Literature DB >> 15931284

Immune deficiency in CHARGE association.

Demetrios S Theodoropoulos1.   

Abstract

CHARGE association is the sporadic, non-random concurrence of Coloboma of the eye, Heart anomalies, choanal Atresia, Retardation of growth and development, Genitourinary anomalies, Ear anomalies and deafness (CHARGE association). Other abnormalities have also been reported in small numbers of patients with CHARGE association. The molecular basis of the CHARGE association is not clear. The spectrum of CHARGE association anomalies is wide and includes multiple systems. CHARGE association shares features with DiGeorge sequence, but no specific immune abnormalities are identified with the CHARGE association. The present study reports immune defects observed in three patients with CHARGE association. All patients presented with frequent upper and lower respiratory infections. The underlying immune abnormalities differ: one patient has impaired T-cell proliferation and poor antibody response to polysaccharide (pneumococcal) antigens; another has T-cell lymphopenia; and the third has a mild IgG2 subclass deficiency. Their course has so far been benign and they are all managed with prophylactic antibiotics. Although no single abnormality of the immune system is recognized in these patients, immune deficiency is considered among the occasional components of the CHARGE association.

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Year:  2003        PMID: 15931284      PMCID: PMC1069020          DOI: 10.3121/cmr.1.1.43

Source DB:  PubMed          Journal:  Clin Med Res        ISSN: 1539-4182


  18 in total

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Journal:  J Pediatr       Date:  1981-08       Impact factor: 4.406

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Journal:  Nature       Date:  1988 Dec 22-29       Impact factor: 49.962

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Authors:  D J Wood; T J David; I L Chrystie; B Totterdell
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  7 in total

1.  Bilateral choanal atresia and paranasal sinus hypoplasia in an adult patient with hypogammaglobulinaemia.

Authors:  Hamid El-Sawy; M Azher Siddiq; Aubu Anbarasu
Journal:  Eur Arch Otorhinolaryngol       Date:  2006-07-15       Impact factor: 2.503

Review 2.  CHARGE syndrome: a review of the immunological aspects.

Authors:  Monica T Y Wong; Elisabeth H Schölvinck; Annechien J A Lambeck; Conny M A van Ravenswaaij-Arts
Journal:  Eur J Hum Genet       Date:  2015-02-18       Impact factor: 4.246

3.  Congenital T cell deficiency in a patient with CHARGE syndrome.

Authors:  Julie Hoover-Fong; William J Savage; Emily Lisi; Jerry Winkelstein; George H Thomas; Lies H Hoefsloot; David M Loeb
Journal:  J Pediatr       Date:  2009-01       Impact factor: 4.406

4.  CHARGE (coloboma, heart defect, atresia choanae, retarded growth and development, genital hypoplasia, ear anomalies/deafness) syndrome and chromosome 22q11.2 deletion syndrome: a comparison of immunologic and nonimmunologic phenotypic features.

Authors:  Soma Jyonouchi; Donna M McDonald-McGinn; Sherri Bale; Elaine H Zackai; Kathleen E Sullivan
Journal:  Pediatrics       Date:  2009-05       Impact factor: 7.124

5.  Helios expression in T-regulatory cells in patients with di George Syndrome.

Authors:  Adam Klocperk; Jarmila Grecová; Kristýna Šišmová; Jana Kayserová; Eva Froňková; Anna Šedivá
Journal:  J Clin Immunol       Date:  2014-07-10       Impact factor: 8.317

Review 6.  Atopic disorders in CHARGE syndrome: A retrospective study and literature review.

Authors:  Fang Kong; Donna M Martin
Journal:  Eur J Med Genet       Date:  2017-11-27       Impact factor: 2.708

7.  Immune Dysfunction in Children with CHARGE Syndrome: A Cross-Sectional Study.

Authors:  Monica T Y Wong; Annechien J A Lambeck; Mirjam van der Burg; Sacha la Bastide-van Gemert; Lianne A Hogendorf; Conny M A van Ravenswaaij-Arts; Elisabeth H Schölvinck
Journal:  PLoS One       Date:  2015-11-06       Impact factor: 3.240

  7 in total

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