Reduced memory B-cell populations in boys with B-cell dysfunction after bone marrow transplantation for X-linked severe combined immunodeficiency

Br J Haematol. 2001 Mar;112(4):1004-11. doi: 10.1046/j.1365-2141.2001.02639.x.

Abstract

X-linked severe combined immunodeficiency (XSCID) is a lethal disease resulting in death in infancy. In many instances, haploidentical bone marrow transplantation (BMT) offers reconstitution of T-cell immunity alone, with residual hypogammaglobulinaemia. The exact nature of B-cell dysfunction in these patients is unclear, although differentiation arrest of the B cells is a potential explanation. To ascertain the differentiation status of peripheral blood B lymphocytes from XSCID patients after BMT, the surface expression of CD19, CD10, CD34, CD5, serum immunoglogulin (sIg)M, sIgD, sIgG and CD27 on these B cells was investigated using three-colour flow cytometry. CD27 is a marker of memory B cells. Populations of CD19+IgM-D- B cells, CD19+IgM-only, CD19+IgG+CD27+ and CD19+IgM+ CD27+ B cells were found to be diminished in the XSCID patients after BMT with persistent hypogammaglobulinaemia, compared with both post-BMT patients with B-cell function and age-matched normal controls. This indicated the lack of CD19+IgM-D- B cells, which represent Ig isotype-switched B cells, as well as CD19+IgM-only and CD19+IgG+CD27+ or CD19+IgM+CD27+ memory B-cell populations. Interaction between CD27 and its ligand CD70 has been shown to induce IgG and IgM production by CD27+ B cells. Therefore, the lack of CD27/70 interaction is a probable explanation for the hypogammaglobulinaemia in these patients after BMT.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Agammaglobulinemia / immunology
  • Antigens, CD19 / analysis
  • Antigens, CD34 / analysis
  • B-Lymphocytes / immunology*
  • Bone Marrow Transplantation*
  • CD5 Antigens / analysis
  • Case-Control Studies
  • Flow Cytometry / methods
  • Follow-Up Studies
  • Humans
  • Immunoglobulin D / blood
  • Immunoglobulin G / blood
  • Immunoglobulin M / blood
  • Immunologic Memory*
  • Immunophenotyping
  • Infant
  • Male
  • Neprilysin / analysis
  • Severe Combined Immunodeficiency / immunology*
  • Severe Combined Immunodeficiency / therapy*
  • Statistics, Nonparametric
  • Tumor Necrosis Factor Receptor Superfamily, Member 7 / analysis
  • X Chromosome*

Substances

  • Antigens, CD19
  • Antigens, CD34
  • CD5 Antigens
  • Immunoglobulin D
  • Immunoglobulin G
  • Immunoglobulin M
  • Tumor Necrosis Factor Receptor Superfamily, Member 7
  • Neprilysin