Literature DB >> 34751866

Neuropsychiatric Functioning in CDLS: A Detailed Phenotype and Genotype Correlation.

Paola Francesca Ajmone1, Beatrice Allegri2, Anna Cereda3, Giovanni Michelini4, Francesca Dall'Ara2, Milena Mariani5, Claudia Rigamonti2, Angelo Selicorni5, Paola Vizziello2, Maria Antonella Costantino2.   

Abstract

Behavioural phenotype and autism-related traits of 38 patients affected by Cornelia de Lange syndrome (CdLS) were assessed using a specific neuropsychiatric protocol. Subsequently,we search for possible genotype-phenotype correlations comparing individuals with NIPBL variants and patients with negative molecular results. Firstly results showed a higher percentage of subjects with normal intellectual quotient (IQ) and borderline IQ; adaptive skills were lower than expected for age in all participants. 39.5% of the sample presented with autism spectrum disorder (ASD), NIPBL mutated individuals demonstrated a worse trend in comparison with the clinical diagnosis group. non-truncating individuals displayed no ASD and better communication abilities than truncating individuals. Findings increase our awareness of the strengths and weaknesses points in CdLS individuals.
© 2021. The Author(s), under exclusive licence to Springer Science+Business Media, LLC, part of Springer Nature.

Entities:  

Keywords:  CdLS; Genotype–phenotype correlations; ID; NIPBL; Neuropsychiatric assessment

Mesh:

Substances:

Year:  2021        PMID: 34751866     DOI: 10.1007/s10803-021-05343-8

Source DB:  PubMed          Journal:  J Autism Dev Disord        ISSN: 0162-3257


  26 in total

1.  Normal language skills and normal intelligence in a child with de Lange syndrome.

Authors:  T H Cameron; D P Kelly
Journal:  J Speech Hear Disord       Date:  1988-05

2.  Predictive value of the Griffiths assessment in extremely low birthweight infants.

Authors:  J R Bowen; F L Gibson; G I Leslie; J D Arnold; P J Ma; D R Starte
Journal:  J Paediatr Child Health       Date:  1996-02       Impact factor: 1.954

3.  The prevalence and phenomenology of self-injurious and aggressive behaviour in genetic syndromes.

Authors:  K Arron; C Oliver; J Moss; K Berg; C Burbidge
Journal:  J Intellect Disabil Res       Date:  2010-10-26

4.  Longitudinal trajectories of intellectual and adaptive functioning in adolescents and adults with Williams syndrome.

Authors:  M H Fisher; M D Lense; E M Dykens
Journal:  J Intellect Disabil Res       Date:  2016-06-08

Review 5.  Behavioral and psychiatric manifestations in Cornelia de Lange syndrome.

Authors:  Marco A Grados; Mustafa H Alvi; Siddharth Srivastava
Journal:  Curr Opin Psychiatry       Date:  2017-03       Impact factor: 4.741

6.  Genotype-phenotype correlations of 39 patients with Cornelia De Lange syndrome: the Dutch experience.

Authors:  Z A Bhuiyan; M Klein; P Hammond; A van Haeringen; M M A M Mannens; I Van Berckelaer-Onnes; R C M Hennekam
Journal:  J Med Genet       Date:  2005-10-19       Impact factor: 6.318

7.  Survey of speech and language skills with prognostic indicators in 116 patients with Cornelia de Lange syndrome.

Authors:  M T Goodban
Journal:  Am J Med Genet       Date:  1993-11-15

8.  NIPBL mutational analysis in 120 individuals with Cornelia de Lange syndrome and evaluation of genotype-phenotype correlations.

Authors:  Lynette A Gillis; Jennifer McCallum; Maninder Kaur; Cheryl DeScipio; Dinah Yaeger; Allison Mariani; Antonie D Kline; Hui-hua Li; Marcella Devoto; Laird G Jackson; Ian D Krantz
Journal:  Am J Hum Genet       Date:  2004-08-18       Impact factor: 11.025

9.  The behavioural phenotype of Cornelia de Lange Syndrome: a study of 56 individuals.

Authors:  E Basile; L Villa; A Selicorni; M Molteni
Journal:  J Intellect Disabil Res       Date:  2007-09

10.  Contralesional White Matter Alterations in Patients After Hemispherotomy.

Authors:  Jennifer Gaubatz; Conrad C Prillwitz; Leon Ernst; Bastian David; Christian Hoppe; Elke Hattingen; Bernd Weber; Hartmut Vatter; Rainer Surges; Christian E Elger; Theodor Rüber
Journal:  Front Hum Neurosci       Date:  2020-07-07       Impact factor: 3.169

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