Scott S Hall1, Amy A Lightbody, Melissa Hirt, Ava Rezvani, Allan L Reiss. 1. Center for Interdisciplinary Brain Sciences Research, Department of Psychiatry and Behavioral Sciences, 401 Quarry Road, Stanford University, Stanford, CA 94305-5795, USA. hallss@stanford.edu
Abstract
OBJECTIVE: Many investigators now routinely classify children with fragile X syndrome (FXS) according to whether or not they also meet diagnostic criteria for autism. To determine whether this classification is appropriate, we examined the profiles of autistic behaviors shown by boys and girls with FXS. METHOD: Individuals with FXS, aged 5 to 25 years, were assessed on two established measures of autism, the Social Communication Questionnaire (SCQ) and the Autism Diagnostic Observation Schedule (ADOS). RESULTS: We found that 35.1% of boys and 4.3% of girls with FXS scored in the "autism" category on both instruments. Analysis of the symptom profile indicated that both boys and girls with FXS showed lower rates of impairment on communication and reciprocal social interaction items than the reference autism samples on the measures. Furthermore, a regression model showed that IQ was significantly negatively associated with the SCQ total score in both boys and girls with FXS, when controlling for age, medication use, and FMRP levels. CONCLUSIONS: These data suggest that there are significant differences in the profile of social and communicative symptomatology in FXS compared with individuals diagnosed with idiopathic autism. Given these differences, the implementation of standard autism interventions for individuals with FXS may not be optimal. Maintaining the conceptual distinction between FXS (an established biological disease) and idiopathic autism (a phenomenologically defined behavioral disorder) may also facilitate the development of more targeted and thus effective interventions for individuals with FXS in the future. 2010 American Academy of Child and Adolescent Psychiatry. Published by Elsevier Inc. All rights reserved.
OBJECTIVE: Many investigators now routinely classify children with fragile X syndrome (FXS) according to whether or not they also meet diagnostic criteria for autism. To determine whether this classification is appropriate, we examined the profiles of autistic behaviors shown by boys and girls with FXS. METHOD: Individuals with FXS, aged 5 to 25 years, were assessed on two established measures of autism, the Social Communication Questionnaire (SCQ) and the Autism Diagnostic Observation Schedule (ADOS). RESULTS: We found that 35.1% of boys and 4.3% of girls with FXS scored in the "autism" category on both instruments. Analysis of the symptom profile indicated that both boys and girls with FXS showed lower rates of impairment on communication and reciprocal social interaction items than the reference autism samples on the measures. Furthermore, a regression model showed that IQ was significantly negatively associated with the SCQ total score in both boys and girls with FXS, when controlling for age, medication use, and FMRP levels. CONCLUSIONS: These data suggest that there are significant differences in the profile of social and communicative symptomatology in FXS compared with individuals diagnosed with idiopathic autism. Given these differences, the implementation of standard autism interventions for individuals with FXS may not be optimal. Maintaining the conceptual distinction between FXS (an established biological disease) and idiopathic autism (a phenomenologically defined behavioral disorder) may also facilitate the development of more targeted and thus effective interventions for individuals with FXS in the future. 2010 American Academy of Child and Adolescent Psychiatry. Published by Elsevier Inc. All rights reserved.
Authors: Maria Johansson; Maria Råstam; Eva Billstedt; Susanna Danielsson; Kerstin Strömland; Marilyn Miller; Christopher Gillberg Journal: Dev Med Child Neurol Date: 2006-01 Impact factor: 5.449
Authors: Walter E Kaufmann; Ranon Cortell; Alice S M Kau; Irena Bukelis; Elaine Tierney; Robert M Gray; Christiane Cox; George T Capone; Pia Stanard Journal: Am J Med Genet A Date: 2004-09-01 Impact factor: 2.802
Authors: R Nick Hernandez; Rachel L Feinberg; Rebecca Vaurio; Natalie M Passanante; Richard E Thompson; Walter E Kaufmann Journal: Am J Med Genet A Date: 2009-06 Impact factor: 2.802
Authors: Heather Cody Hazlett; Michele D Poe; Amy A Lightbody; Guido Gerig; James R Macfall; Allison K Ross; James Provenzale; Arianna Martin; Allan L Reiss; Joseph Piven Journal: J Neurodev Disord Date: 2009-03 Impact factor: 4.025
Authors: Fumiko Hoeft; Elizabeth Walter; Amy A Lightbody; Heather C Hazlett; Catie Chang; Joseph Piven; Allan L Reiss Journal: Arch Gen Psychiatry Date: 2010-11-01
Authors: David P Benjamin; Ann M Mastergeorge; Andrea S McDuffie; Sara T Kover; Randi J Hagerman; Leonard Abbeduto Journal: Res Dev Disabil Date: 2014-07-23