Document Detail


T-cell immunodeficiency in CHARGE syndrome.
MedLine Citation:
PMID:  18976358     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
CHARGE syndrome comprises coloboma of the eye, heart defects, choanal atresia, growth and developmental retardation, genitourinary anomalies and ear and hearing defects. The association between CHARGE syndrome and T-cell immunodeficiency is recognized, but has not been reported widely in the literature. We report four patients meeting the diagnostic criteria for CHARGE syndrome, who had moderate or severe T-cell lymphopenia complicated by infections. The patients presented in Leicester, UK, between 2000 and 2007. All patients were negative for 22q11.2 deletions by FISH analysis, but mutations in the CHD7 gene were identified in three patients in whom the analysis was performed. Our cases indicate that patients with CHARGE syndrome may have a spectrum of T-cell immune deficiency, and that this association may be more common than has previously been appreciated. We recommend that all patients diagnosed with CHARGE syndrome should have lymphocyte subsets evaluated as part of their initial investigation. Conclusion: Thymic hypoplasia should be included in the clinical features associated with CHARGE syndrome. All patients with CHARGE syndrome should have lymphocyte subset analysis performed, to exclude T-cell immunodeficiency.
Authors:
Charu Chopra; Richard Baretto; Michael Duddridge; Michael J Browning
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Publication Detail:
Type:  Case Reports; Journal Article     Date:  2008-10-13
Journal Detail:
Title:  Acta paediatrica (Oslo, Norway : 1992)     Volume:  98     ISSN:  1651-2227     ISO Abbreviation:  Acta Paediatr.     Publication Date:  2009 Feb 
Date Detail:
Created Date:  2009-01-15     Completed Date:  2009-05-05     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  9205968     Medline TA:  Acta Paediatr     Country:  Norway    
Other Details:
Languages:  eng     Pagination:  408-10     Citation Subset:  IM    
Affiliation:
Department of Immunology, Leicester Royal Infirmary, University Hospitals of Leicester NHS Trust, Leicester LE1 5WW, UK.
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MeSH Terms
Descriptor/Qualifier:
Abnormalities, Multiple*
Choanal Atresia / complications*
Coloboma / complications*
Female
Genitalia / abnormalities*
Growth Disorders / complications*
Heart Defects, Congenital / complications*
Humans
Immunologic Deficiency Syndromes / complications*
Infant, Newborn
Male
Syndrome
T-Lymphocytes / immunology*

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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