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.

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

NOVARA, FRANCESCA;PRAMPARO, TIZIANO;ZUFFARDI, ORSETTA;CICCONE, ROBERTO
2013-01-01

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.
2013
Molecular Biology & Genetics considers all aspects of basic and applied genetics, including molecular genetics, prokaryotic and eukaryotic gene expression, mechanisms of mutagenesis, structure, function and regulation of genetic material. Also included are resources concerned with clinical genetics, patterns of inheritance, genetic cause, and screening and treatment of disease. Resources dealing specifically with developmentally regulated gene expression, or with signal transduction pathways that modulate gene expression at the cellular level are excluded and are covered in the Cell and Developmental Biology category.
Esperti anonimi
Inglese
Internazionale
STAMPA
56
1
54
58
5
Abnormalities; Multiple; diagnosis/genetics, Child; Preschool, Chromosome Disorders; diagnosis/genetics, Chromosome Duplication; genetics, Chromosomes; Human; Pair 5, Comparative Genomic Hybridization, Facies, Female, Foot Deformities; Congenital; genetics, Genetic Association Studies, Hand Deformities; genetics, Humans, Phenotype, Proteins; genetics, Syndrome
http://dx.doi.org/10.1016/j.ejmg.2012.10.002
10
info:eu-repo/semantics/article
262
Novara, Francesca; E., Alfei; S., D'Arrigo; C., Pantaleoni; S., Beri; V., Achille; F. L., Sciacca; Pramparo, Tiziano; Zuffardi, Orsetta; Ciccone, Robe...espandi
1 Contributo su Rivista::1.1 Articolo in rivista
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Utilizza questo identificativo per citare o creare un link a questo documento: https://hdl.handle.net/11571/982660
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