[Two children with severe recurrent infections and the X-linked hyper-IgM syndrome]

Ned Tijdschr Geneeskd. 2003 May 24;147(21):1024-8.
[Article in Dutch]

Abstract

A boy suffered from severe recurrent intestinal infections from the age of 8 months onwards; investigation into an immune disorder ultimately resulted in the diagnosis of 'hyper-IgM syndrome'. He was treated successfully with bone marrow transplantation, using an HLA-matched donor. Another boy had severe recurrent respiratory tract infections from the age of 3 months onwards. At the age of 6.5 years, 'hyper-IgM syndrome' was diagnosed. No suitable donor was available. In addition, he developed sclerosing cholangitis and end-stage liver disease, making a combined bone marrow and liver transplantation too risky. He died at 10.5 years of age. X-linked hyper-IgM syndrome is a rare congenital immunodeficiency disorder, characterised by a defect in both humoral and cellular immune responses. Deficiency in the membrane glycoprotein CD40 ligand (expressed on activated T-cells) compromises T-cell interactions with antigen-presenting cells. In a child with severe recurrent infections, and with dysgammaglobulinaemia with a normal or increased IgM level, the diagnosis of 'X-linked hyper-IgM syndrome' should be considered.

Publication types

  • Case Reports

MeSH terms

  • Bone Marrow Transplantation
  • CD40 Ligand / genetics
  • CD40 Ligand / metabolism
  • Child
  • Child, Preschool
  • Chromosomes, Human, X*
  • Diagnosis, Differential
  • Fatal Outcome
  • Genetic Diseases, X-Linked / diagnosis
  • Genetic Diseases, X-Linked / genetics*
  • Genetic Diseases, X-Linked / immunology
  • Genetic Diseases, X-Linked / therapy
  • Humans
  • Hypergammaglobulinemia / diagnosis
  • Hypergammaglobulinemia / genetics*
  • Hypergammaglobulinemia / immunology
  • Hypergammaglobulinemia / therapy
  • Immunoglobulin M* / blood
  • Infant
  • Infections / diagnosis
  • Infections / genetics*
  • Infections / immunology
  • Infections / therapy
  • Male
  • Recurrence
  • Syndrome

Substances

  • Immunoglobulin M
  • CD40 Ligand