.

                Corresponding author: Mariana Jinga, MD, PhD, Professor, Internal Medicine and Gastroenterology, Carol Davila University of Medicine and Pharmacy, Dr. Carol Davila Central Military Emergency University Hospital, No. 37 Dionisie Lupu, Bucharest 020021, Romania. mariana_jinga@ 123456yahoo.com

                Article
                Other ID: jWJCC.v8.i18.pg4151
                DOI: 10.12998/wjcc.v8.i18.4151
                PMC ID: 7520766
                PubMed ID: 33024773
                SO-VID: 0b3deaad-769a-4a9f-841d-5a6a28621dd3
                Copyright © ©The Author(s) 2020. Published by Baishideng Publishing Group Inc. All rights reserved.
                License:

                This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial.

                History
                Date received : 27 February 2020
                Date revision received : 16 May 2020
                Date accepted : 25 August 2020
                Categories
                Subject: Case Report

                Keywords: celiac disease,sjögren syndrome,prevalence,autoimmunity,screening,antibodies,case report
                Data availability:
                Keywords: celiac disease, sjögren syndrome, prevalence, autoimmunity, screening, antibodies, case report

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