Evidence map›Paper›PMID 40200381›Full record

ArticleOrphanet journal of rare diseases2025

Clinical and molecular genetic characteristics of pediatric PFIC3 patients: three novel variants and prognosis for parental liver transplantation.

Jiqiang Hu, Chenyu Yang, Bingqian Tan, Qiang Xiong, Ying Le, Jianyang Hu, Haoming Wang, Xiaoke Dai, Mingman Zhang

Abstract readCase Reports
In one paragraph

Article in Orphanet journal of rare diseases, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

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2 · The registry

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3 · Its place in the literature

Who cites it

1 citing paper in PubMed.

  1. A Rare Nonsense Mutation in theJournal of clinical medicine · 2026
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4 · The record

Corrections and comments

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5 · Who and what money

Authors and funding

9 authors.

Jiqiang HuDepartment of Pediatric Hepatobiliary Surgery National Clinical Research Center for Child Health and Disorders, Ministry of Education Key Laboratory of Child Development and Disorders, Chongqing Key Laboratory of Pediatric Metabolism and Inflammatory Diseases, Children'S Hospital of Chongqing Medical University, Chongqin, China.
Chenyu YangDepartment of Pediatric Hepatobiliary Surgery National Clinical Research Center for Child Health and Disorders, Ministry of Education Key Laboratory of Child Development and Disorders, Chongqing Key Laboratory of Pediatric Metabolism and Inflammatory Diseases, Children'S Hospital of Chongqing Medical University, Chongqin, China.
Bingqian TanDepartment of Pediatric Hepatobiliary Surgery National Clinical Research Center for Child Health and Disorders, Ministry of Education Key Laboratory of Child Development and Disorders, Chongqing Key Laboratory of Pediatric Metabolism and Inflammatory Diseases, Children'S Hospital of Chongqing Medical University, Chongqin, China.
Qiang XiongDepartment of Pediatric Hepatobiliary Surgery National Clinical Research Center for Child Health and Disorders, Ministry of Education Key Laboratory of Child Development and Disorders, Chongqing Key Laboratory of Pediatric Metabolism and Inflammatory Diseases, Children'S Hospital of Chongqing Medical University, Chongqin, China.
Ying LeDepartment of Pediatric Hepatobiliary Surgery National Clinical Research Center for Child Health and Disorders, Ministry of Education Key Laboratory of Child Development and Disorders, Chongqing Key Laboratory of Pediatric Metabolism and Inflammatory Diseases, Children'S Hospital of Chongqing Medical University, Chongqin, China.
Jianyang HuDepartment of Pediatric Hepatobiliary Surgery National Clinical Research Center for Child Health and Disorders, Ministry of Education Key Laboratory of Child Development and Disorders, Chongqing Key Laboratory of Pediatric Metabolism and Inflammatory Diseases, Children'S Hospital of Chongqing Medical University, Chongqin, China.
Haoming WangDepartment of Pediatric Hepatobiliary Surgery National Clinical Research Center for Child Health and Disorders, Ministry of Education Key Laboratory of Child Development and Disorders, Chongqing Key Laboratory of Pediatric Metabolism and Inflammatory Diseases, Children'S Hospital of Chongqing Medical University, Chongqin, China.
Xiaoke DaiDepartment of Pediatric Hepatobiliary Surgery National Clinical Research Center for Child Health and Disorders, Ministry of Education Key Laboratory of Child Development and Disorders, Chongqing Key Laboratory of Pediatric Metabolism and Inflammatory Diseases, Children'S Hospital of Chongqing Medical University, Chongqin, China. xiaokedai2022@163.com.
Mingman ZhangDepartment of Pediatric Hepatobiliary Surgery National Clinical Research Center for Child Health and Disorders, Ministry of Education Key Laboratory of Child Development and Disorders, Chongqing Key Laboratory of Pediatric Metabolism and Inflammatory Diseases, Children'S Hospital of Chongqing Medical University, Chongqin, China. zhangmingman-a@163.com.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Progressive Familial Intrahepatic Cholestasis Type 3 (PFIC3) is a rare inherited liver disease caused by a mutation in the ABCB4 gene, leading to dysfunction of multidrug resistance protein 3 (MDR3). The earlier the onset of PFIC3 in children is, the more severe the prognosis. The diagnosis of PFIC3 is typically based on clinical symptoms, laboratory tests, and imaging assessments, with final confirmation requiring genetic testing. The aim of this study was to investigate the associations between genetic mutations in PFIC3 and clinical features, molecular genetics, and liver histopathology to improve early recognition and understanding of this disease. By analysing the data of three children with PFIC3 who underwent parental liver transplantation, we were able to gain a deeper understanding of the complexity and diversity of the disease. With respect to molecular genetics, we identified five mutation sites in the ABCB4 gene, including three newly discovered mutations. Immunohistochemical analysis revealed reduced expression of the MDR3 protein in child 1 and no expression in child 2 or child 3, revealing an intrinsic link between the ABCB4 gene and the MDR3 protein. Histopathologically, all three patients presented with significant portal vein fibrosis or cholestatic liver cirrhosis. In conclusion, this study emphasizes the importance of molecular genetic and pathological evaluation of patients with PFIC3 mutations and elucidates the impact of these three mutations on the course of the disease in children, for whom early symptomatic treatment and early preparation for liver transplantation are options worth considering.

Indexed as

ATP Binding Cassette Transporter, Subfamily BCholestasis, IntrahepaticLiver TransplantationChildHumansMutationPrognosisATP Binding Cassette Transporter, Subfamily Bmultidrug resistance protein 3ABCB4CholestasisMDR3Progressive familial intrahepatic cholestasis type 3

Identifiers

PMID40200381
PMCPMC11977866

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.