Literature DB >> 24718998

Autism traits in children and adolescents with Cornelia de Lange syndrome.

Siddharth Srivastava1, Colleen Landy-Schmitt, Bennett Clark, Antonie D Kline, Matt Specht, Marco A Grados.   

Abstract

Cornelia de Lange syndrome (CdLS) is a cohesinopathy causing delayed growth and limb deficits. Individuals with CdLS have mild to profound intellectual disability and autistic features. This study characterizes the behavioral phenotype of children with CdLS, focusing on autistic features, maladaptive behaviors, and impact of age. Children with CdLS (5-18 years) were administered normed instruments to characterize autism features (Childhood Autism Rating Scale, CARS), maladaptive behaviors (Aberrant Behavior Checklist), and adaptive skills (Vineland Adaptive Behaviors Scales). CdLS features and severity were rated with Diagnostic Criteria for CdLS. Forty-one children with CdLS (23 females, 18 males) were classified as having "no autism" (n = 7; 17.1%), "mild autism" (n = 17; 41.4%), and "severe autism" (n = 17; 41.4%), using CARS scores. Characteristic items were abnormal emotional response, stereotypies, odd object use, rigidity, lack of verbal communication, and low intellectual functioning. Verbal communication deficits and repetitive behaviors were higher compared to sensory, social cognition, and behavior abnormalities (P ≤ 0.0001). Maladaptive behaviors associated with autism traits were stereotypies (P = 0.003), hyperactivity (P = 0.01), and lethargy (P = 0.03). Activities of daily living were significantly affected; socialization adaptive skills were a relative strength. However, with advancing age, both socialization (P < 0.0001) and communication (P = 0.001) domains declined significantly. CdLS is characterized by autistic features, notably excessive repetitive behaviors and expressive language deficits. While other adaptive skills are impacted, socialization adaptive skills are less affected. Advancing age can worsen communication and socialization deficits relative to neurotypical peers.
© 2014 Wiley Periodicals, Inc.

Entities:  

Keywords:  Cornelia de Lange syndrome; autism; behavioral phenotype; communication; repetitive behaviors; social cognition

Mesh:

Substances:

Year:  2014        PMID: 24718998     DOI: 10.1002/ajmg.a.36573

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  12 in total

Review 1.  The origin and natural history of autism spectrum disorders.

Authors:  James C Harris
Journal:  Nat Neurosci       Date:  2016-10-26       Impact factor: 24.884

2.  Cornelia de Lange syndrome: Correlation of brain MRI findings with behavioral assessment.

Authors:  Tamanna R Roshan Lal; Mark A Kliewer; Thelma Lopes; Susan L Rebsamen; Julia O'Connor; Marco A Grados; Amy Kimball; Julia Clemens; Antonie D Kline
Journal:  Am J Med Genet C Semin Med Genet       Date:  2016-05-10       Impact factor: 3.908

Review 3.  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

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

Authors:  Paola Francesca Ajmone; Beatrice Allegri; Anna Cereda; Giovanni Michelini; Francesca Dall'Ara; Milena Mariani; Claudia Rigamonti; Angelo Selicorni; Paola Vizziello; Maria Antonella Costantino
Journal:  J Autism Dev Disord       Date:  2021-11-09

5.  Development, behaviour and sensory processing in Marshall-Smith syndrome and Malan syndrome: phenotype comparison in two related syndromes.

Authors:  P A Mulder; I D C van Balkom; A M Landlust; M Priolo; L A Menke; I H Acero; F S Alkuraya; P Arias; L Bernardini; E K Bijlsma; T Cole; C Coubes; I Dapia; S Davies; N Di Donato; N H Elcioglu; J A Fahrner; A Foster; N G González; I Huber; M Iascone; A-S Kaiser; A Kamath; K Kooblall; P Lapunzina; J Liebelt; S A Lynch; S M Maas; C Mammì; I B Mathijssen; S McKee; G M Mirzaa; T Montgomery; D Neubauer; T E Neumann; L Pintomalli; M A Pisanti; A S Plomp; S Price; C Salter; F Santos-Simarro; P Sarda; D Schanze; M Segovia; C Shaw-Smith; S Smithson; M Suri; K Tatton-Brown; J Tenorio; R V Thakker; R M Valdez; A Van Haeringen; J M Van Hagen; M Zenker; M Zollino; W W Dunn; S Piening; R C Hennekam
Journal:  J Intellect Disabil Res       Date:  2020-10-09

6.  Development, behavior, and biomarker characterization of Smith-Lemli-Opitz syndrome: an update.

Authors:  Audrey Thurm; Elaine Tierney; Cristan Farmer; Phebe Albert; Lisa Joseph; Susan Swedo; Simona Bianconi; Irena Bukelis; Courtney Wheeler; Geeta Sarphare; Diane Lanham; Christopher A Wassif; Forbes D Porter
Journal:  J Neurodev Disord       Date:  2016-04-05       Impact factor: 4.025

Review 7.  Role of Genetics in the Etiology of Autistic Spectrum Disorder: Towards a Hierarchical Diagnostic Strategy.

Authors:  Cyrille Robert; Laurent Pasquier; David Cohen; Mélanie Fradin; Roberto Canitano; Léna Damaj; Sylvie Odent; Sylvie Tordjman
Journal:  Int J Mol Sci       Date:  2017-03-12       Impact factor: 5.923

8.  Age-related Behavioural Change in Cornelia de Lange and Cri du Chat Syndromes: A Seven Year Follow-up Study.

Authors:  Lisa Cochran; Alice Welham; Chris Oliver; Adam Arshad; Joanna F Moss
Journal:  J Autism Dev Disord       Date:  2019-06

9.  Behavioral Phenotype and Autism Spectrum Disorders in Cornelia de Lange Syndrome.

Authors:  Lucia Parisi; Teresa Di Filippo; Michele Roccella
Journal:  Ment Illn       Date:  2015-09-30

10.  Behavioural and psychological characteristics in Pitt-Hopkins syndrome: a comparison with Angelman and Cornelia de Lange syndromes.

Authors:  Alice Watkins; Stacey Bissell; Jo Moss; Chris Oliver; Jill Clayton-Smith; Lorraine Haye; Mary Heald; Alice Welham
Journal:  J Neurodev Disord       Date:  2019-10-05       Impact factor: 4.025

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