Lin Kyle Ying-Kit, Lam Ching-Wan, Chan Eugene Yu-Hin, Lee Mianne, Chung Brian Hon-Yin, Fung Cheuk-Wing, Rodenburg Richard, Licht Christoph, Lap-Tak Ma Alison
Paediatric Nephrology, Department of Paediatrics and Adolescent Medicine, Hong Kong Children's Hospital, Hong Kong, SAR, China.
Department of Pathology, University of Hong Kong, Hong Kong, SAR, China.
Pediatr Nephrol. 2025 Feb;40(2):377-380. doi: 10.1007/s00467-024-06496-1. Epub 2024 Sep 3.
We report a child with biallelic COQ6 variants presenting with familial thrombotic microangiopathy (TMA). A Chinese boy presented with steroid-resistant nephrotic syndrome at 8 months old and went into kidney failure requiring peritoneal dialysis at 15 months old. He presented with hypertensive encephalopathy with the triad of microangiopathic haemolytic anaemia, thrombocytopenia, and acute on chronic kidney injury at 25 months old following a viral illness. Kidney biopsy showed features of chronic TMA. He was managed with supportive therapy and plasma exchanges and maintained on eculizumab. However, he had another TMA relapse despite complement inhibition a year later. Eculizumab was withdrawn, and supportive therapies, including ubiquinol (50 mg/kg/day) and vitamins, were optimized. He remained relapse-free since then for 4 years. Of note, his elder sister succumbed to multiple organ failure with histological evidence of chronic TMA at the age of 4. Retrospective genetic analysis revealed the same compound heterozygous variants in the COQ6 gene.
我们报告了一名患有双等位基因COQ6变异且表现为家族性血栓性微血管病(TMA)的儿童。一名中国男孩8个月大时出现类固醇抵抗性肾病综合征,15个月大时发展为肾衰竭,需要进行腹膜透析。25个月大时,在一次病毒感染后,他出现了伴有微血管病性溶血性贫血、血小板减少和急性慢性肾损伤三联征的高血压脑病。肾活检显示为慢性TMA特征。他接受了支持治疗和血浆置换,并持续使用依库珠单抗。然而,尽管一年后进行了补体抑制治疗,他还是再次出现了TMA复发。停用了依库珠单抗,并优化了包括泛醇(50mg/kg/天)和维生素在内的支持治疗。从那时起,他持续4年未再复发。值得注意的是,他的姐姐在4岁时死于多器官衰竭,组织学检查有慢性TMA的证据。回顾性基因分析显示COQ6基因存在相同的复合杂合变异。