Frequent mutations in SH2D1A (XLP) in males presenting with high-grade mature B-cell neoplasms

Pediatr Blood Cancer. 2013 Sep;60(9):E85-7. doi: 10.1002/pbc.24525. Epub 2013 Apr 15.

Abstract

X-linked lymphoproliferative syndrome (XLP) is caused by mutations in SH2D1A, and is associated with overwhelming infectious mononucleosis, aplastic anemia, hypogammaglobulinemia, and B-cell lymphomas. However, the frequency of SH2D1A mutations in males who present with B NHL is unknown. Five cases of XLP were diagnosed among 158 males presenting with B NHL (approximately 3.2%). Four of the patients had two episodes of B NHL and one had a single episode of B NHL followed by aggressive infectious mononucleosis. Prospective screening for XLP in males with B-cell lymphoma at the time of initial diagnosis should be considered.

Keywords: B cells; EBV; XLP; lymphoma.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Follow-Up Studies
  • Humans
  • Intracellular Signaling Peptides and Proteins / genetics*
  • Lymphoma, Large B-Cell, Diffuse
  • Lymphoproliferative Disorders / diagnosis
  • Lymphoproliferative Disorders / genetics*
  • Lymphoproliferative Disorders / pathology
  • Lymphoproliferative Disorders / therapy
  • Male
  • Mutation*
  • Registries*
  • Retrospective Studies
  • Signaling Lymphocytic Activation Molecule Associated Protein

Substances

  • Intracellular Signaling Peptides and Proteins
  • SH2D1A protein, human
  • Signaling Lymphocytic Activation Molecule Associated Protein