Skip to main content Accessibility help
×
Home

Narrative skill in boys with fragile X syndrome with and without autism spectrum disorder

  • BRUNO ESTIGARRIBIA (a1), GARY E. MARTIN (a1), JOANNE E. ROBERTS (a1), AMY SPENCER (a2), AGNIESZKA GUCWA (a1) and JOHN SIDERIS (a1)...

Abstract

We examined recalled narratives of boys with fragile X syndrome with autism spectrum disorder (FXS-ASD; N = 28) and without ASD (FXS-O; N = 29), and compared them to those of boys with Down syndrome (N = 33) and typically developing (TD) boys (N = 39). Narratives were scored for mentions of macrostructural story grammar elements (introduction, relationship, initiating events, internal response, attempts/actions, and ending). We found that narrative recall is predicted by short-term memory and nonverbal mental age levels in almost all groups (except TD), but not by expressive syntax or caregiver education. After adjusting for these covariates, there were no differences between the three groups with intellectual disability. The FXS-ASD group, however, had significantly poorer performance than the TD group on the overall story grammar score, and both the FXS-O and FXS-ASD groups had lower attempts/actions scores than the TD group. We conclude that some form of narrative impairment may be associated with FXS, that this impairment may be shared by other forms of intellectual disability, and that the presence of ASD has a significantly detrimental effect on narrative recall.

Copyright

Corresponding author

ADDRESS FOR CORRESPONDENCE Bruno Estigarribia, Department of Psychology, Davie Hall, Room 364a, University of North Carolina at Chapel Hill, Chapel Hill, NC 27599-8180. E-mail estigarr@email.unc.edu

References

Hide All
Abbeduto, L., Pavetto, M., Kesin, E., Weissman, M. D., Karadottir, S., O'Brien, A., et al. (2001). The linguistic and cognitive profile of Down syndrome: Evidence from a comparison with fragile X syndrome. Down Syndrome Research and Practice, 7, 915.
Astington, J. W. (1990). Narrative thought and narrative language. Hillsdale, NJ: Erlbaum.
Bailey, D. B., Hatton, D. D., Mesibov, G. B., Ament, N., & Skinner, M. (2000). Early development, temperament and functional impairment in autism and fragile X syndrome. Journal of Autism and Developmental Disorders, 30, 4959.
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, 499508.
Bamberg, M. G. W. (1987). The acquisition of narratives: Learning to use language. Berlin: Mouton de Gruyter.
Baumgardner, T. L., Reiss, A. L., Freund, L. S., & Abrams, M. T. (1995). Specification of the neurobehavioral phenotype in males with fragile X syndrome. Pediatrics, 95, 744752.
Bishop, D. V. M., & Donlan, C. (2005). The role of syntax in encoding and recall of pictorial narratives: Evidence from specific language impairment. British Journal of Developmental Psychology, 23, 2546.
Boudreau, D. M., & Chapman, R. S. (2000). The relationship between event representation and linguistic skill in narratives of children and adolescents with Down syndrome. Journal of Speech, Language, and Hearing Research, 43, 11461159.
Bowyer-Crane, C., Snowling, M. J., Duff, F. J., Fieldsend, E., Carroll, J. M., Miles, J., et al. (2008). Improving early language and literacy skills: Differential effects of an oral language versus a phonology with reading intervention. Journal of Child Psychology and Psychiatry, 49, 422432.
Burack, J. A., Shulman, C., Katzir, E., Schaap, T., Iarocci, G., & Amir, P. N. (1999). Cognitive and behavioural development of Israeli males with fragile X and Down syndrome. International Journal of Behavioral Development, 23, 519531.
Centers for Disease Control and Prevention. (2006). Improved national prevalence estimates for 18 selected major birth defects—United States, 1999–2001. Morbidity and Mortality Weekly Report, 54, 13011332.
Chang, C.-J. (2004). Telling stories of experiences: Narrative development of young Chinese children. Applied Psycholinguistics, 25, 83104.
Chapman, R. S., & Hesketh, L. J. (2000). Behavioral phenotype of individuals with Down syndrome. Mental Retardation and Developmental Disabilities Research Reviews, 6, 8495.
Chapman, R. S., Schwartz, S., & Kay-Raining Bird, E. (1991). Language skills of children and adolescents with Down syndrome: I. Comprehension. Journal of Speech, Language, and Hearing Research, 34, 11061120.
Chapman, R. S., Seung, H., Schwartz, S. E., & Kay-Raining Bird, E. (1998). Language skills of children and adolescents with Down syndrome: II. Production deficits. Journal of Speech, Language, and Hearing Research, 41, 861873.
Crowley, J., & Glasgow, C. (1994). The Renfrew Bus Story. Language screening by narrative recall. Centreville, DE: Centreville School.
Demark, J. L., Feldman, M. A., & Holden, J. J. A. (2003). Behavioral relationship between autism and fragile X syndrome. American Journal on Mental Retardation, 108, 314326.
Diehl, J. J., Bennetto, L., & Carter-Young, E. (2006). Story recall and narrative coherence of high-functioning children with autism spectrum disorders. Journal of Abnormal Child Psychology, 34, 87102.
Dollaghan, C. A., Campbell, T. F., Paradise, J. L., Feldman, H. M., Janosky, J. E., Pitcairn, D. N., et al. (1999). Maternal education and measures of early speech and language. Journal of Speech, Language, and Hearing Research, 42, 14321443.
Dunn, L. M., & Dunn, L. M. (1997). Peabody Picture Vocabulary Test (3rd ed.). Circle Pines, MN: American Guidance Service.
Durkin, K., & Conti-Ramsden, G. (2007). Language, social behavior, and the quality of friendships in adolescents with and without a history of specific language impairment. Child Development, 78, 14411457.
Eisenberg, A. R. (1985). Learning to describe past experiences in conversation. Discourse Processes, 8, 177204.
Fewell, R. R., & Deutscher, B. (2004). Contributions of early language and maternal facilitation variables to later language and reading abilities. Journal of Early Intervention, 26, 132145.
Fleiss, J. L. (1981). Statistical methods for rates and proportions. New York: Wiley.
Fowler, A. E. (1995). Linguistic variability in persons with Down syndrome: Research and implications. In Nadel, L. & Rosenthal, D. (Eds.), Down syndrome: Living and learning in the community. New York: Wiley–Liss.
Frazier, J. A. (1994). Narration of “Frog, Where Are You?” coding scheme form. Greensboro, NC: Greensboro Early Schooling Study.
García-Pérez, R. M., Hobson, R. P., & Lee, A. (2008). Narrative role-taking in autism. Journal of Autism and Developmental Disorders, 38, 156168.
Hagerman, R. J. (2002). The physical and behavioural phenotype. In Hagerman, R. J. & Hagerman, P. J. (Eds.), Fragile X syndrome: Diagnosis, treatment, and research (3rd ed., pp. 3109). Baltimore, MD: Johns Hopkins University Press.
Hagerman, R. J. (2004). Physical and behavioural characteristics of fragile X syndrome. In Dew-Hughes, D. (Ed.), Educating children with Fragile X syndrome: A multi-professional view. London: Routledge.
Halliday, M. A. K., & Hasan, R. (1976). Cohesion in English. London: Longman.
Hayiou-Thomas, M., Kovas, Y., Harlaar, N., Plomin, R., Bishop, D., & Dale, P. (2006). Common aetiology for diverse language skills in 4 1/2-year-old twins. Journal of Child Language, 33, 339368.
Heath, S. B. (1998). What no bedtime story means: Narrative skills at home and school. In Brenneis, D. & Macaulay, R. K. S. (Eds.), The matrix of language: Contemporary linguistic anthropology (pp. 1238). Boulder, CO: Westview Press.
Hodapp, R. M., Dykens, E. M., Ort, S. I., Zelinsky, D. G., & Leckman, J. F. (1991). Changing patterns of intellectual strengths and weaknesses in males with fragile X syndrome. Journal of Autism and Developmental Disorders, 21, 503516.
Holdaway, D. (1979). The foundations of literacy. Sydney: Ashton Scholastic.
Hudson, J. A., & Shapiro, L. R. (1991). From knowing to telling: The development of children's scripts, stories, and personal narratives. In McCabe, A. & Peterson, C. (Eds.), Developing narrative structure (pp. 89136). Hillsdale, NJ: Erlbaum.
John, S. F., Lui, M., & Tannock, R. (2003). Children's story retelling and comprehension using a new narrative resource. Canadian Journal of School Psychology, 18, 91113.
Johnston, J. R. (1982). Narratives: A new look at communication problems in older language-disordered children. Language, Speech, and Hearing Services in Schools, 13, 145155.
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, 129, 225234.
Kay-Raining Bird, E., & Chapman, R. S. (1994). Sequential recall in individuals with Down syndrome. Journal of Speech and Hearing Research, 37, 13691380.
Kay-Raining Bird, E., Chapman, R. S., & Schwartz, S. E. (2004). Fast mapping of words and story recall by individuals with Down syndrome. Journal of Speech, Language, and Hearing Research, 47, 12861300.
Keller, H. (2003). A bed full of cats. San Diego, CA: Harcourt Children's Books.
Keller-Bell, Y. D., & Abbeduto, L. (2007). Narrative development in adolescents and young adults with fragile X syndrome. American Journal on Mental Retardation, 112, 289299.
Kemper, S., & Edwards, L. L. (1986). Children's expression of causality and their construction of narratives. Topics in Language Disorders, 7, 1120.
Kennedy, C. R., McCann, D. C., Campbell, M. J., Law, C. M., Mullee, M., Petrou, S., et al. (2006). Language ability after early detection of permanent childhood hearing impairment. New England Journal of Medicine, 354, 21312141.
Kuczaj, S. A., & McClain, L. (1984). Of hawks and moozes: The fantasy narratives produced by a young child. In Kuczaj, S. A. (Ed.), Discourse development: Progress in cognitive development research (pp. 125146). New York: Springer–Verlag.
Labov, W., & Waletzky, J. (1967). Narrative analysis: Oral versions of personal experience. In Helm, J. (Ed.), Essays on the verbal and visual arts (pp. 74104). Seattle, WA: University of Washington Press.
Levy, Y., Gottesman, R., Borochowitz, Z., Frydman, M., & Sagi, M. (2006). Language in boys with fragile X syndrome. Journal of Child Language, 33, 125144.
Lewis, P., Abbeduto, L., Murphy, M., Richmond, E., Giles, N., Bruno, L., et al. (2006). Cognitive, language and social–cognitive skills of individuals with fragile X syndrome with and without autism. Journal of Intellectual Disability Research, 50, 532545.
Lord, C., Rutter, M., DiLavore, P. C., & Risi, S. (2002). Autism Diagnostic Observation Schedule. Los Angeles: Western Psychological Services.
Losh, M., & Capps, L. (2003). Narrative ability in high-functioning children with autism or Asperger's syndrome. Journal of Autism and Developmental Disorders, 33, 239251.
Loveland, K. A., McEvoy, R. E., & Tunali, B. (1990). Narrative story telling in autism and Down's syndrome. British Journal of Developmental Psychology, 8, 923.
Lust, B., Flynn, S., & Foley, C. (1996). What children know about what they say: Elicited imitation as a research method for assessing children's syntax. In McDaniel, D., McKee, C., & Cairns, H. Smith (Eds.), Methods for assessing children's syntax (pp. 5576). Cambridge, MA: MIT Press.
MacWhinney, B. (2000). The CHILDES project: Tools for analyzing talk (3rd ed.). Mahwah, NJ: Erlbaum.
MacWhinney, B., & Snow, C. (1985). The child language data exchange system. Journal of Child Language, 12, 271296.
Martin, G. E., Klusek, J., Estigarribia, B., & Roberts, J. E. (2009). Language characteristics of individuals with Down syndrome. Topics in Language Disorders, 29, 112132.
Mayer, M. (1977). Frog goes to dinner. New York: Puffin Books.
Medved, M. I., & Brockmeier, J. (2004). Making sense of traumatic experiences: Telling your life with fragile X syndrome. Qualitative Health Research, 14, 741759.
Miles, S., & Chapman, R. S. (2002). Narrative content as described by individuals with Down syndrome and typically developing children. Journal of Speech, Language, and Hearing Research, 45, 175189.
Miles, S., Chapman, R., & Sindberg, H. (2006). Sampling context affects MLU in the language of adolescents with Down syndrome. Journal of Speech, Language, and Hearing Research, 49, 325337.
Nicolopoulou, A., & Richner, E. S. (2007). From actors to agents to persons: The development of character representation in young children's narratives. Child Development, 78, 412429.
Norbury, C. F., & Bishop, D. V. M. (2002). Inferential processing and story recall in children with communication problems: A comparison of specific language impairment, pragmatic language impairment and high-functioning autism. International Journal of Language and Communication Disorders, 37, 227251.
Norbury, C. F., & Bishop, D. V. M. (2003). Narrative skills of children with communication impairments. International Journal of Language and Communication Disorders, 38, 287313.
O'Donnell, W. T., & Warren, S. T. (2002). A decade of molecular studies of fragile X syndrome. Annual Review of Neuroscience, 25, 315338.
Patterson, D., & Lott, I. (2008). Etiology, diagnosis, and development in Down syndrome. In Roberts, J. E., Chapman, R. S., & Warren, S. F. (Eds.), Speech and language development and intervention in Down syndrome and fragile X syndrome (pp. 326). Baltimore, MD: Paul H. Brookes.
Paul, R., & Smith, R. L. (1993). Narrative skills in 4-year-olds with normal, impaired, and late-developing language. Journal of Speech and Hearing Research, 36, 572598.
Peterson, C., & Dodsworth, P. (1991). A longitudinal analysis of young children's cohesion and noun specification in narratives. Journal of Child Language, 18, 397415.
Peterson, C., & McCabe, A. (1983). Developmental psycholinguistics: Three ways of looking at a child's narrative. New York: Plenum Press.
Philofsky, A., Hepburn, S. L., Hayes, A., Hagerman, R., & Rogers, S. J. (2004). Linguistic and cognitive functioning and autism symptoms in young children with fragile X syndrome. American Journal on Mental Retardation, 109, 208218.
Pradl, G. M. (1979). Learning how to begin and end a story. Language Arts, 56, 2125.
Price, J. R., Roberts, J. E., & Jackson, S. C. (2006). Structural development of the fictional narratives of African American preschoolers. Language, Speech, and Hearing Services in Schools, 37, 178190.
Price, J. R., Roberts, J. E., Vandergrift, N., & Martin, G. (2007). Language comprehension in boys with fragile X syndrome and boys with Down syndrome. Journal of Intellectual Disability Research, 51, 318326.
Renfrew, C. E. (1991). The bus story: A test of continuous speech (2nd ed.). Oxford: C. E. Renfrew.
Roberts, J. E., Hennon, E. A., Price, J. R., Dear, E., Anderson, K., & Vandergrift, N. A. (2007). Expressive language during conversational speech in boys with fragile X syndrome. American Journal on Mental Retardation, 112, 115.
Roberts, J. E., Long, S. H., Malkin, C., Barnes, E. F., Skinner, M., Hennon, E. A., et al. (2005). A comparison of phonological skills of young males with fragile X syndrome and Down Syndrome. Journal of Speech, Language, and Hearing Research, 48, 980995.
Roberts, J. E., Mirrett, P., Anderson, K., & Neebe, M. B. E. (2002). Early communication, symbolic behavior, and social profiles of young males with fragile X syndrome. American Journal of Speech–Language Pathology, 11, 295304.
Rogers, S. J., Wehner, E. A., & 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. Developmental and Behavioral Pediatrics, 22, 409417.
Roid, G. H., & Miller, L. J. (1997). Leiter International Performance Scale—Revised. Wood Dale, IL: Stoelting.
Roizen, N. J., & Patterson, D. (2003). Down's syndrome. Lancet, 361, 12811289.
Roth, F. P., Speece, D. L., Cooper, D. H., & Paz, S. D. L. (1996). Unresolved mysteries: How do metalinguistic and narrative skills connect with early reading? Journal of Special Education, 30, 257277.
Rumelhart, D. E. (1975). Notes on a schema for stories. In Bobrow, D. G. & Collins, A. M. (Eds.), Representation and understanding: Studies in cognitive science (pp. 211236). New York: Academic Press.
Scarborough, H. S. (1990). The Index of Productive Syntax. Applied Psycholinguistics, 11, 122.
Schafer, J. L., & Graham, J. W. (2002). Missing data: Our view of the state of the art. Psychological Methods, 7, 147177.
Sherman, S. (2002). Epidemiology. In Hagerman, R. J. & Hagerman, P. J. (Eds.), Fragile X syndrome: Diagnosis, treatment, and research (3rd ed., pp. 136168). Baltimore, MD: Johns Hopkins University Press.
Shrout, P. E., & Fleiss, J. L. (1979). Intraclass correlations: Uses in assessing rater reliability. Psychological Bulletin, 86, 420428.
Simon, J. A., Keenan, J. M., Pennington, B. F., Taylor, A. K., & Hagerman, R. J. (2001). Discourse processing in women with fragile X syndrome: Evidence for a deficit establishing coherence. Cognitive Neuropsychology, 18, 118.
Stein, N. L. (1978). How children understand stories: A developmental analysis. Technical report. Urbana, IL: University of Illinois at Urbana–Champaign, Center for the Study of Reading.
Stein, N. L. (1988). The development of children's storytelling skill. In Franklin, M. B. & Barten, S. S. (Eds.), Child language: A reader (pp. 282297). Oxford: Oxford University Press.
Stein, N. L., & Glenn, C. G. (1979). Directions in discourse processing. In Freedle, R. O. (Ed.), An analysis of story comprehension in elementary school children (pp. 53120). Norwood, NJ: Ablex.
Tager-Flusberg, H. (1995). “Once upon a ribbit”: Stories narrated by autistic children. British Journal of Developmental Psychology, 13, 4559.
Tan, B. S., Law, H. Y., Zhao, Y., Yoon, C. S., & Ng, I. S. (2000). DNA testing for fragile X syndrome in 255 males from special schools in Singapore. Annals Academy of Medicine Singapore, 29, 207212.
Thomas, M., & Karmiloff-Smith, A. (2005). Can developmental disorders reveal the component parts of the human language faculty? Language Learning and Development, 1, 6592.
Trabasso, T., Secco, T., & van den Broek, P. (1984). Causal cohesion and story coherence. In Mandl, H., Stein, N. L., & Trabasso, T. (Eds.), Learning and comprehension of text (pp. 83111). Hillsdale, NJ: Erlbaum.
Trabasso, T., & van den Broek, P. (1985). Causal thinking and the representation of narrative events. Journal of Memory and Language, 24, 612630.
van den Broek, P. W. (1997). Discovering the cement of the universe: The development of event comprehension from childhood to adulthood. In van den Broek, P., Bauer, P., & Bourg, T., Developmental spans in event comprehension: Bridging fictional and actual events (pp. 321342). Mahwah, NJ: Erlbaum.
Wagner, R. K., Torgesen, J. K., & Rashotte, C. A. (1999). Comprehensive Test of Phonological Processes. Austin, TX: PRO-ED.
Williams, K. T. (1997). Expressive Vocabulary Test. Circle Pines, MN: American Guidance Service.
Wolman, C., van den Broek, P., & Lorch, R. F. (1997). Effects of causal structure on immediate and delayed story recall by children with mild mental retardation, children with learning disabilities, and children without disabilities. Journal of Special Education, 30, 439455.
Woodcock, R. W., McGrew, K. S., & Mather, N. (2001). Woodcock–Johnson Tests of Cognitive Abilities (3rd ed.). Itasca, IL: Riverside.

Metrics

Full text views

Total number of HTML views: 0
Total number of PDF views: 0 *
Loading metrics...

Abstract views

Total abstract views: 0 *
Loading metrics...

* Views captured on Cambridge Core between <date>. This data will be updated every 24 hours.

Usage data cannot currently be displayed