Literature DB >> 9350810

Spectrum of clinical features associated with interstitial chromosome 22q11 deletions: a European collaborative study.

A K Ryan1, J A Goodship, D I Wilson, N Philip, A Levy, H Seidel, S Schuffenhauer, H Oechsler, B Belohradsky, M Prieur, A Aurias, F L Raymond, J Clayton-Smith, E Hatchwell, C McKeown, F A Beemer, B Dallapiccola, G Novelli, J A Hurst, J Ignatius, A J Green, R M Winter, L Brueton, K Brøndum-Nielsen, P J Scambler.   

Abstract

We present clinical data on 558 patients with deletions within the DiGeorge syndrome critical region of chromosome 22q11. Twenty-eight percent of the cases where parents had been tested had inherited deletions, with a marked excess of maternally inherited deletions (maternal 61, paternal 18). Eight percent of the patients had died, over half of these within a month of birth and the majority within 6 months. All but one of the deaths were the result of congenital heart disease. Clinically significant immunological problems were very uncommon. Nine percent of patients had cleft palate and 32% had velopharyngeal insufficiency, 60% of patients were hypocalcaemic, 75% of patients had cardiac problems, and 36% of patients who had abdominal ultrasound had a renal abnormality. Sixty-two percent of surviving patients were developmentally normal or had only mild learning problems. The majority of patients were constitutionally small, with 36% of patients below the 3rd centile for either height or weight parameters.

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Year:  1997        PMID: 9350810      PMCID: PMC1051084          DOI: 10.1136/jmg.34.10.798

Source DB:  PubMed          Journal:  J Med Genet        ISSN: 0022-2593            Impact factor:   6.318


  17 in total

1.  A prospective cytogenetic study of 36 cases of DiGeorge syndrome.

Authors:  D I Wilson; I E Cross; J A Goodship; J Brown; P J Scambler; H H Bain; J F Taylor; K Walsh; A Bankier; J Burn
Journal:  Am J Hum Genet       Date:  1992-11       Impact factor: 11.025

2.  Late-onset psychosis in the velo-cardio-facial syndrome.

Authors:  R J Shprintzen; R Goldberg; K J Golding-Kushner; R W Marion
Journal:  Am J Med Genet       Date:  1992-01-01

3.  DiGeorge syndrome: part of CATCH 22.

Authors:  D I Wilson; J Burn; P Scambler; J Goodship
Journal:  J Med Genet       Date:  1993-10       Impact factor: 6.318

4.  Velocardiofacial syndrome in a mother and daughter: variability of the clinical phenotype.

Authors:  S E Holder; R M Winter; S Kamath; P J Scambler
Journal:  J Med Genet       Date:  1993-10       Impact factor: 6.318

Review 5.  Velocardiofacial (Shprintzen) syndrome: an important syndrome for the dysmorphologist to recognise.

Authors:  A H Lipson; D Yuille; M Angel; P G Thompson; J G Vandervoord; E J Beckenham
Journal:  J Med Genet       Date:  1991-09       Impact factor: 6.318

Review 6.  Velo-cardio-facial syndrome: a review of 120 patients.

Authors:  R Goldberg; B Motzkin; R Marion; P J Scambler; R J Shprintzen
Journal:  Am J Med Genet       Date:  1993-02-01

7.  Cranial hemihypertrophy and neurodevelopmental prognosis.

Authors:  J C Dean; G F Cole; R E Appleton; J Burn; S A Roberts; D Donnai
Journal:  J Med Genet       Date:  1990-03       Impact factor: 6.318

8.  Cerebellar atrophy in a patient with velocardiofacial syndrome.

Authors:  D R Lynch; D M McDonald-McGinn; E H Zackai; B S Emanuel; D A Driscoll; L A Whitaker; K H Fischbeck
Journal:  J Med Genet       Date:  1995-07       Impact factor: 6.318

9.  Transient congenital hypoparathyroidism: resolution and recurrence in chromosome 22q11 deletion.

Authors:  F Greig; E Paul; J DiMartino-Nardi; P Saenger
Journal:  J Pediatr       Date:  1996-04       Impact factor: 4.406

Review 10.  DiGeorge syndrome and related syndromes associated with 22q11.2 deletions. A review.

Authors:  S Demczuk; A Aurias
Journal:  Ann Genet       Date:  1995
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  284 in total

1.  Highly skewed X-chromosome inactivation is associated with idiopathic recurrent spontaneous abortion.

Authors:  M C Lanasa; W A Hogge; C Kubik; J Blancato; E P Hoffman
Journal:  Am J Hum Genet       Date:  1999-07       Impact factor: 11.025

2.  Towards earlier diagnosis of 22q11 deletions.

Authors:  E S Tobias; N Morrison; M L Whiteford; J L Tolmie
Journal:  Arch Dis Child       Date:  1999-12       Impact factor: 3.791

3.  Patients' age at time of testing for chromosome 22q11 microdeletions: missed opportunities for genetic counseling.

Authors:  Y Liu; S Fallet; R Koppel
Journal:  Pediatr Cardiol       Date:  2000 Mar-Apr       Impact factor: 1.655

Review 4.  Science, medicine, and the future: Genetics and cardiovascular risk.

Authors:  I N Day; D I Wilson
Journal:  BMJ       Date:  2001-12-15

5.  Sixteen New Cases Contributing to the Characterization of Patients with Distal 22q11.2 Microduplications.

Authors:  J Wincent; D L Bruno; B W M van Bon; A Bremer; H Stewart; E M H F Bongers; C W Ockeloen; M H Willemsen; D D A Keays; G Baird; D F Newbury; T Kleefstra; C Marcelis; U Kini; Z Stark; R Savarirayan; L J Sheffield; O Zuffardi; H R Slater; B B de Vries; S J L Knight; B-M Anderlid; J Schoumans
Journal:  Mol Syndromol       Date:  2011-05-18

6.  High incidence of recurrent copy number variants in patients with isolated and syndromic Müllerian aplasia.

Authors:  Serena Nik-Zainal; Reiner Strick; Mekayla Storer; Ni Huang; Roland Rad; Lionel Willatt; Tomas Fitzgerald; Vicki Martin; Richard Sandford; Nigel P Carter; Andreas R Janecke; Stefan P Renner; Patricia G Oppelt; Peter Oppelt; Christine Schulze; Sara Brucker; Matthew Hurles; Matthias W Beckmann; Pamela L Strissel; Charles Shaw-Smith
Journal:  J Med Genet       Date:  2011-01-28       Impact factor: 6.318

Review 7.  Cranial neural crest cells on the move: their roles in craniofacial development.

Authors:  Dwight R Cordero; Samantha Brugmann; Yvonne Chu; Ruchi Bajpai; Maryam Jame; Jill A Helms
Journal:  Am J Med Genet A       Date:  2010-12-10       Impact factor: 2.802

8.  Aberrant interchromosomal exchanges are the predominant cause of the 22q11.2 deletion.

Authors:  Sulagna C Saitta; Stacy E Harris; Ann P Gaeth; Deborah A Driscoll; Donna M McDonald-McGinn; Melissa K Maisenbacher; Jill M Yersak; Prabir K Chakraborty; April M Hacker; Elaine H Zackai; Terry Ashley; Beverly S Emanuel
Journal:  Hum Mol Genet       Date:  2003-12-17       Impact factor: 6.150

9.  A Case of CATCH22 Syndrome Diagnosed in Postmenopausal Woman.

Authors:  Seung Kyung Lee; Min Jeong Lee; Hyo Jin Lee; Bu Kyung Kim; Young Bae Sohn; Yoon-Sok Chung
Journal:  J Bone Metab       Date:  2013-05-13

10.  Core histones and HIRIP3, a novel histone-binding protein, directly interact with WD repeat protein HIRA.

Authors:  S Lorain; J P Quivy; F Monier-Gavelle; C Scamps; Y Lécluse; G Almouzni; M Lipinski
Journal:  Mol Cell Biol       Date:  1998-09       Impact factor: 4.272

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