The Relationship Between Autistic Symptomatology and Independent Living Skills in Adolescents and Young Adults with Fragile X Syndrome

Hustyi, Kristin M.; Hall, Scott S.; Quintin, Eve-Marie; Chromik, Lindsay C.; Reiss, Allan L.

doi:10.1007/s10803-014-2342-0

Cited by 18 publications

(13 citation statements)

References 36 publications

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“…Adolescents and young adults with more autism symptoms had lower independent living skills, especially managing money, health and safety, and problem solving, relative to typically developing peers even after controlling for IQ. 79 …”

Section: Resultsmentioning

confidence: 99%

Public Health Literature Review of Fragile X Syndrome

2017

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OBJECTIVES The purpose of this systematic literature review is to describe what is known about fragile X syndrome (FXS) and to identify research gaps. The results can be used to help inform future public health research and provide pediatricians with up-to-date information about the implications of the condition for individuals and their families. METHODS An electronic literature search was conducted, guided by a variety of key words. The search focused on 4 areas of both clinical and public health importance: (1) the full mutation phenotype, (2) developmental trajectories across the life span, (3) available interventions and treatments, and (4) impact on the family. A total of 661 articles were examined and 203 were included in the review. RESULTS The information is presented in the following categories: developmental profile (cognition, language, functional skills, and transition to adulthood), social-emotional profile (cooccurring psychiatric conditions and behavior problems), medical profile (physical features, seizures, sleep, health problems, and physiologic features), treatment and interventions (educational/behavioral, allied health services, and pharmacologic), and impact on the family (family environment and financial impact). Research gaps also are presented. CONCLUSIONS The identification and treatment of FXS remains an important public health and clinical concern. The information presented in this article provides a more robust understanding of FXS and the impact of this complex condition for pediatricians. Despite a wealth of information about the condition, much work remains to fully support affected individuals and their families.

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Section: Resultsmentioning

confidence: 99%

Public Health Literature Review of Fragile X Syndrome

2017

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“…Thus, it is difficult to generalize our findings to males with FXS. Future studies may include males with FXS and corresponding male control groups (see Hustyi et al, 2015). …”

Section: Discussionmentioning

confidence: 99%

The Role of Executive Function in Independent Living Skills in Female Adolescents and Young Adults With Fragile X Syndrome

Martin

Quintin

Hall

et al. 2016

American Journal on Intellectual and Developmental Disabilities

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Fragile X syndrome (FXS) is associated with executive function (EF) and independent living skills (ILS) deficits. We examined the role of childhood EF in ILS during adolescence/early adulthood in females with FXS and two comparison groups in the same age range (matched for IQ [IQ/Age group] and with another genetic condition, Turner syndrome [TS group]). EF and ILS were significantly higher for the FXS group than the IQ/Age group but did not differ from the TS group. For the FXS group, age and EF were significant predictors of ILS during adolescence/early adulthood, but there were no statistically significant longitudinal associations between EF and ILS. Our findings suggest that impairments in EF may have a significant effect on ILS in FXS.

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“…Consistent across studies is the negative impact of autism symptomology on adaptive behavior skill development in children with FXS with autism symptoms exerting an increasingly negative impact over time (Cohen, 1995; Hahn et al, 2015; Hatton et al, 2003; Hustyi et al, 2014; Rogers et al, 2001). Likewise, consistency in the effect of sex is present across multiple studies with girls with FXS exhibiting less severe impairments in their adaptive behavior development and a faster rate of growth than their chronologically age matched, but not mental age matched, male counterparts (Hahn et al, 2015; Hatton et al, 2003; Klaiman et al, 2015).…”

Section: Introductionmentioning

confidence: 88%

Adaptive skill trajectories in infants with fragile X syndrome contrasted to typical controls and infants at high risk for autism

Caravella

Roberts

2017

Research in Autism Spectrum Disorders

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Background Adaptive behaviors are essential for optimal outcomes and independence in individuals with developmental disabilities. This study examined longitudinal trajectories of adaptive behavior in infants with fragile X syndrome (FXS), compared to typical development (TD) and infant siblings of children diagnosed with autism (ASIBs). Method Participants included 76 male infants (FXS =25, ASIBs=27, TD = 24) assessed up to 4 times between 6 and 24 months of age for a total of 215 assessments of adaptive behavior. A sample of 12 females with FXS was included for a comparative sex analysis. Results Results indicate that infant males with FXS displayed lower initial adaptive behavior across all domains that emerged by 9 months-of-age with slower growth rates than both comparison groups. A flat profile across the domains at 24 months was evident. Increased severity of autism symptoms was related to reduced adaptive skills at 24 months-of-age. Females with FXS displayed higher scores than males on the Socialization and Motor domains and equivalent scores on the Communication and Daily Living domains at 9 months-of-age with different rates of growth across domains. Conclusions This is the first study to provide evidence of etiological specificity in adaptive behavior profiles during infancy across two populations at high risk for ASD. These findings support targeted adaptive behavior interventions in young children with FXS beginning as early at 9 months of age to reduce identified deficits and the cascading impacts of these early impairments.

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The Relationship Between Autistic Symptomatology and Independent Living Skills in Adolescents and Young Adults with Fragile X Syndrome

Cited by 18 publications

References 36 publications

Public Health Literature Review of Fragile X Syndrome

Public Health Literature Review of Fragile X Syndrome

The Role of Executive Function in Independent Living Skills in Female Adolescents and Young Adults With Fragile X Syndrome

Adaptive skill trajectories in infants with fragile X syndrome contrasted to typical controls and infants at high risk for autism

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