Literature DB >> 17684005

Immunological abnormalities in CHARGE syndrome.

Karin Writzl1, Catherine M Cale, Christine M Pierce, Louise C Wilson, Raoul C M Hennekam.   

Abstract

Immune deficiency can be part of CHARGE syndrome but often receives only limited attention. We present two patients with CHARGE syndrome confirmed CHD7 mutations who had severe T-cell deficiency, and review 15 CHARGE patients from the literature with immunological problems. Most of them had severe T-cell deficiency, although the spectrum also included mild T-cell deficiency and isolated humoral immune deficiency. We conclude that immunodeficiency can form an important symptom in CHARGE syndrome although the frequency and exact nature are still insufficiently known. We propose to evaluate immune functions in all CHARGE syndrome patients, to estimate the frequency and nature of the accompanying immunodeficiency, and to obtain better data regarding prognosis and management.

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Year:  2007        PMID: 17684005     DOI: 10.1016/j.ejmg.2007.05.002

Source DB:  PubMed          Journal:  Eur J Med Genet        ISSN: 1769-7212            Impact factor:   2.708


  17 in total

Review 1.  Chromodomain proteins in development: lessons from CHARGE syndrome.

Authors:  W S Layman; E A Hurd; D M Martin
Journal:  Clin Genet       Date:  2010-04-08       Impact factor: 4.438

Review 2.  Immunological aspects of 22q11.2 deletion syndrome.

Authors:  A R Gennery
Journal:  Cell Mol Life Sci       Date:  2011-10-09       Impact factor: 9.261

Review 3.  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

4.  More Clinical Overlap between 22q11.2 Deletion Syndrome and CHARGE Syndrome than Often Anticipated.

Authors:  N Corsten-Janssen; S C Saitta; L H Hoefsloot; D M McDonald-McGinn; D A Driscoll; R Derks; K A Dickinson; W S Kerstjens-Frederikse; B S Emanuel; E H Zackai; C M A van Ravenswaaij-Arts
Journal:  Mol Syndromol       Date:  2013-05-28

Review 5.  Molecular and phenotypic aspects of CHD7 mutation in CHARGE syndrome.

Authors:  Gabriel E Zentner; Wanda S Layman; Donna M Martin; Peter C Scacheri
Journal:  Am J Med Genet A       Date:  2010-03       Impact factor: 2.802

6.  Successful cord blood transplantation for a CHARGE syndrome with CHD7 mutation showing DiGeorge sequence including hypoparathyroidism.

Authors:  Hirosuke Inoue; Hidetoshi Takada; Takeshi Kusuda; Takako Goto; Masayuki Ochiai; Tadamune Kinjo; Jun Muneuchi; Yasushi Takahata; Naomi Takahashi; Tomohiro Morio; Kenjiro Kosaki; Toshiro Hara
Journal:  Eur J Pediatr       Date:  2010-01-06       Impact factor: 3.183

7.  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

8.  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

9.  Great vessel development requires biallelic expression of Chd7 and Tbx1 in pharyngeal ectoderm in mice.

Authors:  Victoria Randall; Karen McCue; Catherine Roberts; Vanessa Kyriakopoulou; Sarah Beddow; Angela N Barrett; Francesca Vitelli; Katrina Prescott; Charles Shaw-Smith; Koen Devriendt; Erika Bosman; Georg Steffes; Karen P Steel; Subreena Simrick; M Albert Basson; Elizabeth Illingworth; Peter J Scambler
Journal:  J Clin Invest       Date:  2009-10-12       Impact factor: 14.808

10.  Mutations in CHD7 in patients with CHARGE syndrome cause T-B + natural killer cell + severe combined immune deficiency and may cause Omenn-like syndrome.

Authors:  A R Gennery; M A Slatter; J Rice; L H Hoefsloot; D Barge; A McLean-Tooke; T Montgomery; J A Goodship; A D Burt; T J Flood; M Abinun; A J Cant; D Johnson
Journal:  Clin Exp Immunol       Date:  2008-05-26       Impact factor: 4.330

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