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Validation of Hagerman’s behavioral phenotype for fragile X syndrome among men with intellectual disability

Validation of Hagerman’s behavioral phenotype for fragile X syndrome among men with intellectual... The behavioral phenotype of fragile X syndrome (FXS) and intellectual disability (ID) proposed by Hagerman et al. (2009) was primarily based on data from male children and teens. The purpose of this study was to promote a better understanding of how this condition manifests in adults.Design/methodology/approachA total of 18 men of FXS were paired with men with Down syndrome on the basis of age and level of ID. A screening checklist was created on the basis of existing scales and the Hagerman et al. (2009) behavioral phenotype and completed by care providers.FindingsFive of the 12 features of the phenotype were significantly more present among men with FXS than in men with Down syndrome.Originality/valueThis study provides partial confirmation for Hagerman et al.’s (2009) behavioral phenotype of FXS among men with moderate ID and identified some traits that warrant further investigation. http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Advances in Mental Health and Intellectual Disabilities Emerald Publishing

Validation of Hagerman’s behavioral phenotype for fragile X syndrome among men with intellectual disability

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Publisher
Emerald Publishing
Copyright
© Emerald Publishing Limited
ISSN
2044-1282
DOI
10.1108/amhid-09-2020-0020
Publisher site
See Article on Publisher Site

Abstract

The behavioral phenotype of fragile X syndrome (FXS) and intellectual disability (ID) proposed by Hagerman et al. (2009) was primarily based on data from male children and teens. The purpose of this study was to promote a better understanding of how this condition manifests in adults.Design/methodology/approachA total of 18 men of FXS were paired with men with Down syndrome on the basis of age and level of ID. A screening checklist was created on the basis of existing scales and the Hagerman et al. (2009) behavioral phenotype and completed by care providers.FindingsFive of the 12 features of the phenotype were significantly more present among men with FXS than in men with Down syndrome.Originality/valueThis study provides partial confirmation for Hagerman et al.’s (2009) behavioral phenotype of FXS among men with moderate ID and identified some traits that warrant further investigation.

Journal

Advances in Mental Health and Intellectual DisabilitiesEmerald Publishing

Published: Jul 26, 2021

Keywords: Men; Intellectual disability; Down syndrome; Fragile X syndrome; Autistic symptom; Behavioral phenotype

References