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Novel Mutations of the ALMS1 Gene in Patients with Alström Syndrome.
Wang, Chunmei; Luo, Xiaona; Wang, Yilin; Liu, Zhao; Wu, Shengnan; Wang, Simei; Lan, Xiaoping; Xu, Quanmei; Xu, Wuhen; Yuan, Fang; Wang, Anqi; Zeng, Fanyi; Jia, Jia; Chen, Yucai.
Afiliação
  • Wang C; Department of Neurology, Children's Hospital of Shanghai, Shanghai JiaoTong University, China.
  • Luo X; Department of Neurology, Children's Hospital of Shanghai, Shanghai JiaoTong University, China.
  • Wang Y; Department of Neurology, Children's Hospital of Shanghai, Shanghai JiaoTong University, China.
  • Liu Z; Division of Pediatric Neurology, Department of Pediatrics, University of Illinois and Children's Hospital of Illinois, USA.
  • Wu S; Department of Neurology, Children's Hospital of Shanghai, Shanghai JiaoTong University, China.
  • Wang S; Department of Neurology, Children's Hospital of Shanghai, Shanghai JiaoTong University, China.
  • Lan X; Department of Neurology, Children's Hospital of Shanghai, Shanghai JiaoTong University, China.
  • Xu Q; Department of Neurology, Children's Hospital of Shanghai, Shanghai JiaoTong University, China.
  • Xu W; Department of Neurology, Children's Hospital of Shanghai, Shanghai JiaoTong University, China.
  • Yuan F; Department of Neurology, Children's Hospital of Shanghai, Shanghai JiaoTong University, China.
  • Wang A; Department of Neurology, Children's Hospital of Shanghai, Shanghai JiaoTong University, China.
  • Zeng F; NHC Key Laboratory of Medical Embryogenesis and Developmental Molecular Biology & Shanghai Key Laboratory of Embryo and Reproduction Engineering, China.
  • Jia J; Fuxiang Gene Engineering Research Institute, China.
  • Chen Y; Department of Neurology, Children's Hospital of Shanghai, Shanghai JiaoTong University, China.
Intern Med ; 60(23): 3721-3728, 2021 Dec 01.
Article em En | MEDLINE | ID: mdl-34148947
Objective Alström syndrome is an autosomal recessive genetic disease caused by a mutation in the ALMS1 gene. Alström syndrome is clinically characterized by multisystem involvement, including sensorineural deafness, cone-rod dystrophy, nystagmus, obesity, insulin resistance, type 2 diabetes and hypogonadism. The diagnosis is thus challenging for patients without this characteristic set of clinical symptoms. We explored the effectiveness of whole-exome sequencing in the diagnosis of Alström syndrome. Methods A girl with symptoms of Alström syndrome was tested and diagnosed with the disease by whole-exome sequencing. Results Whole-exome sequencing revealed two novel variants, c.6160_6161insAT: p.Lys2054Asnfs*21 (exon 8) and c.10823_10824 delAG:p.Glu 3608Alafs*9 (exon16) in the ALMS1 gene, leading to premature termination codons and the domain of ALMS1 protein. Blood sample testing of her asymptomatic parents revealed them to be heterozygous carriers of the same mutations. Assembly showed that the mutations on both alleles were located in conserved sequences. A review of the ALMS1 gene nonsense mutation status was performed. Conclusion We herein report two novel variants of the ALMS1 gene discovered in a Chinese Alström syndrome patient that expand the mutational spectrum of ALMS1 and provided new insight into the molecular mechanism underlying Alström syndrome. Our findings add to the current knowledge concerning the diagnosis and treatment of Alström syndrome.
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Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Assunto principal: Proteínas de Ciclo Celular / Diabetes Mellitus Tipo 2 / Síndrome de Alstrom Tipo de estudo: Diagnostic_studies Limite: Female / Humans Idioma: En Ano de publicação: 2021 Tipo de documento: Article

Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Assunto principal: Proteínas de Ciclo Celular / Diabetes Mellitus Tipo 2 / Síndrome de Alstrom Tipo de estudo: Diagnostic_studies Limite: Female / Humans Idioma: En Ano de publicação: 2021 Tipo de documento: Article