Immunodeficiency and infections in ataxia-telangiectasia

J Pediatr. 2004 Apr;144(4):505-11. doi: 10.1016/j.jpeds.2003.12.046.

Abstract

Objective: To characterize the immunodeficiency in ataxia-telangiectasia (A-T) and to determine whether the immunodeficiency is progressive and associated with increased susceptibility to infections.

Study design: Records of 100 consecutive patients with A-T from the Johns Hopkins Ataxia-Telangiectasia Clinical Center (ATCC) were reviewed.

Results: Immunoglobulin (Ig) deficiencies are common, affecting IgG4 in 65% of patients, IgA in 63%, IgG2 in 48%, IgE in 23%, and IgG in 18%. Lymphopenia affected 71% of patients, with reduced B-lymphocyte number in 75%, CD4 T lymphocytes in 69%, and CD8 T lymphocytes in 51%. There was no trend for increased frequency or severity of immune abnormalities with age. Recurrent upper and lower respiratory tract infections were frequent: otitis media in 46% of patients, sinusitis in 27%, bronchitis in 19%, and pneumonia in 15%. Sepsis occurred in 5 patients, in 2 patients concurrent with cancer chemotherapy. Warts affected 17% of patients, herpes simplex 8%, molluscum contagiosum 5%, candidal esophagitis 3%, and herpes zoster 2%. Uncomplicated varicella infection occurred in 44% of patients; 2 patients had more than one clinical episode. No patient had Pneumocystis jerovici pneumonia or a complication of live viral vaccine.

Conclusions: In spite of the high prevalence of laboratory immunologic abnormalities, systemic bacterial, severe viral, and opportunistic infections are uncommon in A-T. Cross-sectional analysis suggests that the immune defect is rarely progressive.

Publication types

  • Research Support, Non-U.S. Gov't
  • Research Support, U.S. Gov't, P.H.S.

MeSH terms

  • Adolescent
  • Adult
  • Age Factors
  • Ataxia Telangiectasia / complications*
  • Ataxia Telangiectasia / drug therapy
  • Ataxia Telangiectasia / immunology*
  • Ataxia Telangiectasia / mortality
  • Chickenpox / complications
  • Chickenpox / immunology
  • Child
  • Child, Preschool
  • Esophagitis / complications
  • Esophagitis / immunology
  • Esophagitis / microbiology
  • Female
  • Herpes Zoster / complications
  • Herpes Zoster / immunology
  • Humans
  • Immunoglobulins / blood
  • Immunoglobulins / deficiency*
  • Immunoglobulins, Intravenous / therapeutic use
  • Infant
  • Killer Cells, Natural / metabolism
  • Leukopenia / immunology
  • Lymphocyte Count
  • Lymphopenia / immunology
  • Male
  • Otitis Media / complications
  • Otitis Media / immunology
  • Respiratory Tract Infections / complications
  • Respiratory Tract Infections / immunology*
  • Sepsis / complications
  • Sepsis / immunology
  • Skin Diseases, Viral / complications
  • Skin Diseases, Viral / immunology

Substances

  • Immunoglobulins
  • Immunoglobulins, Intravenous