Family study of a Swiss patient uncovered a novel genetic basis for the S-s-U+(var) phenotype

Transfusion. 2014 Nov;54(11):2941-5. doi: 10.1111/trf.12681. Epub 2014 Apr 17.

Abstract

Background: The rare S-s- phenotype is typically found in persons of African origin. Three genetic bases underlying this phenotype have been identified so far: a large deletion including the GYPB gene, which encodes the S and s antigens, and two mutations affecting GYPB splicing (commonly called "P2" and "NY"). The discovery of the S-s- phenotype in a Swiss patient prompted this study.

Study design and methods: The GYPB genotype of the patient was analyzed with Beadchip technology and Sanger sequencing. GYPB haplotype analysis was also carried out in the patient's family. A functional splicing assay was developed to determine the impact of the identified mutation on GYPB splicing.

Results: Sanger sequencing of GYPB in the patient indicated that she was homozygous for a GYPB*s allele carrying a novel mutation in the splice donor site of Intron 5 (c.270+5G>A). Analysis of GYPB haplotypes in the patient's family revealed that she actually inherited this mutated GYPB*s allele from her mother of Swiss ancestry and a deleted GYPB allele from her father of Egyptian ancestry. Using a minigene-based splicing assay, we showed that GYPB mutation c.270+5G>A causes the skipping of Exon B5, as previously reported for the P2 mutation (c.270+5G>T). Consistently, the patient's red blood cells were found to be S-s-U+(var) .

Conclusion: A novel GYPB mutation (c.270+5G>A) accounting for the S-s-U+(var) phenotype was identified. In contrast with P2 and NY mutations, which also drive this rare phenotype, this novel GYPB mutation inactivates a GYPB*s allele and does not appear to be of African origin.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Alleles
  • Blood Group Antigens / genetics*
  • Cyclophilins / genetics*
  • Erythrocytes
  • Exons*
  • Family*
  • Female
  • Humans
  • Male
  • Point Mutation*
  • RNA Splice Sites*
  • Switzerland

Substances

  • Blood Group Antigens
  • RNA Splice Sites
  • cyclophilin B
  • Cyclophilins