A novel PRPF31 mutation in a large Chinese family with autosomal dominant retinitis pigmentosa and macular degeneration

PLoS One. 2013 Nov 11;8(11):e78274. doi: 10.1371/journal.pone.0078274. eCollection 2013.

Abstract

Purpose: This study was intended to identify the disease causing genes in a large Chinese family with autosomal dominant retinitis pigmentosa and macular degeneration.

Methods: A genome scan analysis was conducted in this family for disease gene preliminary mapping. Snapshot analysis of selected SNPs for two-point LOD score analysis for candidate gene filter. Candidate gene PRPF31 whole exons' sequencing was executed to identify mutations.

Results: A novel nonsense mutation caused by an insertion was found in PRPF31 gene. All the 19 RP patients in 1085 family are carrying this heterozygous nonsense mutation. The nonsense mutation is in PRPF31 gene exon9 at chr19:54629961-54629961, inserting nucleotide "A" that generates the coding protein frame shift from p.307 and early termination at p.322 in the snoRNA binding domain (NOP domain).

Conclusion: This report is the first to associate PRPF31 gene's nonsense mutation and adRP and JMD. Our findings revealed that PRPF31 can lead to different clinical phenotypes in the same family, resulting either in adRP or syndrome of adRP and JMD. We believe our identification of the novel "A" insertion mutation in exon9 at chr19:54629961-54629961 in PRPF31 can provide further genetic evidence for clinical test for adRP and JMD.

Publication types

  • Clinical Trial
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Aged
  • Asian People
  • China
  • Codon, Nonsense*
  • Eye Proteins / genetics*
  • Female
  • Genes, Dominant*
  • Genetic Diseases, Inborn / genetics*
  • Humans
  • Lod Score
  • Macular Degeneration / genetics*
  • Male
  • Middle Aged
  • Mutagenesis, Insertional*
  • Protein Structure, Tertiary
  • Retinitis Pigmentosa / genetics*

Substances

  • Codon, Nonsense
  • Eye Proteins
  • PRPF31 protein, human

Grants and funding

The authors acknowledge the following grants support: National natural science foundation of China (No.81070761 and 81241001 to FL; No.81025006 to ZY; No.81271048 to JY) and youth science & technology foundation of Sichuan province (to FL and to ZY). The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.