Molecular genetics of the Usher syndrome in Lebanon: identification of 11 novel protein truncating mutations by whole exome sequencing

PLoS One. 2014 Sep 11;9(9):e107326. doi: 10.1371/journal.pone.0107326. eCollection 2014.

Abstract

Background: Usher syndrome (USH) is a genetically heterogeneous condition with ten disease-causing genes. The spectrum of genes and mutations causing USH in the Lebanese and Middle Eastern populations has not been described. Consequently, diagnostic approaches designed to screen for previously reported mutations were unlikely to identify the mutations in 11 unrelated families, eight of Lebanese and three of Middle Eastern origins. In addition, six of the ten USH genes consist of more than 20 exons, each, which made mutational analysis by Sanger sequencing of PCR-amplified exons from genomic DNA tedious and costly. The study was aimed at the identification of USH causing genes and mutations in 11 unrelated families with USH type I or II.

Methods: Whole exome sequencing followed by expanded familial validation by Sanger sequencing.

Results: We identified disease-causing mutations in all the analyzed patients in four USH genes, MYO7A, USH2A, GPR98 and CDH23. Eleven of the mutations were novel and protein truncating, including a complex rearrangement in GPR98.

Conclusion: Our data highlight the genetic diversity of Usher syndrome in the Lebanese population and the time and cost-effectiveness of whole exome sequencing approach for mutation analysis of genetically heterogeneous conditions caused by large genes.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Amino Acid Sequence
  • Cadherin Related Proteins
  • Cadherins / genetics
  • Codon, Nonsense*
  • DNA Mutational Analysis
  • Exome*
  • Extracellular Matrix Proteins / genetics
  • Genetic Association Studies
  • Genetic Linkage
  • Humans
  • Lebanon
  • Molecular Sequence Data
  • Myosin VIIa
  • Myosins / genetics
  • Receptors, G-Protein-Coupled / genetics
  • Sequence Analysis, RNA
  • Usher Syndromes / genetics*
  • Usher Syndromes / metabolism

Substances

  • ADGRV1 protein, human
  • CDH23 protein, human
  • Cadherin Related Proteins
  • Cadherins
  • Codon, Nonsense
  • Extracellular Matrix Proteins
  • MYO7A protein, human
  • Myosin VIIa
  • Receptors, G-Protein-Coupled
  • USH2A protein, human
  • Myosins