| Literature DB >> 22693042 |
Anas M Alazami1, Muneera J Alshammari, Mustafa A Salih, Fatema Alzahrani, Hadia Hijazi, Mohammed Z Seidahmed, Leen Abu Safieh, Mazhor Aldosary, Arif O Khan, Fowzan S Alkuraya.
Abstract
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.Entities:
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Year: 2012 PMID: 22693042 DOI: 10.1002/humu.22134
Source DB: PubMed Journal: Hum Mutat ISSN: 1059-7794 Impact factor: 4.878