Abstract
Social escape behavior is a common behavioral feature of individuals with fragile X syndrome (fraX). In this observational study, we examined the effect of antecedent social and performance demands on problem behaviors in four conditions: face-to-face interview, silent reading, oral reading and a singing task. Results showed that problem behaviors were significantly more likely to occur during the interview and singing conditions. Higher levels of salivary cortisol were predictive of higher levels of fidgeting behavior and lower levels of eye contact in male participants. There were no associations between level of FMRP expression and social escape behaviors. These data suggest that specific antecedent biological and environmental factors evoke social escape behaviors in fragile X syndrome.
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Notes
Three male participants did not participate in the silent and oral reading conditions because they were either uncooperative or were unable to read. Ten male participants did not participate in the silent reading condition because they read aloud despite being prompted to read silently to themselves. One male participant was unable to complete any of the tasks and was therefore excluded from the study.
Eye contact prompts were not delivered in the Silent Reading, Oral Reading or Singing conditions.
It should be noted that 87.3% of the male participants and 97.1% of the female participants reciprocated the handshake greeting with the experimenter. Of those who reciprocated the handshake, 34.6% of the male participants and 70.6% of the female participants established eye contact when shaking hands.
References
Achenbach T. M. (1991). Manual for the child behavior checklist/4–18 and 1991 profile. Burlington, VT: University of Vermont, Department of Psychiatry.
Anderson L. T., Dancis L., & Alpert M. (1978). Behavioral contingencies and self-mutilation in Lesch-Nyhan disease. Journal of Consulting and Clinical Psychology, 46, 529–536.
Bakeman R., & Quera V. (1995). Analyzing interaction: sequential analysis with SDIS and GSEQ. Cambridge: Cambridge University Press.
Bodfish J. W., & Lewis M. H. (2002). Self-injury and comorbid behaviors in developmental, neurological, psychiatric and genetic disorders. In S. R. Schroeder, M. L. Oster-Granite, & T. Thompson (Eds.), Self-injurious behavior: Gene-brain-behavior relationships (pp. 23–40). Washington DC: American Psychological Association.
Call N. A., Wacker D. P., Ringdahl J. E., Cooper-Brown L. J., & Boelter E. W. (2004). An assessment of antecedent events influencing noncompliance in an outpatient clinic. Journal of Applied Behavior Analysis, 37, 145–158.
Carr E. G., & Durand V. M. (1985). The social-communicative basis of severe behavior problems in children. In S. Reiss, & R. Bootzin (Eds.), Theoretical issues in behavior therapy. New York: Academic Press.
Christie R., Bay C., Kaufman I. A., Bakay B., Borden M., & Nyhan W. L. (1982). Lesch-Nyhan disease: Clinical experience with nineteen patients. Developmental Medicine and Child Neurology, 24, 293–306.
Cohen I., Fisch G. S., Sudhalter V., Wolf-Schein E. G., Hanson D., & Hagerman R., et al. (1988). Social gaze, social avoidance, and repetitive behavior in Fragile X males: A controlled study. American Journal on Mental Retardation, 92, 436–446.
Cohen, I. L., Vietze, P. M., Sudhalter, V., Jenkins, E. C., & Brown, W. T. (1991). Effects of age and communication level on eye contact in fragile X males and non-fragile X autistic males. American Journal of Medical Genetics, 38, 498–502.
Crawford D. C., Meadows K. L., Newman J. L., Taft L. F., Stanfeild M. L., & Holmgreen P., et al. (1999). Prevalence and phenotype consequence of FRAXA and FRAXE alleles in a large, ethnically diverse, special education-needs population. American Journal of Human Genetics, 64, 495–507.
Curin J. M., Terzic J., Petkovic Z. B., Zekan L., Terzic I. M., & Susnjara I. M. (2003). Lower cortisol and higher ACTH levels in individuals with autism. Journal of Autism and Developmental Disorders, 33, 443–448.
Derby K. M., Wacker D. P., Sasso G., Steege M., Northup J., & Cigrand K., et al. (1992). Brief functional assessment techniques to evaluate aberrant behavior in an outpatient setting: A summary of 79 cases. Journal of Applied Behavior Analysis, 25, 713–721.
Durand V. M., & Carr E. G. (1987). Social influences on “self-stimulatory” behavior: Analysis and treatment application. Journal of Applied Behavior Analysis, 20, 119–132.
Finucane B. M., Konar D., Haas-Givler B., Kurtz M. B., & Scott C. I. (1994). The spasmodic upper-body squeeze: A characteristic behavior in Smith-Magenis syndrome. Developmental Medicine and Child Neurology, 36, 70–83.
Hagberg B., Aicardi J., Dicas K., & Ramos O. (1983). A progressive syndrome of autism, dementia, ataxia, and loss of purposeful hand use in girls Rett’s syndrome: report of 35 cases. Annals of Neurology, 14, 471–479.
Hall S., Oliver C., & Murphy G. (2001). Self-injurious behaviour in young children with Lesch-Nyhan Syndrome. Developmental Medicine and Child Neurology, 43, 745–749.
Hall S., Thorns T., & Oliver C. (2003). Structural and environmental characteristics of stereotyped behaviors. American Journal on Mental Retardation, 108, 391–402.
Hanley G. P., Iwata B. A., & McCord B. E. (2003). Functional analysis of problem behavior: A review. Journal of Applied Behavior Analysis, 36, 147–185.
Hartmann D. P. (1977). Considerations in the choice of interobserver reliability estimates. Journal of Applied Behavior Analysis, 10, 103–116.
Hessl D., Glaser B., Dyer-Friedman J., Blasey C., Hastie T., & Gunnar M., et al. (2002). Cortisol and behavior in fragile X syndrome. Psychoneuroendocrinology, 27, 855–872.
Iwata B. A., Pace G. M., Dorsey M. F., Zarcone J. R., Vollmer T. R., & Smith R. G., et al. (1994). The functions of self-injurious behavior: An experimental epidemiological analysis. Journal of Applied Behavior Analysis, 27, 215–240.
Kennedy C. H., Caruso M., & Thompson T. (2001). Experimental analyses of gene–brain-behavior relations: Some notes on their application. Journal of Applied Behavior Analysis, 34, 539–549.
Lesniak-Karpiak K., Mazzocco M. M. M., & Ross J. L. (2003). Behavioral assessment of social anxiety in females with Turner or fragile X syndrome. Journal of Autism and Developmental Disorders, 33, 55–67.
Martin N., Oliver C., & Hall S. (1998). Obswin: Software for the collection and analysis of observational data. Birmingham: University of Birmingham.
Oliver C., Murphy G., Crayton L., & Corbett J. (1993). Self-injurious behavior in Rett syndrome: Interactions between features of Rett syndrome and operant conditioning. Journal of Autism and Developmental Disorders, 23, 91–109.
Oliver C., Oxener G., Hearn M., & Hall S. (2001). Effects of social proximity on multiple aggressive behaviors. Journal of Applied Behavior Analysis, 34, 85–88.
Pelios L., Morren J., Tesch D., & Axelrod S. (1999). The impact of functional analysis methodology on treatment choice for self-injurious and aggressive behavior. Journal of Applied Behavior Analysis, 32, 185–195.
Reiss A. L., & Dant C. C. (2003). The behavioral neurogenetics of fragile X syndrome: Analyzing gene-brain-behavior relationships in child developmental psychopathologies. Development and Psychopathology, 15, 927–968.
Sackett G. (1987). Analysis of sequential social interaction data: Some issues, recent developments, and a causal inference model. In J. D. Osofsky (Ed.), Handbook of infant development (2nd edn.). New York: Wiley and Son.
Schroeder S. R., Oster-Granite M. L., Berkson G., Bodfish J. W., Breese G. R., & Cataldo M. F., et al. (2001). Self-injurious behavior: Gene-brain-behavior relationships. Mental Retardation and Developmental Disabilities Research Reviews, 7, 3–12.
Sparrow S. S., Balla D. A., & Cicchetti D. V. (1984). Vineland adaptive behavior scales: Interview edition. survey form manual. Circle Pines, MN: American Guidance Service, Inc.
Summers J. A., Allison D. B., Lynch P. S., & Sandler L. (1995). Behavior problems in Angelman syndrome. Journal of Intellectual Disability Research, 39, 97–106.
Symons F. J., Clark R. D., Hatton D. D., Skinner M., & Bailey D. B. (2003). Self-injurious behavior in young boys with fragile X syndrome. American Journal of Medical Genetics, 118A, 115–121.
Taylor A. K., Safanda J. F., Fall M. Z., Quince C., Lang K. A., & Hull C. E., et al. (1994). Molecular predictors of cognitive involvement in female carriers of fragile X syndrome. Journal of the American Medical Association, 271, 507–514.
Taylor J. C., & Carr E. G. (1992). Severe problem behaviors related to social interaction: I. Attention seeking and social avoidance. Behavior Modification, 16: 305–335.
Thornton L., & Dawson K. P. (1990). Prader-Willi syndrome in New Zealand: A survey of 36 affected people. New Zealand Medical Journal, 103(97–98).
Verkerk A. J., Pieretti M., Sutcliffe J. S., Fu Y. H., Kuhl D. P., & Pizzuti A., et al. (1991). Identification of a gene (FMR-1) containing a CGG repeat coincident with a breakpoint cluster region exhibiting length variation in fragile X syndrome. Cell, 65, 905–914.
Willemsen R., Smits A., Mohkamsing S., van Beerendonk H., de Haan A., & de Vries B., et al. (1997). Rapid antibody test for diagnosing fragile X syndrome: A validation of the technique. Human Genetics, 99, 308–311.
Yoder P. J., & Feurer I. D. (2000). Quantifying the magnitude of sequential association between events or behaviors. In T. Thompson, D. Felce, & F. J. Symons (Eds.), Behavioral observation: Technology and applications in developmental disabilities. Baltimore: Paul H. Brooks Co.
Acknowledgments
The authors would like to thank David Hessl, Jennifer Dyer-Friedman, Jacob Wisbeck, Bronwyn Glaser, Donna Mumme, and Cindy Johnston for their participation in this project. This research was supported by NIH grants MH50047 and MH01142 and by a summer studentship awarded to the second author from the National Fragile X Foundation William Rosen Research Award. Portions of these data were presented at the 30th Annual Convention of the Association for Applied Behavior Analysis, Boston, 2004.
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Hall, S., DeBernardis, M. & Reiss, A. Social Escape Behaviors in Children with Fragile X Syndrome. J Autism Dev Disord 36, 935–947 (2006). https://doi.org/10.1007/s10803-006-0132-z
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DOI: https://doi.org/10.1007/s10803-006-0132-z