Weak D caused by a founder deletion in the RHD gene

Transfusion. 2012 Nov;52(11):2348-55. doi: 10.1111/j.1537-2995.2012.03606.x. Epub 2012 Mar 15.

Abstract

Background: The RhD blood group system exemplifies a genotype-phenotype correlation by virtue of its highly polymorphic and immunogenic nature. Weak D phenotypes are generally thought to result from missense mutations leading to quantitative change of the D antigen in the red blood cell membrane or intracellularly.

Study design and methods: Different sets of polymerase chain reaction primers were designed to map and clone a deletion involving RHD Exon 10, which was found in approximately 3% of approximately 2000 RHD hemizygous subjects with D phenotype ambiguity. D antigen density was measured by flow cytometry. Transcript analysis was carried out by 3'-rapid amplification of complementary DNA ends. Haplotype analysis was performed by microsatellite genotyping.

Results: A 5405-bp deletion that removed nearly two-thirds of Intron 9 and almost all of Exon 10 of the RHD gene was characterized. It is predicted to result in the replacement of the last eight amino acids of the wild-type RhD protein by another four amino acids. The mean RhD antigen density from two deletion carriers was determined to be only 30. A consensus haplotype could be deduced from the deletion carriers based on the microsatellite genotyping data.

Conclusion: The currently reported deletion was derived from a common founder. This deletion appears to represent not only the first large deletion associated with weak D but also the weakest of weak D alleles so far reported. This highly unusual genotype-phenotype relationship may be attributable to the additive effect of three distinct mechanisms that affect mRNA formation, mRNA stability, and RhD/ankyrin-R interaction, respectively.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Erythrocyte Membrane / physiology
  • Exons / genetics
  • Flow Cytometry
  • Founder Effect*
  • Gene Deletion*
  • Genetic Association Studies / methods*
  • Haplotypes
  • Humans
  • Immunophenotyping
  • Microsatellite Repeats / genetics
  • Mutation, Missense / genetics
  • Nucleic Acid Amplification Techniques
  • Polymerase Chain Reaction
  • RNA Stability / genetics
  • Rh-Hr Blood-Group System / blood*
  • Rh-Hr Blood-Group System / genetics*

Substances

  • Rh-Hr Blood-Group System
  • Rho(D) antigen