PMID- 33744411 OWN - NLM STAT- MEDLINE DCOM- 20220207 LR - 20220207 IS - 2210-741X (Electronic) IS - 2210-7401 (Linking) VI - 45 IP - 3 DP - 2021 May TI - Adult autoimmune enteropathy in autoimmune hepatitis patient. Case report and literature review. PG - 101673 LID - S2210-7401(21)00052-8 [pii] LID - 10.1016/j.clinre.2021.101673 [doi] AB - Autoimmune enteropathy (AIE) is a rare disease characterized by prolonged diarrhea, vomiting and weight loss; although it is mainly a rare pediatric disease, over the years a number of adults have also been found to be affected. In this study, we present a case report of a 73-year-old woman with a history of autoimmune hepatitis, antinuclear (ANA) and positive anti-enterocyte antibodies (AEA), who has suffered two months of intractable diarrhea, nausea, anorexia and severe weight loss. The histological examination of the endoscopic duodenal mucosa biopsies revealed severe shortening and flattening of the villi, resulting in mucosal atrophy. The immunohistochemical study revealed a polymorphic lymphoid population, exhibiting a B cell (CD20+) phenotype in follicles and a T cell phenotype (CD3+) in the diffuse component within the lamina propria. Our patient had a complete recovery after two weeks of taking prednisone and following a gluten-rich diet. To our knowledge this is the first case of autoimmune enteropathy in adults with ANA and AEA 7 years after a diagnosis of autoimmune hepatitis. To date, the patient is still in clinical remission on a low dose of orally administered predinisone without any additional immunosuppression. CI - Copyright (c) 2021 Elsevier Masson SAS. All rights reserved. FAU - Iaquinto, Gaetano AU - Iaquinto G AD - Division of Gastroenterology, Department of Internal Medicine, S. Rita Hospital, Atripalda, Avellino, Italy. Electronic address: iaquintog@yahoo.it. FAU - Panico, Luigi AU - Panico L AD - Department of Pathology, AORN dei Colli, Monaldi Hospital, Naples, Italy. FAU - Luongo, Gelsomina AU - Luongo G AD - Department of Pathology, San G. Moscati Hospital, Avellino, Italy. FAU - Tenneriello, Valentina AU - Tenneriello V AD - Department of Pathology, San G. Moscati Hospital, Avellino, Italy. FAU - Iaquinto, Salvatore AU - Iaquinto S AD - Division of Gastroenterology, San Filippo Neri Hospital, Rome, Italy. FAU - Giardullo, Nicola AU - Giardullo N AD - Division of Gastroenterology, San G. Moscati Hospital, Avellino, Italy. FAU - Rotondi Aufiero, Vera AU - Rotondi Aufiero V AD - Institute of Food Sciences, CNR, Avellino, Italy. FAU - Mazzarella, Giuseppe AU - Mazzarella G AD - Institute of Food Sciences, CNR, Avellino, Italy. FAU - Rispoli, Raffaella AU - Rispoli R AD - Department of Mental and Physical Health and Preventive Medicine, Section of Human Anatomy, University of Campania "Luigi Vanvitelli", Naples, Italy. FAU - Lucariello, Angela AU - Lucariello A AD - Department of Sport Sciences and Wellness, University of Naples "Parthenope", Naples, Italy. FAU - Perna, Angelica AU - Perna A AD - Department of Medicine and Health Sciences "Vincenzo Tiberio", University of Molise, Campobasso, Italy. FAU - De Luca, Antonio AU - De Luca A AD - Department of Mental and Physical Health and Preventive Medicine, Section of Human Anatomy, University of Campania "Luigi Vanvitelli", Naples, Italy. LA - eng PT - Case Reports PT - Journal Article PT - Review DEP - 20210317 PL - France TA - Clin Res Hepatol Gastroenterol JT - Clinics and research in hepatology and gastroenterology JID - 101553659 RN - Autoimmune enteropathy SB - IM MH - Aged MH - Diarrhea MH - Female MH - *Hepatitis, Autoimmune/complications/diagnosis/drug therapy MH - Humans MH - *Polyendocrinopathies, Autoimmune/complications/diagnosis MH - Weight Loss OTO - NOTNLM OT - Autoimmune enteropathy OT - Autoimmune hepatitis OT - Diarrhea EDAT- 2021/03/22 06:00 MHDA- 2022/02/08 06:00 CRDT- 2021/03/21 20:57 PHST- 2020/05/28 00:00 [received] PHST- 2021/01/25 00:00 [revised] PHST- 2021/03/02 00:00 [accepted] PHST- 2021/03/22 06:00 [pubmed] PHST- 2022/02/08 06:00 [medline] PHST- 2021/03/21 20:57 [entrez] AID - S2210-7401(21)00052-8 [pii] AID - 10.1016/j.clinre.2021.101673 [doi] PST - ppublish SO - Clin Res Hepatol Gastroenterol. 2021 May;45(3):101673. doi: 10.1016/j.clinre.2021.101673. Epub 2021 Mar 17.