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Case report: A novel WASHC5 variant altering mRNA splicing causes spastic paraplegia in a patient.
Gao, Shan-Yu; Liu, Yu-Xing; Dong, Yi; Fan, Liang-Liang; Ding, Qi; Liu, Lv.
Afiliación
  • Gao SY; Department of Neurology, Changshu No. 2 People's Hospital, Changshu, China.
  • Liu YX; Department of Cell Biology, School of Life Science, Central South University, Changsha, China.
  • Dong Y; Department of Pulmonary and Critical Care Medicine, Research Unit of Respiratory Disease, Hunan Diagnosis and Treatment Center of Respiratory Disease, The Second Xiangya Hospital, Central South University, Changsha, China.
  • Fan LL; Department of Cell Biology, School of Life Science, Central South University, Changsha, China.
  • Ding Q; Department of Nephrology, Xiangya Hospital Central South University, Changsha, China.
  • Liu L; Department of Cell Biology, School of Life Science, Central South University, Changsha, China.
Front Genet ; 14: 1205052, 2023.
Article en En | MEDLINE | ID: mdl-38028608
Background: Hereditary spastic paraplegia (HSP) is a progressive upper-motor neurodegenerative disease. Mutations in the WASHC5 gene are associated with autosomal dominant HSP, spastic paraplegia 8 (SPG8). However, due to the small number of reported cases, the exact mechanism remains unclear. Method: We report a Chinese family with HSP. The proband was referred to our hospital due to restless leg syndrome and insomnia. The preliminary clinical diagnosis of the proband was spastic paraplegia. Whole-exome sequencing (WES) and RNA splicing analysis were conducted to evaluate the genetic cause of the disease in this family. Results: A novel splice-altering variant (c.712-2A>G) in the WASHC5 gene was detected and further verified by RNA splicing analysis and Sanger sequencing. Real-time qPCR analysis showed that the expression of genes involved in the Wiskott-Aldrich syndrome protein and SCAR homolog (WASH) complex and endosomal and lysosomal systems was altered due to this variant. Conclusion: A novel heterozygous splice-altering variant (c.712-2A>G) in the WASHC5 gene was detected in a Chinese family with HSP. Our study provided data for genetic counseling to this family and offered evidence that this splicing variant in the WASHC5 gene is significant in causing HSP.
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Idioma: En Revista: Front Genet Año: 2023 Tipo del documento: Article País de afiliación: China Pais de publicación: Suiza

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Idioma: En Revista: Front Genet Año: 2023 Tipo del documento: Article País de afiliación: China Pais de publicación: Suiza