Literature DB >> 2858158

Deletions of proximal 15q and non-classical Prader-Willi syndrome phenotypes.

S Schwartz, S R Max, S R Panny, M M Cohen.   

Abstract

A deletion of the long arm of chromosome 15 (usually involving bands 15q11-q12) has been seen in approximately 50% of Prader-Willi syndrome (PWS) patients [Ledbetter et al, 1982]. However, 14 patients with non-PWS (or atypical PWS) phenotype with 15q deletion indicate great clinical variability. A deletion was found in a propositus with a de novo translocation [45,XY, -15, -22, +rec(15;22) (22pter----22q13.2::15q14----15qter)], who had anomalies not normally observed in PWS patients. Activities of several enzymes mapped to the involved chromosomes were studied in the patient and control individuals. A 50% decrease in the level of arylsulfatase-A confirmed a small deletion in 22q(22q13.2----qter), and additional studies localized more precisely the loci for alpha-mannosidase (cytoplasmic) and beta-galactosidase.

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Year:  1985        PMID: 2858158     DOI: 10.1002/ajmg.1320200208

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  8 in total

1.  Chromosome abnormalities in pupils attending ESN/M schools.

Authors:  M A Lamont; N R Dennis; M Seabright
Journal:  Arch Dis Child       Date:  1986-03       Impact factor: 3.791

2.  Similar molecular deletions on chromosome 15q11.2 are encountered in both the Prader-Willi and Angelman syndromes.

Authors:  T A Donlon
Journal:  Hum Genet       Date:  1988-12       Impact factor: 4.132

3.  Molecular, cytogenetic, and clinical investigations of Prader-Willi syndrome patients.

Authors:  W P Robinson; A Bottani; Y G Xie; J Balakrishman; F Binkert; M Mächler; A Prader; A Schinzel
Journal:  Am J Hum Genet       Date:  1991-12       Impact factor: 11.025

4.  An interstitial 15q11-q14 deletion: expanded Prader-Willi syndrome phenotype.

Authors:  Merlin G Butler; Douglas C Bittel; Nataliya Kibiryeva; Linda D Cooley; Shihui Yu
Journal:  Am J Med Genet A       Date:  2010-02       Impact factor: 2.802

5.  Evaluation of potential models for imprinted and nonimprinted components of human chromosome 15q11-q13 syndromes by fine-structure homology mapping in the mouse.

Authors:  R D Nicholls; W Gottlieb; L B Russell; M Davda; B Horsthemke; E M Rinchik
Journal:  Proc Natl Acad Sci U S A       Date:  1993-03-01       Impact factor: 11.205

6.  A genetic model for the Prader-Willi syndrome and its implication for Angelman syndrome.

Authors:  I Kennerknecht
Journal:  Hum Genet       Date:  1992 Sep-Oct       Impact factor: 4.132

7.  Prader-Willi syndrome: current understanding of cause and diagnosis.

Authors:  M G Butler
Journal:  Am J Med Genet       Date:  1990-03

8.  Complex chromosome rearrangements related 15q14 microdeletion plays a relevant role in phenotype expression and delineates a novel recurrent syndrome.

Authors:  Maria Cristina Roberti; Cecilia Surace; Maria Cristina Digilio; Gemma D'Elia; Pietro Sirleto; Rossella Capolino; Antonietta Lombardo; Anna Cristina Tomaiuolo; Stefano Petrocchi; Adriano Angioni
Journal:  Orphanet J Rare Dis       Date:  2011-04-19       Impact factor: 4.123

  8 in total

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