Carrier detection in the Wiskott Aldrich syndrome

Blood. 1988 Nov;72(5):1735-9.

Abstract

The Wiskott-Aldrich syndrome (WAS) is an X-linked recessive disease characterized by immunodeficiency and severe thrombocytopenia in affected males, but no demonstrable clinical abnormalities in carrier females. Through analysis of the methylation patterns of X-linked genes that display restriction fragment length polymorphisms (RFLPs), we studied the pattern of X-chromosome inactivation in various cell populations from female relatives of patients with WAS. The peripheral blood T cells, granulocytes, and B cells of eight obligate WAS carriers were found to display specific patterns of X-chromosome inactivation clearly different from these of normal controls. Thus, carriers of WAS could be accurately identified using this analysis.

Publication types

  • Research Support, U.S. Gov't, P.H.S.

MeSH terms

  • B-Lymphocytes / physiology
  • Blotting, Southern
  • Dosage Compensation, Genetic*
  • Genetic Carrier Screening / methods
  • Humans
  • Hypoxanthine Phosphoribosyltransferase / genetics
  • Phosphoglycerate Kinase / genetics
  • Wiskott-Aldrich Syndrome / diagnosis*
  • Wiskott-Aldrich Syndrome / genetics

Substances

  • Hypoxanthine Phosphoribosyltransferase
  • Phosphoglycerate Kinase