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DOI 10.1111/nyas.14229

Медицинская библиотека · Annals of the New York Academy of Sciences · 2019

Dwarfism in Troyer syndrome: a family with SPG20 compound heterozygous mutations and a literature review.

Liang Hanting, Miao Hui, Yang Hongbo, Gong Fengying, Chen Shi, Wang Linjie, Zhu Huijuan, Pan Hui

Клинический случайСтатьяОбзорПеревод пока не подготовлен

Аннотация

Troyer syndrome is an autosomal recessive disease characterized by spastic paralysis, dysarthria, distal amyotrophy, and short stature. Recently, two siblings (an older brother and a younger sister) were admitted to our hospital for the chief complaints of "short stature and intellectual disability." Through whole exome sequencing of the sister, who is the proband, it was found that her SPG20 gene had compound heterozygous mutations: c.364_365delAT (p.Met122Valfs* 2) and c.892delA (p.Thr298Glnfs* 30). Target testing revealed that the brother had the same genotype as the sister, and the former mutation originated from the father, while the latter mutation originated from the mother. In summary, this is the first report of Troyer syndrome in a family caused by SPG20 compound heterozygous mutations. A novel SPG20 mutation was found, namely c.892delA (p.Thr298Glnfs* 30). In addition, we also summarize these Troyer syndrome patients' heights and their clinical characteristics, and provide a brief review of all known pathogenic mutations of SPG20.

Для цитирования: Liang Hanting, Miao Hui, Yang Hongbo, Gong Fengying, Chen Shi, Wang Linjie, Zhu Huijuan, Pan Hui. Dwarfism in Troyer syndrome: a family with SPG20 compound heterozygous mutations and a literature review.. Annals of the New York Academy of Sciences. 2019. DOI: 10.1111/nyas.14229.

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