Literature DB >> 23782367

Microarray analysis of unbalanced translocation in Wolf-Hirschhorn syndrome.

Ying Dai1, Jing Yang, Yuanyuan Chen, Liming Bao, Qian Cheng.   

Abstract

Wolf-Hirschhorn syndrome (WHS) is caused by deletions involving chromosome region 4p16.3, which is characterized by growth delay, mild-to-severe mental retardation, hypotonia, facial dysmorphisms and shows extensive phenotypic variability include feeding difficulties, epilepsy and congenital anomalies. Variation in the size of the deletion involving chromosome region 4p16.3 may explain the clinical variation. However, previous studies indicate that duplication for another chromosome region due to an unbalanced translocation elucidate approximately 40-45% WHS patients. Therefore, we used whole genomic cytogenetics array to analyze the entire genome at a significantly higher resolution over conventional cytogenetics to characterize the exact subtelomeric aberration region of one patient with developmental delay and several facial characteristics reminiscent Wolf-Hirschhorn syndrome. Here we report that our patient had 3.7 Mb deletion at the 4p16.2 and 6.8 Mb duplication at 8p23.1 resulted from the unbalanced translocations der(4)t(4;8)(p16.2;p23.1). We confirmed that our patient with monosomy 4p16.2 which is consistent with Wolf-Hirschhorn syndrome and trisomy 8p23.1. The combination of the 4p deletion with 8p partial trisomy explains the complex phenotype presented by our patient.
© 2013 The Authors. Pediatrics International © 2013 Japan Pediatric Society.

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Year:  2013        PMID: 23782367     DOI: 10.1111/j.1442-200X.2012.03684.x

Source DB:  PubMed          Journal:  Pediatr Int        ISSN: 1328-8067            Impact factor:   1.524


  2 in total

1.  Meiotic and pedigree segregation analyses in carriers of t(4;8)(p16;p23.1) differing in localization of breakpoint positions at 4p subband 4p16.3 and 4p16.1.

Authors:  Alina T Midro; Marcella Zollino; Ewa Wiland; Barbara Panasiuk; Piotr S Iwanowski; Marina Murdolo; Robert Śmigiel; Maria Sąsiadek; Jacek Pilch; Maciej Kurpisz
Journal:  J Assist Reprod Genet       Date:  2015-12-04       Impact factor: 3.412

2.  18q22.1-qter deletion and 4p16.3 microduplication in a boy with speech delay and mental retardation: case report and review of the literature.

Authors:  Chunjing Wang; Huanhuan Ren; Huaifu Dong; Meng Liang; Qi Wu; Yaping Liao
Journal:  Mol Cytogenet       Date:  2018-10-19       Impact factor: 2.009

  2 in total

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