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Which Score for What? Operationalizing Standardized Cognitive Test Performance for the Assessment of Change
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Cristan Farmer; Audrey Thurm; Tanvi Das; E. Martina Bebin; Jonathan A. Bernstein; Elizabeth Berry-Kravis; Joseph D. Buxbaum; Charis Eng; Thomas Frazier; Antonio Y. Hardan; Alexander Kolevzon; Darcy A. Krueger; Julian A. Martinez-Agosto; Hope Northrup; Craig M. Powell; Latha Valluripalli Soorya; Joyce Y. Wu; Mustafa Sahin – American Journal on Intellectual and Developmental Disabilities, 2025
Developmental domains, such as cognitive, language, and motor, are key concepts of interest in longitudinal studies of intellectual and developmental disabilities (IDD). Normative scores (e.g., IQ) are often used to operationalize performance on standardized tests of these concepts, but it is the interval-distributed person-ability scores that are…
Descriptors: Cognitive Tests, Intelligence Tests, Cognitive Ability, Intellectual Disability
Measurement of Sleep Behaviors in Chromosome 15q11.2-13.1 Duplication (Dup15q Syndrome)
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Barstein, Jamie; Jeste, Shafali; Saravanapandian, Vidya; Hyde, Carly; Distefano, Charlotte – American Journal on Intellectual and Developmental Disabilities, 2021
Duplication of chromosome 15q11.2-q13.1 (dup15q syndrome) results in hypotonia, intellectual disability (ID), and autism symptomatology. Clinical electroencephalography has shown abnormal sleep physiology, but no studies have characterized sleep behaviors. The present study used the Children's Sleep Habits Questionnaire (CSHQ) in 42 people with…
Descriptors: Genetic Disorders, Sleep, Children, Cognitive Ability
Block Design Performance in Williams Syndrome: Visuospatial Abilities or Task Approach Skills?
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Block, Elisa; Farran, Emily K.; Van Herwegen, Jo – American Journal on Intellectual and Developmental Disabilities, 2022
The block design task (BDT) is a visuospatial measure that individuals with Williams syndrome (WS) perform poorly on. However, it is unclear what underlies their impaired performance. This study investigated whether poorer performance is a result of visuospatial difficulties, executive function (EF) difficulties, atypical looking strategies, or a…
Descriptors: Task Analysis, Visual Perception, Spatial Ability, Executive Function
Attention/Deficit Hyperactivity Disorder in Adolescent and Young Adult Males with Fragile X Syndrome
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Klusek, Jessica; O'Connor, Shannon L.; Hickey, Alexandra; Hills, Kimberly J.; Abbeduto, Leonard; Roberts, Jane E. – American Journal on Intellectual and Developmental Disabilities, 2022
This study characterized the rates of attention-deficit/hyperactivity disorder (ADHD) in adolescent and young adult males with fragile X syndrome (FXS) using a multi-method approach integrating a DSM-based parent interview (Children's Interview for Psychiatric Syndromes; P-ChIPS, Fristad et al., 1998) and a parent rating scale (Child Behavior…
Descriptors: Attention Deficit Hyperactivity Disorder, Adolescents, Young Adults, Males
Daily Living Skills in Adolescent and Young Adult Males with Fragile X Syndrome
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Thurman, Angela John; Swinehart, Stephanie Summers; Klusek, Jessica; Roberts, Jane E.; Bullard, Lauren; Marzan, Jocelyn Christina B.; Brown, W. Ted; Abbeduto, Leonard – American Journal on Intellectual and Developmental Disabilities, 2022
By adulthood, most males with fragile X syndrome (FXS) require support to navigate day-to-day settings. The present study cross-sectionally: (1) characterized the profile of daily living skills in males with FXS and (2) examined associated participant characteristics (i.e., fragile X mental retardation protein [FMRP] expression, nonverbal…
Descriptors: Genetic Disorders, Daily Living Skills, Males, Adolescents
Neurogenesis, Myelination, and Circuitry: The Case for a Distributed Therapeutic Regimen in Down Syndrome
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Haydar, Tarik F. – American Journal on Intellectual and Developmental Disabilities, 2020
One of the overriding hopes of the Down syndrome (DS) research community is to arrive at a better understanding of how trisomy 21 affects brain development and function, and that doing so will improve quality of life and independence for people with DS. In searching for the underlying causes of intellectual disability in DS, researchers and…
Descriptors: Down Syndrome, Medical Research, Brain Hemisphere Functions, Genetic Disorders
Examination of the ADOS-2 Expressive Language Score in Fragile X Syndrome
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Fielding-Gebhardt, Heather; Bredin-Oja, Shelley L.; Warren, Steven F. – American Journal on Intellectual and Developmental Disabilities, 2021
The development of an expressive language score for people with autism based on the ADOS-2 was recently reported by Mazurek et al. (2019). The current study examined the construct validity of the ADOS-2 expressive language score (ELS) in a sample of adolescents with fragile X syndrome (n = 45, 10 girls), a neurodevelopmental disorder with high…
Descriptors: Autism, Pervasive Developmental Disorders, Expressive Language, Genetic Disorders
Executive Functions and Prader-Willi Syndrome: Global Deficit Linked with Intellectual Level and Syndrome-Specific Associations
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Chevalère, Johann; Postal, Virginie; Jauregui, Joseba; Copet, Pierre; Laurier, Virginie; Thuilleaux, Denise – American Journal on Intellectual and Developmental Disabilities, 2015
The aim of this study was to support the growing evidence suggesting that Prader-Willi Syndrome (PWS) might present with an impairment of executive functions (EFs) and to investigate whether this impairment is specific to patients with PWS or due to their intellectual disability (ID). Six tasks were administered to assess EFs (inhibition,…
Descriptors: Genetic Disorders, Disabilities, Executive Function, Neurological Impairments
Individuals with 22q11.2 Deletion Syndrome Are Impaired at Explicit, but Not Implicit, Discrimination of Local Forms Embedded in Global Structures
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Giersch, Anne; Glaser, Bronwyn; Pasca, Catherine; Chabloz, Mélanie; Debbané, Martin; Eliez, Stephan – American Journal on Intellectual and Developmental Disabilities, 2014
Individuals with 22q11.2 deletion syndrome (22q11.2DS) are impaired at exploring visual information in space; however, not much is known about visual form discrimination in the syndrome. Thirty-five individuals with 22q11.2DS and 41 controls completed a form discrimination task with global forms made up of local elements. Affected individuals…
Descriptors: Genetic Disorders, Cognitive Ability, Visual Perception, Visual Discrimination
Parent and Teacher Perspectives about Problem Behavior in Children with Williams Syndrome
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Klein-Tasman, Bonita P.; Lira, Ernesto N.; Li-Barber, Kirsten T.; Gallo, Frank J.; Brei, Natalie G. – American Journal on Intellectual and Developmental Disabilities, 2015
Problem behavior of 52 children with Williams syndrome ages 6 to 17 years old was examined based on both parent and teacher report. Generally good inter-rater agreement was found. Common areas of problem behavior based both on parent and teacher report included attention problems, anxiety difficulties, repetitive behaviors (e.g., obsessions,…
Descriptors: Behavior Problems, Genetic Disorders, Children, Adolescents
Social Cognition in Adolescent Girls with Fragile X Syndrome
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Turkstra, Lyn S.; Abbeduto, Leonard; Meulenbroek, Peter – American Journal on Intellectual and Developmental Disabilities, 2014
This study aimed to characterize social cognition, executive functions (EFs), and everyday social functioning in adolescent girls with fragile X syndrome, and identify relationships among these variables. Participants were 20 girls with FXS and 20 age-matched typically developing peers. Results showed significant between-groups differences in…
Descriptors: Executive Function, Social Cognition, Interpersonal Competence, Females