Sequence variation within the RPGR gene: evidence for a founder complex allele

Hum Mutat. 2000 Sep;16(3):273-4. doi: 10.1002/1098-1004(200009)16:3<273::AID-HUMU19>3.0.CO;2-W.

Abstract

In our study of sequence variation within the RPGR gene associated with X-linked retinitis pigmentosa, we and others have observed a high rate of new mutation within this gene, as all reported mutations are unique or uncommon. In this article we report the identification in a single family of a complex allele of 7 sequence variants in linkage disequilibrium, of which four result in amino-acid alterations (Arg425Lys, DGlu, Thr533Met and Gly566Glu). This complex allele was initially found in a family with XLRP. However, further study revealed an estimated prevalence of 4.3% (15/344 chromosomes) with this complex allele in the European population indicating the non-pathogenic nature of this allele and, along with previously reported polymorphisms, further supporting a high level of human protein diversity for RPGR. This common complex allele may have been established in the population as a founder effect. Complete gene sequencing identified a potential pathogenic sequence variant in the family described (IVS6+5G>A). This study emphasises the need to create a more complete picture of the allelic variation within a gene, suggests cautious interpretation of a phenotypic association with variant sequences, and highlights the potential problems associated with interpreting genetic studies for diagnostic purposes.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Alleles*
  • Amino Acid Substitution / genetics
  • Base Sequence
  • Carrier Proteins / genetics*
  • Eye Proteins*
  • Female
  • Founder Effect*
  • Genetic Variation / genetics*
  • Humans
  • Male
  • Pedigree
  • Retinitis Pigmentosa / genetics*

Substances

  • Carrier Proteins
  • Eye Proteins
  • RPGR protein, human