Hostname: page-component-7dd5485656-k8lnt Total loading time: 0 Render date: 2025-10-31T19:39:56.565Z Has data issue: false hasContentIssue false
  • English
  • Français

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

Published online by Cambridge University Press:  25 March 2011

BRUNO ESTIGARRIBIA ,
GARY E. MARTIN ,
JOANNE E. ROBERTS ,
AMY SPENCER ,
AGNIESZKA GUCWA  and
JOHN SIDERIS
BRUNO ESTIGARRIBIA*
Affiliation:
University of North Carolina at Chapel Hill
GARY E. MARTIN
Affiliation:
University of North Carolina at Chapel Hill
JOANNE E. ROBERTS
Affiliation:
University of North Carolina at Chapel Hill
AMY SPENCER
Affiliation:
University of North Carolina at Greensboro
AGNIESZKA GUCWA
Affiliation:
University of North Carolina at Chapel Hill
JOHN SIDERIS
Affiliation:
University of North Carolina at Chapel Hill
*
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
Get access Rights & Permissions [Opens in a new window]

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.

Information

Type
Articles
Information
Applied Psycholinguistics , Volume 32 , Issue 2 , April 2011 , pp. 359 - 388
Copyright
Copyright © Cambridge University Press 2011

Access options

Get access to the full version of this content by using one of the access options below. (Log in options will check for institutional or personal access. Content may require purchase if you do not have access.)

Article purchase

Temporarily unavailable

References

REFERENCES

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, 9–15.Google Scholar
Astington, J. W. (1990). Narrative thought and narrative language. Hillsdale, NJ: Erlbaum.Google Scholar
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, 49–59.Google Scholar
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, 499–508.Google Scholar
Bamberg, M. G. W. (1987). The acquisition of narratives: Learning to use language. Berlin: Mouton de Gruyter.Google Scholar
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, 744–752.Google Scholar
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, 25–46.Google Scholar
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, 1146–1159.Google Scholar
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, 422–432.Google Scholar
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, 519–531.Google Scholar
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, 1301–1332.Google Scholar
Chang, C.-J. (2004). Telling stories of experiences: Narrative development of young Chinese children. Applied Psycholinguistics, 25, 83–104.Google Scholar
Chapman, R. S., & Hesketh, L. J. (2000). Behavioral phenotype of individuals with Down syndrome. Mental Retardation and Developmental Disabilities Research Reviews, 6, 84–95.Google Scholar
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, 1106–1120.Google Scholar
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, 861–873.Google Scholar
Crowley, J., & Glasgow, C. (1994). The Renfrew Bus Story. Language screening by narrative recall. Centreville, DE: Centreville School.Google Scholar
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, 314–326.Google Scholar
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, 87–102.Google Scholar
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, 1432–1443.Google Scholar
Dunn, L. M., & Dunn, L. M. (1997). Peabody Picture Vocabulary Test (3rd ed.). Circle Pines, MN: American Guidance Service.Google Scholar
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, 1441–1457.Google Scholar
Eisenberg, A. R. (1985). Learning to describe past experiences in conversation. Discourse Processes, 8, 177–204.Google Scholar
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, 132–145.Google Scholar
Fleiss, J. L. (1981). Statistical methods for rates and proportions. New York: Wiley.Google Scholar
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.Google Scholar
Frazier, J. A. (1994). Narration of “Frog, Where Are You?” coding scheme form. Greensboro, NC: Greensboro Early Schooling Study.Google Scholar
García-Pérez, R. M., Hobson, R. P., & Lee, A. (2008). Narrative role-taking in autism. Journal of Autism and Developmental Disorders, 38, 156–168.Google Scholar
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. 3–109). Baltimore, MD: Johns Hopkins University Press.Google Scholar
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.Google Scholar
Halliday, M. A. K., & Hasan, R. (1976). Cohesion in English. London: Longman.Google Scholar
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, 339–368.Google Scholar
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. 12–38). Boulder, CO: Westview Press.Google Scholar
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, 503–516.Google Scholar
Holdaway, D. (1979). The foundations of literacy. Sydney: Ashton Scholastic.Google Scholar
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. 89–136). Hillsdale, NJ: Erlbaum.Google Scholar
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, 91–113.Google Scholar
Johnston, J. R. (1982). Narratives: A new look at communication problems in older language-disordered children. Language, Speech, and Hearing Services in Schools, 13, 145–155.Google Scholar
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, 225–234.Google Scholar
Kay-Raining Bird, E., & Chapman, R. S. (1994). Sequential recall in individuals with Down syndrome. Journal of Speech and Hearing Research, 37, 1369–1380.Google Scholar
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, 1286–1300.Google Scholar
Keller, H. (2003). A bed full of cats. San Diego, CA: Harcourt Children's Books.Google Scholar
Keller-Bell, Y. D., & Abbeduto, L. (2007). Narrative development in adolescents and young adults with fragile X syndrome. American Journal on Mental Retardation, 112, 289–299.Google Scholar
Kemper, S., & Edwards, L. L. (1986). Children's expression of causality and their construction of narratives. Topics in Language Disorders, 7, 11–20.Google Scholar
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, 2131–2141.Google Scholar
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. 125–146). New York: Springer–Verlag.Google Scholar
Labov, W., & Waletzky, J. (1967). Narrative analysis: Oral versions of personal experience. In Helm, J. (Ed.), Essays on the verbal and visual arts (pp. 74–104). Seattle, WA: University of Washington Press.Google Scholar
Levy, Y., Gottesman, R., Borochowitz, Z., Frydman, M., & Sagi, M. (2006). Language in boys with fragile X syndrome. Journal of Child Language, 33, 125–144.Google Scholar
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, 532–545.Google Scholar
Lord, C., Rutter, M., DiLavore, P. C., & Risi, S. (2002). Autism Diagnostic Observation Schedule. Los Angeles: Western Psychological Services.Google Scholar
Losh, M., & Capps, L. (2003). Narrative ability in high-functioning children with autism or Asperger's syndrome. Journal of Autism and Developmental Disorders, 33, 239–251.Google Scholar
Loveland, K. A., McEvoy, R. E., & Tunali, B. (1990). Narrative story telling in autism and Down's syndrome. British Journal of Developmental Psychology, 8, 9–23.Google Scholar
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. 55–76). Cambridge, MA: MIT Press.Google Scholar
MacWhinney, B. (2000). The CHILDES project: Tools for analyzing talk (3rd ed.). Mahwah, NJ: Erlbaum.Google Scholar
MacWhinney, B., & Snow, C. (1985). The child language data exchange system. Journal of Child Language, 12, 271–296.Google Scholar
Martin, G. E., Klusek, J., Estigarribia, B., & Roberts, J. E. (2009). Language characteristics of individuals with Down syndrome. Topics in Language Disorders, 29, 112–132.Google Scholar
Mayer, M. (1977). Frog goes to dinner. New York: Puffin Books.Google Scholar
Medved, M. I., & Brockmeier, J. (2004). Making sense of traumatic experiences: Telling your life with fragile X syndrome. Qualitative Health Research, 14, 741–759.Google Scholar
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, 175–189.Google Scholar
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, 325–337.Google Scholar
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, 412–429.Google Scholar
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, 227–251.Google Scholar
Norbury, C. F., & Bishop, D. V. M. (2003). Narrative skills of children with communication impairments. International Journal of Language and Communication Disorders, 38, 287–313.Google Scholar
O'Donnell, W. T., & Warren, S. T. (2002). A decade of molecular studies of fragile X syndrome. Annual Review of Neuroscience, 25, 315–338.Google Scholar
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. 3–26). Baltimore, MD: Paul H. Brookes.Google Scholar
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, 572–598.Google Scholar
Peterson, C., & Dodsworth, P. (1991). A longitudinal analysis of young children's cohesion and noun specification in narratives. Journal of Child Language, 18, 397–415.Google Scholar
Peterson, C., & McCabe, A. (1983). Developmental psycholinguistics: Three ways of looking at a child's narrative. New York: Plenum Press.Google Scholar
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, 208–218.Google Scholar
Pradl, G. M. (1979). Learning how to begin and end a story. Language Arts, 56, 21–25.Google Scholar
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, 178–190.Google Scholar
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, 318–326.Google Scholar
Renfrew, C. E. (1991). The bus story: A test of continuous speech (2nd ed.). Oxford: C. E. Renfrew.Google Scholar
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, 1–15.Google Scholar
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, 980–995.Google Scholar
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, 295–304.Google Scholar
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, 409–417.Google Scholar
Roid, G. H., & Miller, L. J. (1997). Leiter International Performance Scale—Revised. Wood Dale, IL: Stoelting.Google Scholar
Roizen, N. J., & Patterson, D. (2003). Down's syndrome. Lancet, 361, 1281–1289.Google Scholar
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, 257–277.Google Scholar
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. 211–236). New York: Academic Press.Google Scholar
Scarborough, H. S. (1990). The Index of Productive Syntax. Applied Psycholinguistics, 11, 1–22.Google Scholar
Schafer, J. L., & Graham, J. W. (2002). Missing data: Our view of the state of the art. Psychological Methods, 7, 147–177.Google Scholar
Sherman, S. (2002). Epidemiology. In Hagerman, R. J. & Hagerman, P. J. (Eds.), Fragile X syndrome: Diagnosis, treatment, and research (3rd ed., pp. 136–168). Baltimore, MD: Johns Hopkins University Press.Google Scholar
Shrout, P. E., & Fleiss, J. L. (1979). Intraclass correlations: Uses in assessing rater reliability. Psychological Bulletin, 86, 420–428.Google Scholar
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, 1–18.Google Scholar
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.Google Scholar
Stein, N. L. (1988). The development of children's storytelling skill. In Franklin, M. B. & Barten, S. S. (Eds.), Child language: A reader (pp. 282–297). Oxford: Oxford University Press.Google Scholar
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. 53–120). Norwood, NJ: Ablex.Google Scholar
Tager-Flusberg, H. (1995). “Once upon a ribbit”: Stories narrated by autistic children. British Journal of Developmental Psychology, 13, 45–59.Google Scholar
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, 207–212.Google Scholar
Thomas, M., & Karmiloff-Smith, A. (2005). Can developmental disorders reveal the component parts of the human language faculty? Language Learning and Development, 1, 65–92.Google Scholar
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. 83–111). Hillsdale, NJ: Erlbaum.Google Scholar
Trabasso, T., & van den Broek, P. (1985). Causal thinking and the representation of narrative events. Journal of Memory and Language, 24, 612–630.Google Scholar
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. 321–342). Mahwah, NJ: Erlbaum.Google Scholar
Wagner, R. K., Torgesen, J. K., & Rashotte, C. A. (1999). Comprehensive Test of Phonological Processes. Austin, TX: PRO-ED.Google Scholar
Williams, K. T. (1997). Expressive Vocabulary Test. Circle Pines, MN: American Guidance Service.Google Scholar
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, 439–455.Google Scholar
Woodcock, R. W., McGrew, K. S., & Mather, N. (2001). Woodcock–Johnson Tests of Cognitive Abilities (3rd ed.). Itasca, IL: Riverside.Google Scholar