Literature DB >> 21738022

Phenotypic variability in a three-generation Northern Irish family with Sotos syndrome.

Deirdre E Donnelly1, Peter Turnpenny, Vivienne P M McConnell.   

Abstract

Sotos syndrome is an overgrowth disorder with autosomal dominant inheritance caused by mutations and deletions in the nuclear receptor Set domain-containing protein 1 gene. In general, affected individuals have an advanced bone age, macrocephaly, characteristic facial gestalt and learning difficulties. Genotype-phenotype correlations are unclear. Full penetrance is seen and 95% of cases are de novo. Here, we report a three-generation pedigree, with at least eight affected individuals, shown to harbour the nuclear receptor Set domain-containing protein 1 missense mutation c. 6115C>T. To our knowledge, this is the largest Sotos family reported. The observed phenotype is extremely variable, thus highlighting the clinical heterogeneity that may occur.

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Year:  2011        PMID: 21738022     DOI: 10.1097/MCD.0b013e328349182d

Source DB:  PubMed          Journal:  Clin Dysmorphol        ISSN: 0962-8827            Impact factor:   0.816


  2 in total

1.  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

2.  A Novel Missense Mutation of the NSD1 Gene Associated with Overgrowth in Three Generations of an Italian Family: Case Report, Differential Diagnosis, and Review of Mutations of NSD1 Gene in Familial Sotos Syndrome.

Authors:  Gianluigi Laccetta; Francesca Moscuzza; Angela Michelucci; Andrea Guzzetta; Sara Lunardi; Francesca Lorenzoni; Paolo Ghirri
Journal:  Front Pediatr       Date:  2017-11-07       Impact factor: 3.418

  2 in total

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