Large granular lymphocyte proliferation and revertant mosaicism: two rare events in a Wiskott-Aldrich syndrome patient

Haematologica. 2007 Mar;92(3):e43-5. doi: 10.3324/haematol.11222.

Abstract

We report on a 6 year old patient with an unusual clinical presentation of WAS and oligoclonal proliferation of TCR+ large granular lymphocytes (LGL). Flow cytometry demonstrated two distinct populations of lymphocytes with strongly decreased (WASP-) or normal expression levels of WASP (WASP+), respectively. Molecular analysis confirmed a splice site mutation in intron 2 of the WASP gene in the WASP- cells but not in WASP+ cells. LGL cells were WASP+, suggesting that two independent rare events, somatic revertant mosaicism and LGL expansion, have occurred in a child with WAS. Our report points to diagnostic difficulties in the presence of partial WASP reversions and LGL.

Publication types

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

MeSH terms

  • Cell Separation
  • Child
  • Diagnosis, Differential
  • Flow Cytometry
  • Haemophilus Infections / complications
  • Humans
  • Introns / genetics
  • Lymphoproliferative Disorders / etiology*
  • Lymphoproliferative Disorders / genetics
  • Lymphoproliferative Disorders / pathology
  • Male
  • Mosaicism*
  • Mutation
  • Purpura, Thrombocytopenic, Idiopathic / diagnosis
  • RNA Splice Sites / genetics*
  • Receptors, Antigen, T-Cell, gamma-delta / analysis
  • Recurrence
  • T-Lymphocyte Subsets / chemistry
  • T-Lymphocyte Subsets / pathology*
  • Wiskott-Aldrich Syndrome / blood
  • Wiskott-Aldrich Syndrome / complications*
  • Wiskott-Aldrich Syndrome / diagnosis
  • Wiskott-Aldrich Syndrome / genetics
  • Wiskott-Aldrich Syndrome Protein / analysis
  • Wiskott-Aldrich Syndrome Protein / deficiency
  • Wiskott-Aldrich Syndrome Protein / genetics

Substances

  • RNA Splice Sites
  • Receptors, Antigen, T-Cell, gamma-delta
  • WAS protein, human
  • Wiskott-Aldrich Syndrome Protein