Literature DB >> 8237917

Velo-cardio-facial syndrome. Intrafamilial variability of the phenotype.

S D McLean1, H M Saal, N B Spinner, B S Emanuel, D A Driscoll.   

Abstract

We describe a mother and son with velo-cardio-facial syndrome (VCFS) in whom cytogenetic and DNA molecular studies demonstrate an interstitial deletion of the long arm of chromosome 22. Although these two individuals manifest the typical facial and cognitive features of VCFS, they are discordant for the cardiovascular and palatal anomalies, which are seminal manifestations of the disorder. Previously, this degree of phenotypic variability had not been well appreciated within a single family segregating the VCFS deletion. A review of other familial cases of VCFS suggests that the family described in this article is not atypical. Because a microdeletion would be expected to be inherited without alteration within individual families, the phenotypic variability observed in these families appears to be an intrinsic quality of the syndrome and not wholly due to genetic heterogeneity.

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Year:  1993        PMID: 8237917     DOI: 10.1001/archpedi.1993.02160350086013

Source DB:  PubMed          Journal:  Am J Dis Child        ISSN: 0002-922X


  7 in total

1.  Evaluation of potential modifiers of the palatal phenotype in the 22q11.2 deletion syndrome.

Authors:  Deborah A Driscoll; Torrey Boland; Beverly S Emanuel; Richard E Kirschner; Don LaRossa; Jeanne Manson; Donna McDonald-McGinn; Peter Randall; Cynthia Solot; Elaine Zackai; Laura E Mitchell
Journal:  Cleft Palate Craniofac J       Date:  2006-07

Review 2.  22q11 deletion syndrome: a genetic subtype of schizophrenia.

Authors:  A S Bassett; E W Chow
Journal:  Biol Psychiatry       Date:  1999-10-01       Impact factor: 13.382

Review 3.  Phenotype of adults with the 22q11 deletion syndrome: A review.

Authors:  E Cohen; E W Chow; R Weksberg; A S Bassett
Journal:  Am J Med Genet       Date:  1999-10-08

Review 4.  When half is not enough: gene expression and dosage in the 22q11 deletion syndrome.

Authors:  D W Meechan; T M Maynard; D Gopalakrishna; Y Wu; A S LaMantia
Journal:  Gene Expr       Date:  2007

Review 5.  Excess of deletions of maternal origin in the DiGeorge/velo-cardio-facial syndromes. A study of 22 new patients and review of the literature.

Authors:  S Demczuk; A Lévy; M Aubry; M F Croquette; N Philip; M Prieur; U Sauer; P Bouvagnet; G A Rouleau; G Thomas
Journal:  Hum Genet       Date:  1995-07       Impact factor: 4.132

6.  Chromosome 22q11.2 microdeletion in monozygotic twins with discordant phenotype and deletion size.

Authors:  Ashutosh Halder; Manish Jain; Isha Chaudhary; Binuja Varma
Journal:  Mol Cytogenet       Date:  2012-03-13       Impact factor: 2.009

Review 7.  Genetics of bipolar disorder.

Authors:  Berit Kerner
Journal:  Appl Clin Genet       Date:  2014-02-12
  7 in total

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