Literature DB >> 3079686

Hypogammaglobulinemia with hyper-IgM, severe T-cell defect, and abnormal recirculation of OKT4 lymphocytes in a girl with chronic lymphadenopathy.

M Fiorilli, G Russo, R Paganelli, C Papetti, M Carbonari, M Crescenzi, M Calvani, I Quinti, F Aiuti.   

Abstract

We describe here one 8-year-old girl with an unusual form of immunodeficiency, characterized by hypogammaglobulinemia with hyper-IgM, severe T-cell defect, and chronic lymphadenopathy. Patient's B cells failed to produce IgG or IgA in vitro following stimulation with either pokeweed mitogen or Epstein-Barr virus, suggesting an intrinsic B-cell defect. Abnormal T-cell function was demonstrated by impaired in vivo delayed type hypersensitivity, reduction of mitogen-induced proliferation and interleukin 2 production, reduction of interferon-gamma production, and marked decrease of circulating OKT4+ cells. The latter cells were found in normal proportion in the patient's lymph node tissue. This finding suggests that the decrease of OKT4+ cells in peripheral blood was due to the abnormal recirculation of these cells. The identity of this syndrome with the infantile form of the acquired immunodeficiency syndrome was apparently ruled out by the failure to demonstrate HTLV-III-related sequences in patient's lymphocytes or virus-specific serum antibodies.

Entities:  

Mesh:

Substances:

Year:  1986        PMID: 3079686     DOI: 10.1016/0090-1229(86)90143-1

Source DB:  PubMed          Journal:  Clin Immunol Immunopathol        ISSN: 0090-1229


  4 in total

Review 1.  The hyper-IgM (HIM) syndrome.

Authors:  N Ramesh; M Seki; L D Notarangelo; R S Geha
Journal:  Springer Semin Immunopathol       Date:  1998

2.  Prevalence of gallstones in patients with primary immunoglobulin deficiency. Evidence for lack of association.

Authors:  A De Santis; R Paganelli; R Stefanoni; A Di Sabatino; M Angelico
Journal:  Dig Dis Sci       Date:  1990-06       Impact factor: 3.199

3.  An imbalance of naive and memory/effector subsets and altered expression of CD38 on T lymphocytes in two girls with hyper-IgM syndrome.

Authors:  B T Costa-Carvalho; M A Viana; M K C Brunialti; E G Kallas; R Salomao
Journal:  Clin Exp Immunol       Date:  2004-05       Impact factor: 4.330

4.  Evidence that defective gamma interferon production in patients with primary immunodeficiencies is due to intrinsic incompetence of lymphocytes.

Authors:  R Paganelli; M R Capobianchi; B Ensoli; G P D'Offizi; J Facchini; F Dianzani; F Aiuti
Journal:  Clin Exp Immunol       Date:  1988-04       Impact factor: 4.330

  4 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.