Benign Recurrent Intrahepatic Cholestasis Type 1 with Novel Gene Mutation Complicated by Distal Renal Tubular Acidosis: A Case Report

Introduction: Benign recurrent intrahepatic cholestasis (BRIC) is a rare autosomal recessive inherited disorder characterized by intermittent jaundice episodes. Case Presentation: We report a 25-year-old male patient with numerous hospitalizations for jaundice. The diagnosis of BRIC type...

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Bibliographic Details
Main Authors: Van Huy Vo, Chuong Dinh Nguyen, Sang The Phan, Phong Tien Quach, Thu Dang Anh Phan, Khue Minh Nguyen, Thong Duy Vo, Hoang Huu Bui
Format: Article
Language:English
Published: Karger Publishers 2025-04-01
Series:Case Reports in Gastroenterology
Online Access:https://karger.com/article/doi/10.1159/000544786
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Summary:Introduction: Benign recurrent intrahepatic cholestasis (BRIC) is a rare autosomal recessive inherited disorder characterized by intermittent jaundice episodes. Case Presentation: We report a 25-year-old male patient with numerous hospitalizations for jaundice. The diagnosis of BRIC type 1 was established by liver biopsy, genetic analysis, and the exclusion of alternative etiologies of cholestasis. A novel missense heterozygous variant was identified in the ATP8B1 gene (c.2081T>A). The acute cholestatic attack, along with associated complications such as distal renal tubular acidosis and acute pancreatitis, was successfully managed with plasmapheresis and rifampicin. Conclusion: Novel mutations that differ from those documented in the literature in combination with renal tubular acidosis may enhance our comprehension of this topic.
ISSN:1662-0631