Literature DB >> 9462277

Common variable immunodeficiency with CD4+ T lymphocytopenia and overproduction of soluble IL-2 receptor associated with Turner's syndrome and dorsal kyphoscoliosis.

R A al-Attas1, A H Rahi.   

Abstract

An unusual combination of common variable immunodeficiency (CVID) and Turner's syndrome in a Saudi woman aged 20 years is presented. In addition to panhypogammaglobulinaemia, the patient had CD4+ T lymphocytopenia; however, there was evidence of in vivo activation of T cells and overproduction of soluble interleukin 2 receptor in culture supernate. Mantoux test was positive, but lymphoblastic response to non-specific mitogen was impaired. Immunogenetically the patient was HLA-DR3 positive and karyotypically she was a mosaic (45XO/46XX) with ring X chromosome (46Xr(X)). The presence of severe kyphoscoliosis was possibly related to ring X chromosome. This case highlights the grave consequences of the delayed diagnosis of immunodeficiency and emphasises the heterogeneous nature of CVID.

Entities:  

Mesh:

Substances:

Year:  1997        PMID: 9462277      PMCID: PMC500276          DOI: 10.1136/jcp.50.10.876

Source DB:  PubMed          Journal:  J Clin Pathol        ISSN: 0021-9746            Impact factor:   3.411


  14 in total

1.  Plasma and tissue interleukin-2 receptor levels in inflammatory bowel disease.

Authors:  Y R Mahida; A Gallagher; L Kurlak; C J Hawkey
Journal:  Clin Exp Immunol       Date:  1990-10       Impact factor: 4.330

2.  Common variable immunodeficiency in association with Turner's syndrome.

Authors:  S C Robson; P C Potter
Journal:  J Clin Lab Immunol       Date:  1990-07

3.  Immunoglobulins and the X-chromosome.

Authors:  C B Wood; W Martin; M Adinolfi; P E Polani
Journal:  Br Med J       Date:  1969-10-11

4.  Three patients with ring (X) chromosomes and a severe phenotype.

Authors:  N R Dennis; A L Collins; J A Crolla; A E Cockwell; A M Fisher; P A Jacobs
Journal:  J Med Genet       Date:  1993-06       Impact factor: 6.318

5.  Serum immunoglobulins and lymphocyte subpopulations derangement in Turner's syndrome.

Authors:  E Cacciari; M Masi; M P Fantini; F Licastro; A Cicognani; P Pirazzoli; M P Villa; F Specchia; A Forabosco; C Franceschi; L Martoni
Journal:  J Immunogenet       Date:  1981-10

6.  Characterization of common variable immunodeficiency: identification of a subset of patients with distinctive immunophenotypic and clinical features.

Authors:  J J Wright; D K Wagner; R M Blaese; C Hagengruber; T A Waldmann; T A Fleisher
Journal:  Blood       Date:  1990-11-15       Impact factor: 22.113

7.  Idiopathic CD4+ T-lymphocytopenia--an analysis of five patients with unexplained opportunistic infections.

Authors:  T J Spira; B M Jones; J K Nicholson; R B Lal; T Rowe; A C Mawle; C B Lauter; J A Shulman; R A Monson
Journal:  N Engl J Med       Date:  1993-02-11       Impact factor: 91.245

8.  CD4+ lymphocytopenia due to common variable immunodeficiency mimicking AIDS.

Authors:  R S Kaczmarski; A D Webster; J Moxham; F Davison; S Sutherland; G J Mufti
Journal:  J Clin Pathol       Date:  1994-04       Impact factor: 3.411

9.  Immunologic studies in Turner syndrome before and during treatment with growth hormone. The Dutch Growth Hormone Working Group.

Authors:  C Rongen-Westerlaken; G T Rijkers; E J Scholtens; A van Es; J M Wit; J L van den Brande; B J Zegers
Journal:  J Pediatr       Date:  1991-08       Impact factor: 4.406

Review 10.  Molecular basis for three X-linked immune disorders.

Authors:  J M Puck
Journal:  Hum Mol Genet       Date:  1994       Impact factor: 6.150

View more
  3 in total

Review 1.  Do ribosomopathies explain some cases of common variable immunodeficiency?

Authors:  S Khan; J Pereira; P J Darbyshire; S Holding; P C Doré; W A C Sewell; A Huissoon
Journal:  Clin Exp Immunol       Date:  2010-11-09       Impact factor: 4.330

2.  Increased prevalence of autoimmunity in Turner syndrome--influence of age.

Authors:  K H Mortensen; L Cleemann; B E Hjerrild; E Nexo; H Locht; E M Jeppesen; C H Gravholt
Journal:  Clin Exp Immunol       Date:  2009-02-26       Impact factor: 4.330

3.  Prevalence of Celiac Disease in Patients With Turner Syndrome: Systematic Review and Meta-Analysis.

Authors:  Ghada S M Al-Bluwi; Asma H AlNababteh; Linda Östlundh; Saif Al-Shamsi; Rami H Al-Rifai
Journal:  Front Med (Lausanne)       Date:  2021-06-17
  3 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.