Literature DB >> 33571694

Hypersociability associated with developmental delay, macrocephaly and facial dysmorphism points to CHD3 mutations.

Juliette Coursimault1, François Lecoquierre1, Pascale Saugier-Veber1, Valérie Drouin-Garraud2, Joël Lechevallier3, Anne Boland4, Jean-François Deleuze4, Thierry Frebourg1, Gaël Nicolas1, Anne-Claire Brehin5.   

Abstract

CHD3-related syndrome, also known as Snijders Blok-Campeau syndrome, is a rare developmental disorder described in 2018, caused by de novo pathogenic variants in the CHD3 gene. This syndrome is characterized by global developmental delay, speech delay, intellectual disability, hypotonia and behavioral disorders including autism spectrum disorder (ASD). Typical dysmorphic features include macrocephaly, hypertelorism, enophthalmia, sparse eyebrows, bulging forehead, midface hypoplasia, prominent nose and pointed chin. To our knowledge, there have been no other clinical descriptions of patients since the initial publication. We report the clinical description of a 21-year-old patient harboring a pathogenic de novo variant in CHD3. We reviewed the clinical features of the 35 previously reported patients. Main features were severe intellectual disability, dysmorphic facies, macrocephaly, cryptorchidism, pectus carinatum, severe ophthalmologic abnormalities and behavioral disorders including ASD, and a frank happy demeanor. Hypersociability, which was a noticeable clinical feature in our case, despite ASD, is an uncommon behavioral feature in syndromic intellectual disabilities. Our report supports hypersociability as a suggestive feature of CHD3-related syndrome along with developmental delay, macrocephaly and a dysmorphic facies.
Copyright © 2021 Elsevier Masson SAS. All rights reserved.

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Keywords:  CHD3; Happy demeanor; Hypersociability; Macrocephaly; SNIBCPS; Snijders blok-campeau syndrome

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Year:  2021        PMID: 33571694     DOI: 10.1016/j.ejmg.2021.104166

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


  1 in total

1.  Snijders Blok-Campeau syndrome caused by CHD3 gene mutation: a case report.

Authors:  Xi-Yong Fan
Journal:  Zhongguo Dang Dai Er Ke Za Zhi       Date:  2021 Sept 15
  1 in total

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