Advertisement

Journal of Autism and Developmental Disorders

, Volume 45, Issue 6, pp 1836–1844 | Cite as

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

  • Kristin M. Hustyi
  • Scott S. HallEmail author
  • Eve-Marie Quintin
  • Lindsay C. Chromik
  • Amy A. Lightbody
  • Allan L. Reiss
Original Paper

Abstract

Few studies have examined the relationship between autistic symptomatology and competence in independent living skills in adolescents and young adults with fragile X syndrome (FXS). In this study, 70 individuals with FXS, aged 15–25 years, and 35 matched controls were administered direct measures of independent living skills and autistic symptomatology. Results showed that higher levels of autistic symptomatology were associated with lower levels of competence in independent living skills in individuals with FXS, but not in controls. These data indicated that the relationship between autistic symptomatology and independent living skills was syndrome-specific. Early intervention strategies that address autistic symptomatology are sorely needed to improve functional outcomes in this population.

Keywords

Fragile X syndrome Functional skills Independent living skills Autistic symptomatology 

Notes

Acknowledgments

This study was supported by Grant Numbers MH50047 (PI Allan Reiss) and K08MH081998 (PI Scott Hall) from the National Institute of Mental Health, a gift from the Canel Family, and the Fonds de Recherche Société et Culture Québec (Postdoctoral Grant to Eve-Marie Quintin).

References

  1. Bailey, D. B., Mesibov, G. B., Hatton, D. D., Clark, R. D., Roberts, J. E., & Mayhew, L. (1998). Autistic behavior in young boys with fragile X syndrome. Journal of Autism and Developmental Disorders, 28(6), 499–508.CrossRefPubMedGoogle Scholar
  2. Bailey, D. B., Raspa, M., Holiday, D., Bishop, E., & Olmsted, M. (2009). Functional skills of individuals with fragile X syndrome: A lifespan cross-sectional analysis. American Journal on Intellectual and Developmental Disabilities, 114(4), 289–303.CrossRefPubMedGoogle Scholar
  3. Brock, M., & Hatton, D. (2010). Distinguishing features of autism in boys with fragile X syndrome. Journal of Intellectual Disability Research, 54(10), 894–905.CrossRefPubMedGoogle Scholar
  4. Clifford, S., Dissanayake, C., Bui, Q. M., Huggins, R., Taylor, A. K., & Loesch, D. Z. (2007). Autism spectrum phenotype in males and females with fragile X full mutation and premutation. Journal of Autism and Developmental Disorders, 37(4), 738–747.CrossRefPubMedGoogle Scholar
  5. Coppus, A. M. (2013). People with intellectual disability: What do we know about adulthood and life expectancy? Developmental Disabilities Research Reviews, 18(1), 6–16.CrossRefPubMedGoogle Scholar
  6. Cornish, K., Cole, V., Longhi, E., Karmiloff-Smith, A., & Scerif, G. (2012). Does attention constrain developmental trajectories in fragile X syndrome? A 3-year prospective longitudinal study. American Journal on Intellectual and Developmental Disabilities, 117(2), 103–120.CrossRefPubMedGoogle Scholar
  7. Demark, J. L., Feldman, M. A., & Holden, J. J. (2003). Behavioral relationship between autism and fragile X syndrome. American Journal on Mental Retardation, 108(5), 314–326.CrossRefPubMedGoogle Scholar
  8. Hagerman, R. J., Jackson, A. W., Levitas, A., Rimland, B., & Braden, M. (1986). An analysis of autism in fifty males with the fragile X syndrome. American Journal of Medical Genetics, 23(1–2), 359–374.CrossRefPubMedGoogle Scholar
  9. Hall, S. S. (2009). Treatments for fragile X syndrome: A closer look at the data. Developmental Disabilities Research Reviews, 15, 353–360.CrossRefPubMedCentralPubMedGoogle Scholar
  10. Hall, S. S., Lightbody, A. A., Hirt, M., Rezvani, A., & Reiss, A. L. (2010). Autism in fragile X syndrome: A category mistake? Journal of the American Academy of Child and Adolescent Psychiatry, 49(9), 921–933.CrossRefPubMedCentralPubMedGoogle Scholar
  11. Hall, S. S., Lightbody, A. A., & Reiss, A. L. (2008). Compulsive, self-injurious, and autistic behavior in children and adolescents with fragile X syndrome. American Journal on Mental Retardation, 113(1), 44–53.CrossRefPubMedGoogle Scholar
  12. Harris, S. W., Hessl, D., Goodlin-Jones, B., Ferranti, J., Bacalman, S., Barbato, I., et al. (2008). Autism profiles of males with fragile X syndrome. American Journal on Mental Retardation, 113(6), 427–438.CrossRefPubMedCentralPubMedGoogle Scholar
  13. Hartley, S. L., Seltzer, M. M., Raspa, M., Olmstead, M., Bishop, E., & Bailey, D. B. (2011). Exploring the adult life of men and women with fragile X syndrome: Results from a national survey. American Journal on Intellectual and Developmental Disabilities, 116(1), 16–35.CrossRefPubMedCentralPubMedGoogle Scholar
  14. Hatton, D. D., Sideris, J., Skinner, M., Mankowski, J., Bailey, D. B, Jr, Roberts, J., & Mirrett, P. (2006). Autistic behavior in children with fragile X syndrome: Prevalence, stability, and the impact of FMRP. American Journal of Medical Genetics. Part A, 140A(17), 1804–1813.CrossRefPubMedGoogle Scholar
  15. Hernandez, R. N., Feinberg, R. L., Vaurio, R., Passanante, N. M., Thompson, R. E., & Kaufmann, W. E. (2009). Autism spectrum disorder in fragile X syndrome: A longitudinal evaluation. American Journal of Medical Genetics. Part A, 149A(6), 1125–1137.CrossRefPubMedCentralPubMedGoogle Scholar
  16. Hessl, D., Dyer-Friedman, J., Glaser, B., Wisbeck, J., Barajas, R. G., Taylor, A., et al. (2001). The influence of environmental and genetic factors on behavior problems and autistic symptoms in boys and girls with fragile X syndrome. Pediatrics, 108(5), E88.CrossRefPubMedGoogle Scholar
  17. Howlin, P., Goode, S., Hutton, J., & Rutter, M. (2004). Adult outcome for children with autism. Journal of Child Psychology and Psychiatry and Allied Disciplines, 45(2), 212–229.CrossRefGoogle Scholar
  18. Karagiozis, H., Gray, S., Sacco, J., Shapiro, M., & Kawas, C. (1998). The direct assessment of functional abilities (DAFA): A comparison to an indirect measure of instrumental activities of daily living. Gerontologist, 38(1), 113–121.CrossRefPubMedGoogle Scholar
  19. Katz, S. (1983). Assessing self-maintenance: Activities of daily living, mobility, and instrumental activities of daily living. Journal of the American Geriatrics Society, 31(12), 721–727.CrossRefPubMedGoogle Scholar
  20. Kau, A. S., Tierney, E., Bukelis, I., Stump, M. H., Kates, W. R., Trescher, W. H., et al. (2004). Social behavior profile in young males with fragile X syndrome: Characteristics and specificity. American Journal of Medical Genetics. Part A, 126A(1), 9–17.CrossRefPubMedGoogle Scholar
  21. Kaufmann, W. E., Cortell, R., Kau, A. S., Bukelis, I., Tierney, E., Gray, R. M., et al. (2004). Autism spectrum disorder in fragile X syndrome: Communication, social interaction, and specific behaviors. American Journal of Medical Genetics. Part A, 129A(3), 225–234.CrossRefPubMedGoogle Scholar
  22. Klaiman, C., Quintin, E., Jo, B., Lightbody, A. A., Hazlett, H. C., Piven, J., et al. (2014). Longitudinal profiles of adaptive behavior in fragile X syndrome. Pediatrics, 134(2), 315–324.CrossRefPubMedGoogle Scholar
  23. Loeb, P. A. (1996). Independent living scales manual. San Antonio, TX: Psychological Corporation.Google Scholar
  24. Loewenstein, D. A., Amigo, E., Duara, R., Guterman, A., Hurwitz, D., Berkowitz, N., et al. (1989). A new scale for the assessment of functional status in Alzheimer’s disease and related disorders. Journal of Gerontology, 44(4), 114–121.CrossRefGoogle Scholar
  25. Lord, C., Risi, S., Lambrecht, L., Cook, E. H, Jr, Leventhal, B. L., DiLavore, P. C., et al. (2000). The autism diagnostic observation schedule-generic: A standard measure of social and communication deficits associated with the spectrum of autism. Journal of Autism and Developmental Disorders, 30(3), 205–223.CrossRefPubMedGoogle Scholar
  26. McCary, L. M., & Roberts, J. E. (2013). Early identification of autism in fragile X syndrome: A review. Journal of Intellectual Disability Research, 57(9), 803–814.CrossRefPubMedCentralPubMedGoogle Scholar
  27. McDuffie, A., Abbeduto, L., Lewis, P., Kover, S., Kim, J. S., Weber, A., et al. (2010). Autism spectrum disorder in children and adolescents with fragile X syndrome: Within-syndrome differences and age-related changes. American Journal on Intellectual and Developmental Disabilities, 115(4), 307–326.CrossRefPubMedCentralPubMedGoogle Scholar
  28. Moss, J., Oliver, C., Nelson, L., Richards, C., & Hall, S. (2013). Delineating the profile of autism spectrum disorder characteristics in Cornelia de Lange and Fragile X syndromes. American Journal on Intellectual and Developmental Disabilities, 118(1), 55–73.CrossRefPubMedGoogle Scholar
  29. Partington, M. W., Robinson, H., Laing, S., & Turner, G. (1992). Mortality in the fragile X syndrome: Preliminary data. American Journal of Medical Genetics, 43(1–2), 120–123.CrossRefPubMedGoogle Scholar
  30. Reuben, D. B., Valle, L. A., Hays, R. D., & Siu, A. L. (1995). Measuring physical function in community-dwelling older persons: a comparison of self-administered, interviewer-administered, and performance-based measures. Journal of the American Geriatrics Society, 43(1), 17–23.CrossRefPubMedGoogle Scholar
  31. Revhiem, N., & Medalia, A. (2004). The independent living scales as a measure of functional outcome for schizophrenia. Psychiatric Services, 55, 1052–1054.CrossRefGoogle Scholar
  32. Rogers, S. J., Wehner, D. E., & Hagerman, R. (2001). The behavioral phenotype in fragile X: Symptoms of autism in very young children with fragile X syndrome, idiopathic autism, and other developmental disorders. Journal of Developmental and Behavioral Pediatrics, 22(6), 409–417.CrossRefPubMedGoogle Scholar
  33. Rozzini, R., Frisoni, G. B., Bianchetti, A., Zanetti, O., & Trabucchi, M. (1993). Physical performance test and activities of daily living scales in the assessment of health status in elderly people. Journal of the American Geriatrics Society, 41(10), 1109–1113.CrossRefPubMedGoogle Scholar
  34. Sabaratnam, M. (2000). Pathological and neuropathological findings in two males with fragile-X syndrome. Journal of Intellectual Disability Research, 44(1), 81–85.CrossRefPubMedGoogle Scholar
  35. Seltzer, M. M., & Krauss, M. W. (1989). Aging parents with adult mentally retarded children: Family risk factors and sources of support. American Journal on Mental Retardation, 94(3), 303–312.PubMedGoogle Scholar
  36. Smith, L. E., Barker, E. T., Seltzer, M. M., Abbeduto, L., & Greenberg, J. S. (2012). Behavioral phenotype of fragile X syndrome in adolescence and adulthood. American Journal on Intellectual and Developmental Disabilities, 117, 1–17.CrossRefPubMedCentralPubMedGoogle Scholar
  37. Turner, G., Webb, T., Wake, S., & Robinson, H. (1996). Prevalence of fragile X syndrome. American Journal of Medical Genetics, 64(1), 196–197.CrossRefPubMedGoogle Scholar
  38. Wechsler, D. (1991). Wechsler intelligence scale for children (3rd ed.). San Antonio, TX: The Psychological Corporation.Google Scholar
  39. Wechsler, D. (1999). Wechsler abbreviated scale of intelligence. San Antonio, TX: The Psychological Corporation.Google Scholar
  40. Wolff, J. J., Bodfish, J. W., Hazlett, H. C., Lightbody, A. A., Reiss, A. L., & Piven, J. (2012). Evidence of a distinct behavioral phenotype in young boys with fragile X syndrome and autism. Journal of the American Academy of Child and Adolescent Psychiatry, 51(12), 1324–1332.CrossRefPubMedCentralPubMedGoogle Scholar

Copyright information

© Springer Science+Business Media New York 2014

Authors and Affiliations

  • Kristin M. Hustyi
    • 1
  • Scott S. Hall
    • 1
    Email author
  • Eve-Marie Quintin
    • 1
    • 2
  • Lindsay C. Chromik
    • 1
  • Amy A. Lightbody
    • 1
  • Allan L. Reiss
    • 1
  1. 1.Department of Psychiatry and Behavioral Sciences, Center for Interdisciplinary Brain Sciences Research (CIBSR)Stanford University School of MedicineStanfordUSA
  2. 2.Department of Educational and Counseling PsychologyMcGill UniversityMontrealCanada

Personalised recommendations