Literature DB >> 23085304

5p13 microduplication syndrome: a new case and better clinical definition of the syndrome.

Francesca Novara1, Enrico Alfei, Stefano D'Arrigo, Chiara Pantaleoni, Silvana Beri, Valentina Achille, Francesca L Sciacca, Roberto Giorda, Orsetta Zuffardi, Roberto Ciccone.   

Abstract

Chromosome 5p13 duplication syndrome (OMIM #613174), a contiguous gene syndrome involving duplication of several genes on chromosome 5p13 including NIPBL (OMIM 608667), has been described in rare patients with developmental delay and learning disability, behavioral problems and peculiar facial dysmorphisms. 5p13 duplications described so far present with variable sizes, from 0.25 to 13.6 Mb, and contain a variable number of genes. Here we report another patient with 5p13 duplication syndrome including NIPBL gene only. Proband's phenotype overlapped that reported in patients with 5p13 microduplication syndrome and especially that of subjects with smaller duplications. Moreover, we better define genotype-phenotype relationship associated with this duplication and confirmed that NIPBL was likely the major dosage sensitive gene for the 5p13 microduplication phenotype.
Copyright © 2012 Elsevier Masson SAS. All rights reserved.

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Year:  2012        PMID: 23085304     DOI: 10.1016/j.ejmg.2012.10.002

Source DB:  PubMed          Journal:  Eur J Med Genet        ISSN: 1769-7212            Impact factor:   2.708


  5 in total

1.  Electroclinical characteristics and neuropsychological profile of a female child with chromosome 5p13.2 duplication syndrome.

Authors:  Elisabetta Lucarelli; Maria Grazia Pasca; Isabella Fanizza; Antonio Trabacca
Journal:  Neurol Sci       Date:  2017-01-20       Impact factor: 3.307

2.  Copy number analysis of NIPBL in a cohort of 510 patients reveals rare copy number variants and a mosaic deletion.

Authors:  Yu-Wei Cheng; Christopher A Tan; Agata Minor; Kelly Arndt; Latrice Wysinger; Dorothy K Grange; Beth A Kozel; Nathaniel H Robin; Darrel Waggoner; Carrie Fitzpatrick; Soma Das; Daniela Del Gaudio
Journal:  Mol Genet Genomic Med       Date:  2013-11-14       Impact factor: 2.183

3.  Novel phenotype of 5p13.3-q11.2 duplication resulting from supernumerary marker chromosome 5: implications for management and genetic counseling.

Authors:  Margaret E Armstrong; David D Weaver; Melissa D Lah; Gail H Vance; Benjamin J Landis; Stephanie M Ware; Benjamin M Helm
Journal:  Mol Cytogenet       Date:  2018-03-27       Impact factor: 2.009

4.  A de novo triplication on 2q22.3 including the entire ZEB2 gene associated with global developmental delay, multiple congenital anomalies and behavioral abnormalities.

Authors:  Haiming Yuan; Lina Zhang; Mengfan Chen; Junping Zhu; Zhe Meng; Liyang Liang
Journal:  Mol Cytogenet       Date:  2015-12-23       Impact factor: 2.009

5.  A boy with developmental delay and mosaic supernumerary inv dup(5)(p15.33p15.1) leading to distal 5p tetrasomy - case report and review of the literature.

Authors:  Pavel Tesner; Jana Drabova; Miroslav Stolfa; Martin Kudr; Martin Kyncl; Veronika Moslerova; Drahuse Novotna; Radka Kremlikova Pourova; Eduard Kocarek; Tereza Rasplickova; Zdenek Sedlacek; Marketa Vlckova
Journal:  Mol Cytogenet       Date:  2018-05-09       Impact factor: 2.009

  5 in total

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