Literature DB >> 3688017

Duplication of proximal 15q as a cause of Prader-Willi syndrome.

A L Pettigrew1, S M Gollin, F Greenberg, V M Riccardi, D H Ledbetter.   

Abstract

We describe an apparent duplication of proximal 15q, i.e., 15q11q12 or 15q12q13 in two patients. Prometaphase chromosome analysis, C-banding and distamycin A/DAPI staining were used to exclude a translocation between the abnormal 15 homolog and another chromosome. The 2 patients have many manifestations of the Prader-Willi syndrome (PWS) including at least 5 of the following: obesity, compulsive eating, mental retardation, short stature, central hypotonia, hypogonadism, small hands and feet, hypopigmentation, and feeding problems in infancy. Results of high resolution chromosome analysis of the parents of both patients were normal. A comparison between these patients and 2 subjects from previous reports demonstrates phenotypic heterogeneity among the duplication 15q PWS patients. Two patients have the hypopigmentation seen in chromosomally normal and deletion PWS patients. These cases add to the variety of chromosome 15 aberrations which are associated with PWS.

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Year:  1987        PMID: 3688017     DOI: 10.1002/ajmg.1320280403

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


  15 in total

1.  Inherited DNA amplification of the proximal 15q region: cytogenetic and molecular studies.

Authors:  C Mignon; F Parente; C Stavropoulou; P Collignon; A Moncla; C Turc-Carel; M G Mattei
Journal:  J Med Genet       Date:  1997-03       Impact factor: 6.318

2.  Reciprocal translocation between the proximal regions of the long arms of chromosomes 13 and 15 resulting in unbalanced offspring: characterization by fluorescence in situ hybridization and DNA analysis.

Authors:  K Mangelschots; B Van Roy; F Speleman; N Van Roy; J Gheuens; J Beuten; I Buntinx; M N Van Thienen; H Willekens; J Dumon
Journal:  Hum Genet       Date:  1992-06       Impact factor: 4.132

3.  Supernumerary marker 15 chromosomes: a clinical, molecular and FISH approach to diagnosis and prognosis.

Authors:  J A Crolla; J F Harvey; F L Sitch; N R Dennis
Journal:  Hum Genet       Date:  1995-02       Impact factor: 4.132

4.  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

Review 5.  Angelman ("happy puppet") syndrome--seven new cases documented by cerebral computed tomography: review of the literature.

Authors:  A Dörries; H L Spohr; J Kunze
Journal:  Eur J Pediatr       Date:  1988-12       Impact factor: 3.183

6.  Molecular characterisation of four cases of intrachromosomal triplication of chromosome 15q11-q14.

Authors:  P Ungaro; S L Christian; J A Fantes; A Mutirangura; S Black; J Reynolds; S Malcolm; W B Dobyns; D H Ledbetter
Journal:  J Med Genet       Date:  2001-01       Impact factor: 6.318

7.  Molecular characterization of two proximal deletion breakpoint regions in both Prader-Willi and Angelman syndrome patients.

Authors:  S L Christian; W P Robinson; B Huang; A Mutirangura; M R Line; M Nakao; U Surti; A Chakravarti; D H Ledbetter
Journal:  Am J Hum Genet       Date:  1995-07       Impact factor: 11.025

Review 8.  Duplication of chromosome 15 in the region 15q11-13 in a patient with developmental delay and ataxia with similarities to Angelman syndrome.

Authors:  J Clayton-Smith; T Webb; X J Cheng; M E Pembrey; S Malcolm
Journal:  J Med Genet       Date:  1993-06       Impact factor: 6.318

9.  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

Review 10.  The comorbidity of autism with the genomic disorders of chromosome 15q11.2-q13.

Authors:  Amber Hogart; David Wu; Janine M LaSalle; N Carolyn Schanen
Journal:  Neurobiol Dis       Date:  2008-09-18       Impact factor: 5.996

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