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Direct linkBlock, 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
Britton, Tobias C.; Wilkinson, Ellen H.; Hall, Scott S. – American Journal on Intellectual and Developmental Disabilities, 2020
Limited information is available concerning the specificity of the forms and functions of aggressive behavior exhibited by boys with fragile X syndrome (FXS). To investigate these relationships, we conducted indirect functional assessments of aggressive behavior exhibited by 41 adolescent boys with FXS and 59 age and symptom-matched controls with…
Descriptors: Aggression, Males, Genetic Disorders, Adolescents
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
Adams, Dawn; Clarke, Samantha; Griffith, Gemma; Howlin, Pat; Moss, Jo; Petty, Jane; Tunnicliffe, Penny; Oliver, Chris – American Journal on Intellectual and Developmental Disabilities, 2018
It is well documented that mothers of children with challenging behavior (CB) experience elevated levels of stress and that this persists over time, but less is known about the experience of mothers of children with rare genetic syndromes. This article describes 2 studies, 1 cross-sectional and 1 longitudinal, comparing well-being in mothers of…
Descriptors: Mothers, Stress Variables, Genetic Disorders, Depression (Psychology)
Raspa, Melissa; Bann, Carla M.; Gwaltney, Angela; Benke, Timothy A.; Fu, Cary; Glaze, Daniel G.; Haas, Richard; Heydemann, Peter; Jones, Mary; Kaufmann, Walter E.; Lieberman, David; Marsh, Eric; Peters, Sarika; Ryther, Robin; Standridge, Shannon; Skinner, Steven A.; Percy, Alan K.; Neul, Jeffrey L. – American Journal on Intellectual and Developmental Disabilities, 2020
Rett syndrome (RTT) is a neurodevelopmental disorder that primarily affects females. Recent work indicates the potential for disease modifying therapies. However, there remains a need to develop outcome measures for use in clinical trials. Using data from a natural history study (n = 1,075), we examined the factor structure, internal consistency,…
Descriptors: Genetic Disorders, Psychometrics, Psychomotor Skills, Physical Disabilities
Physiological Correlates of Maternal Responsivity in Mothers of Preschoolers with Fragile X Syndrome
Robinson, Ashley N.; Roberts, Jane E.; Brady, Nancy C.; McQuillin, Samuel D.; Warren, Steven F. – American Journal on Intellectual and Developmental Disabilities, 2016
The present study examined the relationship between salivary cortisol and maternal responsiveness in mothers of boys with fragile X syndrome (FXS). Maternal responsivity is strongly associated with child outcomes, and children with FXS are at risk for compromised development due to intellectual disability and problem behavior. Increased…
Descriptors: Correlation, Metabolism, Mothers, Responses
Pitts, C. Holley; Mervis, Carolyn B. – American Journal on Intellectual and Developmental Disabilities, 2016
We describe the performance of 292 4- to 17-year-olds with Williams syndrome (WS) on the Kaufman Brief Intelligence Test-2 (KBIT-2; Kaufman & Kaufman, 2004). Mean IQ Composite, Verbal standard score (SS), and Nonverbal SS were in the borderline range relative to the general population, with variability similar to the general population.…
Descriptors: Intelligence Tests, Genetic Disorders, Intelligence Quotient, Children
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
Kidd, Sharon A.; Raspa, Melissa; Clark, Renée; Usrey-Roos, Holly; Wheeler, Anne C.; Liu, Jessica A.; Wylie, Amanda; Sherman, Stephanie L. – American Journal on Intellectual and Developmental Disabilities, 2017
The objectives were to describe the demographic characteristics of children with Fragile X syndrome (FXS) and to determine predictors of attendance at Fragile X (FX) clinics. Findings from the Community Support Network (CSN) and Our Fragile X World (OFXW) samples showed that children who attended FX Clinics were mostly male, high-school aged or…
Descriptors: Genetic Disorders, Predictor Variables, Individual Characteristics, Participation
Nelson, Lisa; Moss, Jo; Oliver, Chris – American Journal on Intellectual and Developmental Disabilities, 2014
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…
Descriptors: Longitudinal Studies, Followup Studies, Children, Adults
Tonnsen, Bridgette L.; Shinkareva, Svetlana V.; Deal, Sara C.; Hatton, Deborah D.; Roberts, Jane E. – American Journal on Intellectual and Developmental Disabilities, 2013
Anxiety is among the most impairing conditions associated with Fragile X syndrome (FXS) and is putatively linked to atypical physiological arousal. However, few studies have examined this association in young children with FXS. The authors examined whether patterns of arousal and behavior during an experimental stranger approach paradigm differ…
Descriptors: Anxiety, Genetic Disorders, Physiology, Arousal Patterns
Greenberg, Jan S.; Seltzer, Marsha Mailick; Baker, Jason K.; Smith, Leann E.; Warren, Steven F.; Brady, Nancy; Hong, Jinkuk – American Journal on Intellectual and Developmental Disabilities, 2012
We examine how the family environment is associated with aspects of the Fragile X syndrome phenotype during childhood, adolescence, and adulthood. Mothers of children (n = 48), adolescents (n = 85), and adults (n = 34) with Fragile X syndrome participated in a multisite study. For children and adults with Fragile X syndrome, the presence of warmth…
Descriptors: Behavior Problems, Adolescents, Criticism, Family Environment
Elsabbagh, Mayada; Cohen, Henri; Karmiloff-Smith, Annette – American Journal on Intellectual and Developmental Disabilities, 2010
We examined auditory perception in Williams syndrome by investigating strategies used in organizing sound patterns into coherent units. In Experiment 1, we investigated the streaming of sound sequences into perceptual units, on the basis of pitch cues, in a group of children and adults with Williams syndrome compared to typical controls. We showed…
Descriptors: Cues, Mental Retardation, Auditory Perception, Genetic Disorders
Berry-Kravis, Elizabeth; Raspa, Melissa; Loggin-Hester, Lisa; Bishop, Ellen; Holiday, David; Bailey, Donald B., Jr. – American Journal on Intellectual and Developmental Disabilities, 2010
A national survey of caregivers of individuals with fragile X syndrome addressed characteristics of epilepsy and co-occurring conditions. Of the 1,394 individuals (1,090 males and 304 females) with the full mutation, 14% of males and 6% of females reported seizures. Seizures were more often partial, began between ages 4 and 10 years, and were…
Descriptors: Seizures, Caregivers, National Surveys, Disabilities
Stinton, Chris; Elison, Sarah; Howlin, Patricia – American Journal on Intellectual and Developmental Disabilities, 2010
Although many researchers have investigated emotional and behavioral difficulties in individuals with Williams syndrome, few have used standardized diagnostic assessments. We examined mental health problems in 92 adults with Williams syndrome using the Psychiatric Assessment Schedule for Adults with Developmental Disabilities--PAS-ADD (Moss,…
Descriptors: Developmental Disabilities, Mental Health, Depression (Psychology), Mental Disorders
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