Novel splice receptor-site mutation of RPGR in a Chinese family with X-linked retinitis pigmentosa

Medicine (Baltimore). 2018 Oct;97(41):e12779. doi: 10.1097/MD.0000000000012779.

Abstract

Rationale: Retinitis pigmentosa (RP) is a group of clinically and genetically heterogeneous diseases; X-linked retinitis pigmentosa (XLRP) is the most serious type. Mutations in RP GTPase regulator (RPGR) account for over 70% of patients with XLRP.

Patient concerns: We report a Chinese family with RP, 5 males presented with night blindness and decreased vision, and 8 females showed different severities of myopia.

Diagnoses: Targeted exome capture sequencing was performed in 2 affected males, which revealed a novel variant (NM_000328.2, c.470-1G>A) in the RPGR gene. The mis-splicing causes a substitution of the 157th amino acid from glutamic acid to glycine and finally the 165th codon is changed to stop codon, possibly resulting in a truncated protein and/or a nonsense-mediated mRNA decay. The mutation cosegregated with the disease phenotype in the family.

Interventions: Medication and cataract surgery.

Outcomes: The phenotype of affected males is more serious than that of the carrier females, and the effect of clinical treatment is not very well.

Lessons: Next-generation sequencing is a suitable method for early detection of pathogenic mutations in RP, which would be helpful for prenatal diagnosis of the disease.

MeSH terms

  • Adolescent
  • Adult
  • Asian People / genetics
  • Child
  • Child, Preschool
  • China
  • Codon
  • Exons
  • Eye Proteins / genetics*
  • Female
  • Genetic Diseases, X-Linked / genetics*
  • Humans
  • Male
  • Mutation / genetics*
  • Pedigree
  • Phenotype
  • RNA Splice Sites / genetics*
  • Retinitis Pigmentosa / genetics*
  • Young Adult

Substances

  • Codon
  • Eye Proteins
  • RNA Splice Sites
  • RPGR protein, human