Hostname: page-component-8448b6f56d-t5pn6 Total loading time: 0 Render date: 2024-04-25T00:41:40.793Z Has data issue: false hasContentIssue false

Behavioral profiles of children with infantile nephropathic cystinosis

Published online by Cambridge University Press:  12 May 2005

Gustavo Delgado
Affiliation:
Southern California Permanente Medical Group, USA.
Amy Schatz
Affiliation:
Department of Neurosciences, School of Medicine, USA.
Sharon Nichols
Affiliation:
Department of Neurosciences, School of Medicine, USA.
Mark Appelbaum
Affiliation:
Department of Psychology, University of California, San Diego, School of Medicine, La Jolla, USA.
Doris Trauner
Affiliation:
Departments of Neurosciences and Pediatrics, University of California, San Diego, School of Medicine, La Jolla, USA.
Get access

Abstract

Children with infantile nephropathic cystinosis have evidence of visuospatial and arithmetic deficits on a background of normal intellectual and verbal skills. This study aimed to define further their behavioral phenotype. The Achenbach Child Behavior Checklist was completed by parents of: 64 children and adolescents with cystinosis (33 females, 31 males; mean age 8y 8mo, range 4 to 16y, SD 2y 11mo); 101 healthy controls (47 females, 54 males; mean age 8y 4mo, range 4 to 16y, SD2y 11mo); 21 children and adolescents with cystic fibrosis (CF), termed chronic-disease controls (9 females, 12 males; mean age 11y 3mo, age range 4 to 17y, SD3y 5mo). Compared with healthy controls, individuals with cystinosis had evidence of a significantly higher incidence of behavioral problems, including social problems, somatic complaints, and attention problems. Compared with the chronic-disease control group, the cystinosis group differed only on the Social Problems scale, with 22% of participants with cystinosis scoring in the ‘at risk’ range whereas no participant with CF received an elevated score on this scale. We conclude that children and adolescents with cystinosis have evidence of a significant incidence of social difficulties compared with individuals with another chronic illness and healthy participants. The combination of visuospatial problems, difficulty with arithmetic, attention problems, and social difficulties seen in the cystinosis group constitutes a behavioral phenotype of this genetic disorder. This cluster of cognitive and behavioral symptoms is also seen in the nonverbal learning disabilities syndrome, and suggests a possible early difference in brain development in children with cystinosis compared with children who do not share this genetic disorder.

Type
Original Articles
Copyright
© 2005 Mac Keith Press

Access options

Get access to the full version of this content by using one of the access options below. (Log in options will check for institutional or personal access. Content may require purchase if you do not have access.)