Case Report
Copyright ©The Author(s) 2020. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Sep 26, 2020; 8(18): 4151-4161
Published online Sep 26, 2020. doi: 10.12998/wjcc.v8.i18.4151
Celiac disease and Sjögren’s syndrome: A case report and review of literature
Daniel Vasile Balaban, Ancuta Mihai, Alina Dima, Alina Popp, Mariana Jinga, Ciprian Jurcut
Daniel Vasile Balaban, Alina Dima, Mariana Jinga, Internal Medicine and Gastroenterology, Carol Davila University of Medicine and Pharmacy, Dr. Carol Davila Central Military Emergency University Hospital, Bucharest 020021, Romania
Ancuta Mihai, Ciprian Jurcut, Internal Medicine, Dr. Carol Davila Central Military Emergency University Hospital, Bucharest 010825, Romania
Alina Popp, Pediatrics, Carol Davila University of Medicine and PharmacyCarol Davila University of Medicine and Pharmacy, Bucharest 020021, Romania
Alina Popp, Alfred Rusescu Institute for Mother and Child Care, Bucharest 020021, Romania
Alina Popp, Tampere Center for Child Health Research, University of Tampere and Tampere University Hospital, Bucharest 020021, Romania
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.
Informed consent statement: Informed written consent was obtained from the patient for publication of this report and any accompanying images.
Conflict-of-interest statement: The authors declare that they have no conflict of interest.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (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. See: http://creativecommons.org/licenses/by-nc/4.0/
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@yahoo.com
Received: February 27, 2020
Peer-review started: February 27, 2020
First decision: April 8, 2020
Revised: May 16, 2020
Accepted: August 25, 2020
Article in press: August 25, 2020
Published online: September 26, 2020
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.

Keywords: Celiac disease, Sjögren syndrome, Prevalence, Autoimmunity, Screening, Antibodies, Case report

Core Tip: Celiac disease (CD) and Sjögren's syndrome (SS) are associated autoimmune diseases, sharing a common genetic background. Although there is some evidence supporting the association of the two diseases, currently available guidelines are not sufficiently reporting it. We herein report a case of a patient with SS who was diagnosed with CD and summarize existing data in the literature regarding the association of the two diseases and discuss future research topics that remain to be answered with regard to this association. To the best of our knowledge this is the first literature review regarding these two pathologies concomitance.