Literature DB >> 23633107

Expanded Prader-Willi syndrome due to chromosome 15q11.2-14 deletion: report and a review of literature.

Anthony P Y Liu1, Wing Fai Tang, Elizabeth T Lau, Kelvin Y K Chan, Anita S Y Kan, Kar Yin Wong, Winnie W Y Tso, Khair Jalal, So Lun Lee, Christy S K Chau, Brian H Y Chung.   

Abstract

We report on a male infant with de novo unbalanced t(5;15) translocation resulting in a 17.23 Mb deletion within 15q11.2-q14 and a 25.12 kb deletion in 5pter. The 15q11.2-q14 deletion encompassed the 15q11.2-q13 Prader-Willi syndrome (PWS) critical region and the recently described 15q13.3 microdeletion syndrome region while the 5pter deletion contained no RefSeq genes. From our literature review, patients with similar deletions in chromosome 15q exhibit expanded phenotype of severe developmental delay, protracted feeding problem, absent speech, central visual impairment, congenital malformations and epilepsy in addition to those typical of PWS. The patient reported herein had previously unreported anomalies of mega cisterna magna, horseshoe kidney and the rare neonatal interstitial lung disease known as pulmonary interstitial glycogenosis. Precise breakpoint delineation by microarray is useful in patients with atypical PWS deletions to guide investigation and prognostication.
Copyright © 2013 Wiley Periodicals, Inc.

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Year:  2013        PMID: 23633107     DOI: 10.1002/ajmg.a.35909

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  3 in total

1.  The clinical impact of chromosomal microarray on paediatric care in Hong Kong.

Authors:  Victoria Q Tao; Kelvin Y K Chan; Yoyo W Y Chu; Gary T K Mok; Tiong Y Tan; Wanling Yang; So Lun Lee; Wing Fai Tang; Winnie W Y Tso; Elizabeth T Lau; Anita S Y Kan; Mary H Tang; Yu-Lung Lau; Brian H Y Chung
Journal:  PLoS One       Date:  2014-10-15       Impact factor: 3.240

2.  Neural crest cells require Meis2 for patterning the mandibular arch via the Sonic hedgehog pathway.

Authors:  Jaroslav Fabik; Katarina Kovacova; Zbynek Kozmik; Ondrej Machon
Journal:  Biol Open       Date:  2020-07-02       Impact factor: 2.422

3.  Genetic subtypes and phenotypic characteristics of 110 patients with Prader-Willi syndrome.

Authors:  Lu Zhang; Xiaoliang Liu; Yunjing Zhao; Qingyi Wang; Yuanyuan Zhang; Haiming Gao; Bijun Zhang; Wanting Cui; Yanyan Zhao
Journal:  Ital J Pediatr       Date:  2022-07-23       Impact factor: 3.288

  3 in total

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