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PMID: 41158795 已发表 · epublish 英语

Recurrent fever-associated acute liver failure and cranial dysmorphism in children caused by RINT1 gene mutations: a rare case report.

Frontiers in pediatrics ·第 13 卷 ·2025-00-00

Cui Y, Abudu F, Simijiang Y

摘要

Mutations in the RINT1 gene represent a rare genetic cause of recurrent fever-associated acute liver failure (ALF) accompanied by skeletal abnormalities in infants and children. We report the case of a 9-month-old infant presenting with multisystem involvement, primarily characterized by recurrent fever-associated ALF and cranial dysmorphism, due to compound heterozygous mutations in the RINT1 gene. The patient exhibited abnormal liver function tests and coagulation dysfunction following febrile episodes. Over a period of more than one year, the patient initially experienced two episodes of acute liver injury, followed by two episodes of ALF, with progressively worsening clinical manifestations. Whole-exome sequencing (WES) identified compound heterozygous variants in the RINT1 gene (exons 12-14 deletion; intron 11, c.1672-1G > T, p.?), consistent with a diagnosis of infantile liver failure syndrome-3 (ILFS3). Between episodes, liver function failed to return fully to baseline and was accompanied by growth retardation, delayed psychomotor development, cranial dysmorphism, and beak-like deformities of vertebral bodies. This case highlights the critical role of RINT1 mutations in the pathogenesis of recurrent fever-associated ALF and emphasizes the importance of recognizing associated skeletal developmental abnormalities, including cranial dysmorphism. Early genetic diagnosis and prompt antipyretic intervention may mitigate liver injury and improve long-term outcomes. By documenting cranial dysmorphism in this context, we aim to expand the recognized phenotypic spectrum of ILFS3 and improve clinical awareness among pediatricians and geneticists.

关键词
RINT1 gene acute liver failure coagulation dysfunction cranial dysmorphism infantile liver failure syndrome type 3
文献信息
期刊
Frontiers in pediatrics
期刊简称
Front Pediatr
ISSN
2296-2360
发表日期
2025-00-00
语言
英语
国家/地区
Switzerland
NLM ID
101615492
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