Suppr超能文献

一名患有脊髓小脑共济失调21型新突变的巴林儿童反复出现急性肝衰竭。

Recurrent Acute Liver Failure in a Bahraini Child With a Novel Mutation of Spinocerebellar Ataxia-21.

作者信息

Isa Hasan M, Alkaabi Jawaher F, Alhammadi Wasan H, Marjan Khadija A

机构信息

Department of Pediatrics, Arabian Gulf University, Manama, BHR.

Department of Pediatrics, Salmaniya Medical Complex, Manama, BHR.

出版信息

Cureus. 2023 Mar 16;15(3):e36249. doi: 10.7759/cureus.36249. eCollection 2023 Mar.

Abstract

Acute liver failure (ALF) in children is a rare life-threatening condition. ALF is caused by different etiologies. The most common causes are drug-induced liver injury, infections, and metabolic diseases. Other rare causes of ALF are genetic disorders including spinocerebellar ataxia-21 (SCAR21). Herein, we describe the first Bahraini child who was diagnosed with a novel homozygous mutation in the SCYL1 gene. He was admitted to the hospital twice by the age of two and five years due to acute hepatic failure triggered by a febrile illness. Drug-induced, infectious causes, and metabolic diseases were excluded. The liver function then gradually recovered. The patient had delayed gross motor development as he started to walk at 20 months of age. After the first episode of ALF, he had progressive difficulty in walking leading to frequent falls and ending with a complete inability to walk. A whole-exome sequencing (WES) test revealed that the patient has previously unreported autosomal recessive pathogenic non-sense variation c.895A>T (p.Lys299Ter) in exon 7 of the SCYL1 gene in a homozygous status. It is confirmed that the pathogenicity of this variant in the SCYL1 gene was associated with SCAR21 disease.

摘要

儿童急性肝衰竭(ALF)是一种罕见的危及生命的病症。ALF由不同病因引起。最常见的病因是药物性肝损伤、感染和代谢性疾病。ALF的其他罕见病因是遗传性疾病,包括脊髓小脑共济失调21型(SCAR21)。在此,我们描述了首例被诊断出SCYL1基因存在新型纯合突变的巴林儿童。他在两岁和五岁时因发热性疾病引发急性肝衰竭而两次入院。排除了药物性、感染性病因和代谢性疾病。随后肝功能逐渐恢复。该患者大运动发育迟缓,20个月时才开始走路。首次发生ALF后,他行走逐渐困难,频繁跌倒,最终完全无法行走。全外显子组测序(WES)检测显示,该患者在SCYL1基因第7外显子中存在此前未报道的纯合状态的常染色体隐性致病无义变异c.895A>T(p.Lys299Ter)。已证实该SCYL1基因变异的致病性与SCAR21疾病相关。

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验