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Heterozygous RHO p.R135W missense mutation in a large Han-Chinese family with retinitis pigmentosa and different refractive errors.
Wu, Yuan; Guo, Yi; Yi, Junhui; Xu, Hongbo; Yuan, Lamei; Yang, Zhijian; Deng, Hao.
Afiliación
  • Wu Y; Center for Experimental Medicine and Department of Neurology, The Third Xiangya Hospital, Central South University, Changsha, China.
  • Guo Y; Department of Clinical Laboratory, The Third Xiangya Hospital, Central South University, Changsha, China.
  • Yi J; Center for Experimental Medicine and Department of Neurology, The Third Xiangya Hospital, Central South University, Changsha, China.
  • Xu H; Department of Medical Information, School of Life Sciences, Central South University, Changsha, China.
  • Yuan L; Department of Ophthalmology, The Third Xiangya Hospital, Central South University, Changsha, China.
  • Yang Z; Center for Experimental Medicine and Department of Neurology, The Third Xiangya Hospital, Central South University, Changsha, China.
  • Deng H; Center for Experimental Medicine and Department of Neurology, The Third Xiangya Hospital, Central South University, Changsha, China.
Biosci Rep ; 39(7)2019 07 31.
Article en En | MEDLINE | ID: mdl-31239368
Retinitis pigmentosa (RP), the most common type of inherited retinal degeneration causing blindness, initially manifests as severely impaired rod function followed by deteriorating cone function. Mutations in the rhodopsin gene (RHO) are the most common cause of autosomal dominant RP (adRP). The present study aims to identify the disease-causing mutation in a numerous, four-generation Han-Chinese family with adRP detected by whole exome sequencing and Sanger sequencing. Afflicted family members present classic adRP along with heterogeneous clinical phenotypes including differing refractive errors, cataracts, astigmatism and epiretinal membranes. A missense mutation, c.403C>T (p.R135W), in the RHO gene was identified in nine subjects and it co-segregated with family members. The mutation is predicted to be disease-causing and results in rhodopsin protein abnormalities. The present study extends the genotype-phenotype relationship between RHO gene mutations and adRP clinical findings. The results have implications for familial genetic counseling, clinical management and developing RP target gene therapy strategies.
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Rodopsina / Retinitis Pigmentosa / Predisposición Genética a la Enfermedad Tipo de estudio: Prognostic_studies Límite: Adult / Female / Humans / Male / Middle aged Idioma: En Revista: Biosci Rep Año: 2019 Tipo del documento: Article País de afiliación: China Pais de publicación: Reino Unido

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Rodopsina / Retinitis Pigmentosa / Predisposición Genética a la Enfermedad Tipo de estudio: Prognostic_studies Límite: Adult / Female / Humans / Male / Middle aged Idioma: En Revista: Biosci Rep Año: 2019 Tipo del documento: Article País de afiliación: China Pais de publicación: Reino Unido