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A SEPSECS mutation in a 23-year-old woman with microcephaly and progressive cerebellar ataxia.


ABSTRACT: Mutations in the SEPSECS gene are associated with pontocerebellar hypoplasia type 2D. Pontocerebellar hypoplasia (PCH) is a heterogeneous group of rare autosomal recessive neurodegenerative disorders, mainly affecting pons and cerebellum. Patients have severe motor and cognitive impairments and often die during infancy. Here, we report a 23-year-old woman with slowly progressive cerebellar ataxia and cognitive impairment, in whom a homozygous missense mutation in the SEPSECS gene (c.1321G>A; p.Gly441Arg) was identified with whole exome sequencing. Our findings underline that defects in selenoprotein synthesis can also result in milder cerebellar atrophy presenting at a later age.

SUBMITTER: van Dijk T 

PROVIDER: S-EPMC6133186 | biostudies-literature | 2018 Sep

REPOSITORIES: biostudies-literature

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A SEPSECS mutation in a 23-year-old woman with microcephaly and progressive cerebellar ataxia.

van Dijk Tessa T   Vermeij Jan-Dirk JD   van Koningsbruggen Silvana S   Lakeman Phillis P   Baas Frank F   Poll-The Bwee Tien BT  

Journal of inherited metabolic disease 20180220 5


Mutations in the SEPSECS gene are associated with pontocerebellar hypoplasia type 2D. Pontocerebellar hypoplasia (PCH) is a heterogeneous group of rare autosomal recessive neurodegenerative disorders, mainly affecting pons and cerebellum. Patients have severe motor and cognitive impairments and often die during infancy. Here, we report a 23-year-old woman with slowly progressive cerebellar ataxia and cognitive impairment, in whom a homozygous missense mutation in the SEPSECS gene (c.1321G>A; p.G  ...[more]

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