Isa Hasan M, Alkaabi Jawaher F, Alhammadi Wasan H, Marjan Khadija A
Department of Pediatrics, Arabian Gulf University, Manama, BHR.
Department of Pediatrics, Salmaniya Medical Complex, Manama, BHR.
Cureus. 2023 Mar 16;15(3):e36249. doi: 10.7759/cureus.36249. eCollection 2023 Mar.
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.
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。