Abstract
We compared the developmental status, functional abilities, and temperament of 31 young boys with fragile X syndrome (FXS) who did not have autism, matched on chronological age, gender, and race, with 31 boys with autism but no FXS. Children with autism exhibited a more variable profile of development in comparison with a relatively flat profile for children with FXS. Children with autism were significantly more delayed in social skills and were rated by observers as exhibiting a greater degree of impairment in cognitive, communication, and social skills. On temperament ratings, both groups were slower to adapt, less persistent, and more withdrawing than the reference group. Boys with FXS were rated as more active than the referent group, whereas boys with autism were rated as less intense, more distractible, having a higher threshold for response, and less rhythmic than the reference group. A smaller three-group analysis compared boys with FXS, boys with autism, and boys with both FXS and autism. Children with both autism and FXS were substantially more delayed than children with autism or FXS alone.
This is a preview of subscription content, log in via an institution to check access.
Similar content being viewed by others
REFERENCES
Bailey, D. B., & Nelson, D. (1995). The nature and consequences of fragile X syndrome. Mental Retardation and Developmental Disabilities Research Review, 1, 238-244.
Bailey, D., Simeonsson, R., Buysse, V., & Smith, T. (1993). Reliability of an index of child characteristics. Developmental Medicine and Child Neurology, 35, 806-815.
Bailey, D., Buysse, V., Simeonsson, R., Smith, T., & Keyes, L. (1995). Individual and team consensus ratings of child functioning. Developmental Medicine and Child Neurology, 37, 246-259.
Bailey, A., Phillips, W., & Rutter, M. (1996). Autism: Towards an integration of clinical, genetic, neuropsychological, and neurobiological perspectives. Journal of Child Psychology and Psychiatry, 37, 89-126.
Bailey, D., Hatton, D., & Skinner, M. (1998). Early developmental trajectories of males with fragile X syndrome. American Journal on Mental Retardation, 103, 29-39.
Bailey, D., Mesibov, G., Hatton, D., Clark, R., Roberts, J., & Mayhew, L. (1999). Autistic behavior in young boys with fragile X syndrome. Journal of Autism and Developmental Disorders, 28, 499-508.
Baumgardner, T., Reiss, A., Freund, L., & Abrams, B. (1995). Specification of the neurobehavioral phenotype in males with fragile X syndrome. Pediatrics, 45, 744-752.
Behl, D. D., & Akers, J. F. (1996). The use of the Battelle Developmental Inventory in the prediction of later development. Diagnostique, 21(4), 1-16.
Burack, J., & Volkmar, F. (1992). Development of low-and highfunctioning autistic children. Journal of Child Psychology and Psychiatry, 33, 607-616.
Buysse, V., Smith, T. M., Bailey, D. B., & Simeonsson, R. J. (1993). Consumer validation of an index characterizing the functional abilities of young children with disabilities. Journal of Early Intervention, 17, 224-238.
Carpentieri, S., & Morgan, S. (1996). Adaptive and intellectual functioning in autistic and nonautistic retarded children. Journal of Autism and Developmental Disorders, 26, 611-620.
Cohen, I. L. (1995). Behavioral profiles of autistic and nonautistic fragile X males. Developmental Brain Dysfunction, 8, 252-269.
Cook, E. (1998). Genetics of autism. Mental Retardation and Developmental Disabilities Research Reviews, 4, 113-120.
Dykens, E., Ort, S., Cohen, I., Finucane, B., Spiridigliozzi, G., Lachiewicz, A., Reiss, A., Freund, L., Hagerman, R., & O'Connor, R. (1996). Trajectories and profiles of adaptive behavior in males with fragile X syndrome: Multicenter studies. Journal of Autism and Developmental Disorders, 26, 287-301.
Dykens, E., & Volkmar, F. R. (1997). Medical conditions associated with autism. In D. J. Cohen & F. R. Volkmar (Eds.), Handbook of autism and pervasive developmental disorders (2nd ed., pp. 388-410). New York: Wiley.
Feinstein, C., & Reiss, A. L. (1998). Autism: The point of view from fragile X studies. Journal of of Autism and Developmental Disorders, 28, 393-405.
Ferrier, L., Bashir, A., Meryash, D., Johnson, J., & Wolff, P. (1991). Conversational skills of individuals with fragile X syndrome: A comparison with autism and Down syndrome. Developmental Medicine and Child Neurology, 33, 776-788.
Fisch, G. (1992). Is autism associated with the fragile X syndrome? American Journal of Medical Genetics, 43, 47-55.
Fisch, G. S., Holden, J. J., Simeonssen, R., Carpenter, N., Howard-Peebles, P. N., Maddalena, A., Sandgrund, A., Jacques, J. R., & McGann, B. (1994). Is fragile X syndrome a pervasive developmental disability? Cognitive ability and adaptive behavior in males with the full mutation. American Journal of Medical Genetics, 51, 346-352.
Freeman, B., Ritvo, E., Yokota, A., Childs, J., & Pollard, J. (1988). WISC-R and Vineland Adaptive Behavior Scale scores in autistic children. Journal of the American Academy of Child and Adolescent Psychiatry, 27, 428-429.
Hatton, D., Bailey, D., Burchinal, M., & Ferrell, K. (1997). Developmental growth curves of preschool children with vision impairments. Child Development, 68, 788-806.
Jacobson, J., & Ackerman, L. (1990). Differences in adaptive functioning among people with autism or mental retardation. Journal of Autism and Developmental Disorders, 20, 205-219.
Kerby, D., & Dawson, B. (1994). Autistic features, personality, and adaptive behavior in males with the fragile X syndrome and no autism. American Journal on Mental Retardation, 98, 455-462.
Klin, A., Volkmar, F., & Sparrow, S. (1992). Autistic social dysfunction: Some limitations of the theory of mind hypothesis. Journal of Child Psychology and Psychiatry, 33, 861-876.
McDevitt, S., & Carey, W. (1978). The measurement of temperament in three-to seven-year-old children. Journal of Child Psychology and Psychiatry, 19, 245-253.
Newborg J., Stock, J., Wnek, L., Guidubaldi, J., & Svinicki, J. (1984). The Battelle Developmental Inventory. Allen, TX: DLM/Teaching Resources.
Paul, R., Dykens, E., Leckman, J., Watson, M., Breg, W., & Cohen, D. (1987). A comparison of language characteristics of mentally retarded adults with fragile X syndrome and those with nonspecific mental retardation and autism. Journal of Autism and Developmental Disorders, 17, 454-468.
Reiss, A., & Freund, L. (1990). Fragile X syndrome, DSM-III-R, and autism. Journal of the American Academy of Child and Adolescent Psychiatry, 29, 885-891.
Rodrigue, J., Morgan, S., & Geffken, G. (1991). A comparative evaluation of adaptive behavior in children and adolescents with autism, Down syndrome, and normal development. Journal of Autism and Developmental Disorders, 21, 187-196.
Schopler, E., & Mesibov, G. B. (1988). Diagnosis and assessment in autism. New York: Plenum Press.
Schopler, E., Reichler, R., & Renner, B. (1988). The Childhood Autism Rating Scale (CARS). Los Angeles: Western Psychological Services.
Sexton, D., McLean, M., Boyd, R., Thompson, B., & McCormick, K. (1988). Criterion-related validity of a new standardized developmental measure for use with infants who are handicapped. Measurement and Evaluation in Counseling and Development, 21, 16-24.
Simeonssen, R. F., & Bailey, D. B. (1988). Essential elements of the assessment process. In T. D. Wachs & B. Sheehan (Eds.), Assessment of developmentally disabled infants and preschool children (pp. 25-41). New York: Plenum Press.
Simeonsson, R. J., Bailey, D., Smith, T., & Buysse, V. (1995). Young children with disabilities: Functional assessment by teachers. Journal of Developmental and Physical Disabilities, 7, 267-284.
Snyder, P., Lawson, S., Thompson, B., Stricklin, S., & Sexton, D. (1993). Evaluating the psychometric integrity of instruments used in early intervention research: The Battelle Developmental Inventory. Topics in Early Childhood Special Education, 13, 216-232.
Sponheim, E. (1996). Changing criteria of autistic disorders: A comparison of the ICD-10 research criteria and DSM-IV with DSMIII-R, CARS, and ABC. Journal of Autism and Developmental Disorders, 26, 513-525.
Sudhalter, V. (1996). The emergence of tangential language in fragile X males. In V. Sudhalter (Chair), The Phenotype of Males and Females with Fragile X Syndrome. Symposium conducted at the 29th Annual Gatlinburg Conference on Research and Theory in Mental Retardation and Developmental Disabilities, Gatlinburg, Tennessee.
Sudhalter, V., Cohen, I., Silverman, W., & Wolf-Schein, E. (1990). Conversational analyses of males with fragile X, Down syndrome, and autism: Comparison of the emergence of deviant language. American Journal on Mental Retardation, 94, 431-441.
Thomas, A., Chess, S., Birch, H., Hertzig, M., & Korn, S. (1963). Behavioral individuality in early childhood. New York: University Press.
Travis, L., & Sigman, S. (1998). Social deficits and interpersonal relationships in autism. Mental Retardation and Developmental Disabilities Research Reviews, 4, 65-72.
Turk J., & Graham, P. (1997). Fragile X syndrome, autism, and autistic features. Autism, 1, 175-197.
Author information
Authors and Affiliations
Rights and permissions
About this article
Cite this article
Bailey, D.B., Hatton, D.D., Mesibov, G. et al. Early Development, Temperament, and Functional Impairment in Autism and Fragile X Syndrome. J Autism Dev Disord 30, 49–59 (2000). https://doi.org/10.1023/A:1005412111706
Issue Date:
DOI: https://doi.org/10.1023/A:1005412111706