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一名患有辅酶Q10缺乏相关肾小球病儿童的家族性血栓性微血管病

Familial thrombotic microangiopathy in a child with coenzyme Q10 deficiency-associated glomerulopathy.

作者信息

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

机构信息

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.

出版信息

Pediatr Nephrol. 2025 Feb;40(2):377-380. doi: 10.1007/s00467-024-06496-1. Epub 2024 Sep 3.

DOI:10.1007/s00467-024-06496-1
PMID:39225810
Abstract

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 Eculizumab was withdrawn, and supportive therapies, including ubiquinol (50 mg/kg/day) and vitamins, were optimized.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.

摘要

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。

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本文引用的文献

1
Oral Coenzyme Q10 supplementation leads to better preservation of kidney function in steroid-resistant nephrotic syndrome due to primary Coenzyme Q10 deficiency.口服辅酶 Q10 补充剂可导致原发性辅酶 Q10 缺乏引起的类固醇抵抗性肾病综合征的肾功能更好地保留。
Kidney Int. 2022 Sep;102(3):604-612. doi: 10.1016/j.kint.2022.04.029. Epub 2022 May 25.
2
Variation of the clinical spectrum and genotype-phenotype associations in Coenzyme Q10 deficiency associated glomerulopathy.辅酶 Q10 缺乏相关性肾小球病的临床表型谱和基因型-表型相关性的变异。
Kidney Int. 2022 Sep;102(3):592-603. doi: 10.1016/j.kint.2022.02.040. Epub 2022 Apr 26.
3
Two cases of idiopathic steroid-resistant nephrotic syndrome complicated with thrombotic microangiopathy.
两例特发性类固醇抵抗型肾病综合征合并血栓性微血管病。
BMC Nephrol. 2020 Aug 3;21(1):323. doi: 10.1186/s12882-020-01985-5.
4
Extra-renal manifestations of atypical hemolytic uremic syndrome.非肾性表现的非典型溶血尿毒综合征。
Pediatr Nephrol. 2019 Aug;34(8):1337-1348. doi: 10.1007/s00467-018-4039-7. Epub 2018 Aug 14.
5
Genetic bases and clinical manifestations of coenzyme Q10 (CoQ 10) deficiency.辅酶Q10(CoQ 10)缺乏症的遗传基础和临床表现。
J Inherit Metab Dis. 2015 Jan;38(1):145-56. doi: 10.1007/s10545-014-9749-9. Epub 2014 Aug 5.