Case Report: Reversible cholestatic steatohepatitis after two decades of gluten-free diet non-adherence in celiac disease
- 1Department of Nutrition, Fattouma Bourguiba University Hospital, University of Monastir, Monastir, Tunisia
- 2Department of Gastroenterology and Hepatology, Fattouma Bourguiba University Hospital, University of Monastir, Monastir, Tunisia
Abstract
IntroductionMild aminotransferase elevation is a recognized hepatic manifestation of celiac disease, whereas conjugated jaundice with steatohepatitis is uncommon. This case illustrates a rare hepatic phenotype associated with prolonged gluten exposure and severe malnutrition.MethodsWe report the presentation, diagnostic investigations, nutritional management and nine-month follow-up of a 38-year-old Tunisian woman with childhood-onset celiac disease and approximately 18 years of nonadherence to a gluten-free diet.ResultsThe patient presented with diarrhea, 20-kg weight loss, a body mass index of 16 kg/m2 and jaundice. Total and conjugated bilirubin were 192 and 168 μmol/L, respectively. Ultrasonography showed steatosis without biliary dilatation. Duodenal biopsy demonstrated villous atrophy; liver biopsy revealed approximately 40% macrovesicular steatosis, inflammatory injury and moderate fibrosis without cholangitis. A strict gluten-free diet and nutritional rehabilitation were initiated. Aminotransferases rose transiently after a seven-day parenteral nutrition course; an adapted RUCAM score of 4 indicated only a possible contribution. Jaundice and diarrhea resolved by two months. At nine months, body mass index was 25 kg/m2; bilirubin, aminotransferases and alkaline phosphatase were normal, while gamma-glutamyl transferase remained minimally elevated.DiscussionProlonged gluten exposure and severe malnutrition may be associated with reversible cholestatic steatohepatitis. Causal attribution remains cautious because several alternative hepatobiliary diagnoses could not be definitively excluded and the proposed mechanisms were not directly measured.
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BibTeX
@article{sallem2026case,
title = {{Case Report: Reversible cholestatic steatohepatitis after two decades of gluten-free diet non-adherence in celiac disease}},
author = {Om Kolthoum Sallem and Wided Bouhlel and Nabil Ben Chaabene and Leila Safer},
journal = {Frontiers in Nutrition},
year = {2026},
month = nov,
doi = {10.3389/fnut.2026.1942195},
url = {https://doi.org/10.3389/fnut.2026.1942195},
note = {CC BY},
}
RIS
TY - JOUR
TI - Case Report: Reversible cholestatic steatohepatitis after two decades of gluten-free diet non-adherence in celiac disease
AU - Om Kolthoum Sallem
AU - Wided Bouhlel
AU - Nabil Ben Chaabene
AU - Leila Safer
PY - 2026
DA - 2026/11/06
JO - Frontiers in Nutrition
DO - 10.3389/fnut.2026.1942195
UR - https://doi.org/10.3389/fnut.2026.1942195
AB - IntroductionMild aminotransferase elevation is a recognized hepatic manifestation of celiac disease, whereas conjugated jaundice with steatohepatitis is uncommon. This case illustrates a rare hepatic phenotype associated with prolonged gluten exposure and severe malnutrition.MethodsWe report the presentation, diagnostic investigations, nutritional management and nine-month follow-up of a 38-year-old Tunisian woman with childhood-onset celiac disease and approximately 18 years of nonadherence to a gluten-free diet.ResultsThe patient presented with diarrhea, 20-kg weight loss, a body mass index of 16 kg/m2 and jaundice. Total and conjugated bilirubin were 192 and 168 μmol/L, respectively. Ultrasonography showed steatosis without biliary dilatation. Duodenal biopsy demonstrated villous atrophy; liver biopsy revealed approximately 40% macrovesicular steatosis, inflammatory injury and moderate fibrosis without cholangitis. A strict gluten-free diet and nutritional rehabilitation were initiated. Aminotransferases rose transiently after a seven-day parenteral nutrition course; an adapted RUCAM score of 4 indicated only a possible contribution. Jaundice and diarrhea resolved by two months. At nine months, body mass index was 25 kg/m2; bilirubin, aminotransferases and alkaline phosphatase were normal, while gamma-glutamyl transferase remained minimally elevated.DiscussionProlonged gluten exposure and severe malnutrition may be associated with reversible cholestatic steatohepatitis. Causal attribution remains cautious because several alternative hepatobiliary diagnoses could not be definitively excluded and the proposed mechanisms were not directly measured.
SN - 2296-861X
ER -