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Molecular characterization of Joubert syndrome in Saudi Arabia.
Alazami, Anas M; Alshammari, Muneera J; Salih, Mustafa A; Alzahrani, Fatema; Hijazi, Hadia; Seidahmed, Mohammed Z; Abu Safieh, Leen; Aldosary, Mazhor; Khan, Arif O; Alkuraya, Fowzan S.
Afiliação
  • Alazami AM; Department of Genetics, King Faisal Specialist Hospital and Research Center, Riyadh, Saudi Arabia.
Hum Mutat ; 33(10): 1423-8, 2012 Oct.
Article em En | MEDLINE | ID: mdl-22693042
Joubert syndrome (JS) is a ciliopathy that is defined primarily by typical cerebellar structural and ocular motility defects. The genetic heterogeneity of this condition is significant with 16 genes identified to date. We have used a combination of autozygome-guided candidate gene mutation analysis and exome sequencing to identify the causative mutation in a series of 12 families. The autozygome approach identified mutations in RPGRIP1L, AHI1, TMEM237, and CEP290, while exome sequencing revealed families with truncating mutations in TCTN1 and C5ORF42. Our study, the largest comprehensive molecular series on JS, provides independent confirmation of the recently reported TCTN1, TMEM237, and C5ORF42 as bona fide JS disease genes, and expands the allelic heterogeneity of this disease.
Assuntos

Texto completo: 1 Bases de dados: MEDLINE Assunto principal: Doenças Cerebelares / Anormalidades do Olho / Doenças Renais Císticas Limite: Child / Child, preschool / Female / Humans / Infant País/Região como assunto: Asia Idioma: En Revista: Hum Mutat Assunto da revista: GENETICA MEDICA Ano de publicação: 2012 Tipo de documento: Article País de afiliação: Arábia Saudita

Texto completo: 1 Bases de dados: MEDLINE Assunto principal: Doenças Cerebelares / Anormalidades do Olho / Doenças Renais Císticas Limite: Child / Child, preschool / Female / Humans / Infant País/Região como assunto: Asia Idioma: En Revista: Hum Mutat Assunto da revista: GENETICA MEDICA Ano de publicação: 2012 Tipo de documento: Article País de afiliação: Arábia Saudita