A Longitudinal Follow-Up Study of Affect in Children and Adults With Cornelia de Lange Syndrome

A Longitudinal Follow-Up Study of Affect in Children and Adults With Cornelia de Lange Syndrome

Nelson L; Moss J; Oliver C

American Journal on Intellectual and Developmental Disabilities

2014

Rehabilitation; behavior; Education & Educational Research; Cornelia de Lange syndrome; behavioral-phenotype; mental-retardation; affect; autistic; behavioral phenotypes; delange-syndrome; follow-up; fragile-x-syndrome; genetic syndromes; genotype-phenotype correlations; longitudinal; pervasive developmental disorders; spectrum disorders; williams-syndrome; behavioral problems; De Lange syndrome; Cri-du-chat; trajectory; characteristics; mood; interest; pleasure

Studies of individuals with Cornelia de Lange syndrome (CdLS) have described changes in mood and behavior with age, although no empirical or longitudinal studies have been conducted. Caregivers of individuals with CdLS (N = 67), cri du chat syndrome (CdCS; N = 42), and Fragile X syndrome (FXS; N = 142) completed the Mood, Interest and Pleasure Questionnaire (MIPQ) at Time 1 and 2 years later (Time 2). Scores on the MIPQ were significantly lower in the CdLS group compared with the CdCS and FXS groups at Time 1 and Time 2. Lower MIPQ scores were characteristic of older adolescents (> 15 years) and adults with CdLS. However, there were no significant differences in MIPQ scores between Time 1 and Time 2. Age and insistence on sameness predicted MIPQ scores in CdLS.

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