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SPG7显性遗传性视神经萎缩表型扩展:无痉挛性截瘫的婴儿性眼球震颤和视神经萎缩。

Expanding SPG7 dominant optic atrophy phenotype: Infantile nystagmus and optic atrophy without spastic paraplegia.

作者信息

Seo Yuri, Lim Hyun Taek, Lee Byung Joo, Han Jinu

机构信息

Department of Ophthalmology, Institute of Vision Research, Yongin Severance Hospital, Yonsei University College of Medicine, Yongin, South Korea.

Seoul Orthopia Eye Clinic, Seoul, South Korea.

出版信息

Am J Med Genet A. 2023 Feb;191(2):582-585. doi: 10.1002/ajmg.a.63037. Epub 2022 Nov 11.

Abstract

Spastic paraplegia is a neurodegenerative disorder characterized by progressive leg weakness and spasticity due to degeneration of corticospinal axons. SPG7 encodes paraplegin, and pathogenic variants in the gene cause hereditary spastic paraplegia as an autosomal recessive trait. Various ophthalmological findings including optic atrophy, ophthalmoplegia, or nystagmus have been reported in patients with spastic paraplegia type 7. We report a 15-year-old male patient with a novel heterozygous variant, c.1224T>G:p.(Asp408Glu) in SPG7 (NM_003119.3) causing early onset isolated optic atrophy and infantile nystagmus prior to the onset of neurological symptoms. Therefore, SPG7 should be considered a cause of infantile nystagmus with optic atrophy.

摘要

痉挛性截瘫是一种神经退行性疾病,其特征是由于皮质脊髓轴突变性导致进行性腿部无力和痉挛。SPG7编码 paraplegin,该基因的致病性变异导致遗传性痉挛性截瘫,呈常染色体隐性遗传特征。7型痉挛性截瘫患者已报告有包括视神经萎缩、眼肌麻痹或眼球震颤在内的各种眼科表现。我们报告一名15岁男性患者,其SPG7(NM_003119.3)基因存在一种新的杂合变异,即c.1224T>G:p.(Asp408Glu),该变异导致在神经症状出现之前就出现早发性孤立性视神经萎缩和婴儿型眼球震颤。因此,SPG7应被视为婴儿型眼球震颤伴视神经萎缩的一个病因。

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