Literature DB >> 19596467

MLPA analysis in 30 Sotos syndrome patients revealed one total NSD1 deletion and two partial deletions not previously reported.

Claudia Fagali1, Fernando Kok, Pablo Nicola, Chong Kim, Débora Bertola, Lílian Albano, Célia P Koiffmann.   

Abstract

Sotos syndrome (MIM #117550) is an autosomal dominant condition characterized by pre and postnatal overgrowth, macrocephaly and typical facial gestalt with frontal bossing, hypertelorism, antimongoloid slant of the palpebral fissures, prominent jaw and high and narrow palate. This syndrome is also frequently associated with brain, cardiovascular, and urinary anomalies and is occasionally accompanied by malignant lesions such as Wilms tumour and hepatocarcinoma. The syndrome is known to be caused by mutations or deletions of the NSD1 gene. To detect both 5q35 microdeletions and partial NSD1 gene deletions we screened 30 Brazilian patients with clinical diagnosis of Sotos syndrome by multiplex ligation dependent probe amplification. We identified one patient with a total deletion of NSD1 and neighbouring FGFR4, other with missing NSD1 exons 13-14 and another with a deletion involving FGFR4 and spanning up to NSD1 exon 17. All deletions were de novo. The two NSD1 partial deletions have not been previously reported. The clinical features of the three patients included a typical facial gestalt with frontal bossing, prominent jaw and high anterior hairline; macrocephaly, dolichocephaly, large hands; neonatal hypotonia and jaundice. All presented normal growth at birth but postnatal overgrowth. Two patients with NSD1 and FGFR4 gene deletions presented congenital heart anomalies.

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Year:  2009        PMID: 19596467     DOI: 10.1016/j.ejmg.2009.07.001

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


  6 in total

1.  Deletion of NSD1 exon 14 in Sotos syndrome: first description.

Authors:  Maria Piccione; Valeria Consiglio; Antonella Di Fiore; Marina Grasso; Massimiliano Cecconi; Lucia Perroni; Giovanni Corsello
Journal:  J Genet       Date:  2011-04       Impact factor: 1.166

2.  Clinical and molecular heterogeneity in brazilian patients with sotos syndrome.

Authors:  Gustavo H Vieira; Melissa M Cook; Renata L Ferreira De Lima; Carlos E Frigério Domingues; Daniel R de Carvalho; Isaias Soares de Paiva; Danilo Moretti-Ferreira; Anand K Srivastava
Journal:  Mol Syndromol       Date:  2015-01-21

3.  Steric Clash in the SET Domain of Histone Methyltransferase NSD1 as a Cause of Sotos Syndrome and Its Genetic Heterogeneity in a Brazilian Cohort.

Authors:  Kyungsoo Ha; Priya Anand; Jennifer A Lee; Julie R Jones; Chong Ae Kim; Debora Romeo Bertola; Jonathan D J Labonne; Lawrence C Layman; Wolfgang Wenzel; Hyung-Goo Kim
Journal:  Genes (Basel)       Date:  2016-11-09       Impact factor: 4.096

4.  Case Report of Neonatal Sotos Syndrome with a New Missense Mutation in the NSD1 Gene and Literature Analysis in the Chinese Han Population.

Authors:  Hui-Ying Jin; Hai-Feng Li; Jia-Lu Xu; Wang Hui; Wen-Cong Ruan; Cheng-Cheng Lv; Ren-Ai Xu; Shu Qiang
Journal:  Medicina (Kaunas)       Date:  2022-07-21       Impact factor: 2.948

5.  Identification of novel genomic imbalances in Saudi patients with congenital heart disease.

Authors:  Waad Albawardi; Faten Almutairi; Zuhair N Al-Hassnan; Rawan AlMass; Albandary AlBakheet; Osama M Mustafa; Laila AlQuait; Zarghuna M A Shinwari; Salma Wakil; Mustafa A Salih; Majid Al-Fayyadh; Saeed M Hassan; Mansour Aljoufan; Osima Al-Nakhli; Brynn Levy; Balsam AlMaarik; Hana A Al-Hakami; Maysoon Alsagob; Dilek Colak; Namik Kaya
Journal:  Mol Cytogenet       Date:  2018-01-25       Impact factor: 2.009

Review 6.  Genetic Predisposition to Solid Pediatric Cancers.

Authors:  Mario Capasso; Annalaura Montella; Matilde Tirelli; Teresa Maiorino; Sueva Cantalupo; Achille Iolascon
Journal:  Front Oncol       Date:  2020-10-28       Impact factor: 6.244

  6 in total

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