[Agammaglobulinemia with the absence of circulating B-lymphocytes. 9 cases]

Presse Med. 1998 Mar 28;27(12):562-6.
[Article in French]

Abstract

Objectives: Agammaglobulinemia with absence of circulating B lymphocytes is a rare genetically transmitted immunodeficiency that appears in early childhood and affect mainly boys. The clinical manifestations of the disease are rather heterogeneous.

Patients and methods: Nine patients (7 boys and 2 girls) were diagnosed as suffering from agammaglobulinemia with absence of circulating B lymphocytes, over a period of 6 years. Quantitation of immunoglobulins and search for circulating B lymphocytes were respectively performed by the Mancini method and immunofluorescence using T specific (anti-CD3, anti-CD4 and anti-CD8) and B (anti-CD19) monoclonal antibody.

Results: The disease started to manifest clinically at the mean age of 8.7 months (4-16 months). The mean age at diagnosis is 4 years (1-11 years). The clinical manifestations were essentially recurrent infections of the lung and the gastrointestinal tract. However, bacterial meningitidis was observed in 3 patients. Severe complications such as an echovirus 27 meningoencephalitis and a chronic active hepatitis (1 patient) and a pericarditis (1 patient) were observed. All of our patients lacked circulating B lymphocytes and had low or null immunoglobulin levels. Five patients were treated by intravenous immunoglobulin (Ig) and 3 were treated by intramuscular immunoglobulin with a residual IgG level respectively of 5.5 g/l and 3.3 g/l.

Conclusion: Recurrent infections are the principal manifestation of the agammaglobulinemia, early Ig treatment is the only therapy allowing improved.

MeSH terms

  • Adolescent
  • Agammaglobulinemia / genetics
  • Agammaglobulinemia / immunology*
  • Agammaglobulinemia / therapy
  • Age Factors
  • Antibodies, Monoclonal
  • Antigens, CD19 / analysis
  • B-Lymphocytes / immunology*
  • CD3 Complex / analysis
  • CD4 Antigens / analysis
  • CD8 Antigens / analysis
  • Child
  • Echovirus Infections
  • Female
  • Humans
  • Immunization, Passive
  • Immunoglobulin A / blood
  • Immunoglobulin G / blood
  • Immunoglobulin M / blood
  • Immunoglobulins, Intravenous / therapeutic use
  • Infant
  • Injections, Intramuscular
  • Lymphopenia / genetics
  • Lymphopenia / immunology*
  • Lymphopenia / therapy
  • Male
  • Meningitis, Bacterial / etiology
  • Meningoencephalitis / virology
  • Opportunistic Infections / etiology
  • Retrospective Studies

Substances

  • Antibodies, Monoclonal
  • Antigens, CD19
  • CD3 Complex
  • CD4 Antigens
  • CD8 Antigens
  • Immunoglobulin A
  • Immunoglobulin G
  • Immunoglobulin M
  • Immunoglobulins, Intravenous