Literature DB >> 30258122

Variant in SCYL1 gene causes aberrant splicing in a family with cerebellar ataxia, recurrent episodes of liver failure, and growth retardation.

Adi Shohet1, Lior Cohen2,3, Danielle Haguel3,4, Yael Mozer3,5, Noam Shomron3,4, Shay Tzur6, Lily Bazak7, Lina Basel Salmon2,3,7,8, Irit Krause9,10.   

Abstract

Herein, we describe two members of one family who presented with recurrent episodes of hepatic failure, cerebellar ataxia, peripheral neuropathy, and short stature. Liver transplantation was considered. Whole-exome sequencing (Trio) revealed a synonymous variant in exon 4 of SCYL1:c.459C>T p. (Gly153Gly), which did not appear to affect the protein sequence. Computational prediction analysis suggested that this modification could alter the SCYL1 mRNA splicing processing to create a premature termination codon. The SCYL1 mRNAs in our patient's lymphocytes were analyzed and aberrant splicing was found. Molecular analysis of family members identified the parents as heterozygous recessive carriers and the proband as well as an affected aunt as homozygous. Evidently, harmless synonymous variants in the SCYL1 gene can damage gene splicing and hence the expression. We confirmed that the pathogenicity of this variant in the SCYL1 gene was associated with spinocerebellar ataxia, autosomal recessive 21 (SCAR21). Other reported cases (accept one) of liver failure found in the SCYL1 variants resolved during childhood, therefore orthotropic liver transplantation was no longer appropriate.

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Year:  2018        PMID: 30258122      PMCID: PMC6336772          DOI: 10.1038/s41431-018-0268-2

Source DB:  PubMed          Journal:  Eur J Hum Genet        ISSN: 1018-4813            Impact factor:   4.246


  13 in total

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Journal:  EMBO Rep       Date:  2007-06-15       Impact factor: 8.807

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Authors:  Jonathon L Burman; Jason N R Hamlin; Peter S McPherson
Journal:  PLoS One       Date:  2010-03-04       Impact factor: 3.240

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Journal:  J Biol Chem       Date:  2008-06-13       Impact factor: 5.157

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Journal:  Nature       Date:  2016-08-18       Impact factor: 49.962

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Journal:  Genet Med       Date:  2018-02-08       Impact factor: 8.822

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  9 in total

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Review 3.  The Classification of Autosomal Recessive Cerebellar Ataxias: a Consensus Statement from the Society for Research on the Cerebellum and Ataxias Task Force.

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Journal:  Cerebellum       Date:  2019-12       Impact factor: 3.847

4.  SCYL1 arginine methylation by PRMT1 is essential for neurite outgrowth via Golgi morphogenesis.

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6.  Whole exome sequencing in Alopecia Areata identifies rare variants in KRT82.

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Review 7.  Lipid Dyshomeostasis and Inherited Cerebellar Ataxia.

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Journal:  Mol Neurobiol       Date:  2022-04-14       Impact factor: 5.682

8.  mTORC1 controls Golgi architecture and vesicle secretion by phosphorylation of SCYL1.

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Journal:  Nat Commun       Date:  2022-08-10       Impact factor: 17.694

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