Skip to main content
Log in

Is Theory of Mind Understanding Impaired in Males with Fragile X Syndrome?

  • Original Paper
  • Published:
Journal of Abnormal Child Psychology Aims and scope Submit manuscript

Abstract

Males with fragile X syndrome (FXS) have difficulties with social interaction and many show autistic features. This study examined whether the social deficits characteristic of FXS are associated with theory of mind difficulties. Two groups of boys with FXS participated: a group with few autistic features and a group with many autistic features. An intellectual disability control group also participated. In addition to using standard theory of mind tasks, new techniques were used that were able to separate out the various processing demands of the task (e.g., memory, inhibitory control). Overall, the findings indicate that both groups of boys with FXS have difficulty with theory of mind tasks compared to an intellectual disability control group. However, both groups with FXS also performed worse on comparison trials that required working memory but not theory of mind. Theory of mind difficulties are likely to be an important aspect of the FXS clinical profile, but are most likely the result from a more basic difficulty with working memory.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Fig. 1
Fig. 2

Similar content being viewed by others

References

  • Apperly, I. A., Samson, D., Chiavarino, C., & Humphreys, G. W. (2004). Frontal and temporo-parietal lobe contribution to theory of mind: Neuropsychological evidence from a false belief task with reduced language and executive demands. Journal of Cognitive Neuroscience, 16(10), 1773–1784.

    Article  PubMed  Google Scholar 

  • Apperly, I. A., Samson, D., & Humphreys, G. W. (2005). Domain-specificity and theory of mind: Evaluating evidence from neuropsychology. Trends in Cognitive Sciences, 9(12), 572–577.

    Article  PubMed  Google Scholar 

  • Astington, J. W. (2000). Minds in the Making: Essays in honour of David R. Olson. Oxford: Blackwell.

    Google Scholar 

  • Astington, J. W., & Jenkins, J. M. (1999) A longitudinal study of the relation between language and theory-of-mind development. Developmental Psychology, 35, 1311–1320.

    Article  PubMed  Google Scholar 

  • Bailey, D. B., Mesibov, G., 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.

    Article  PubMed  Google Scholar 

  • Bailey, D. B., & Nelson, D. (1995). The nature and consequences of fragile X syndrome. Mental Retardation and Developmental Disability Research Review, 1, 238–244.

    Article  Google Scholar 

  • Baron-Cohen, S., Leslie, A. M., & Frith, U. (1985). Does the autistic child have a theory of mind? Cognition, 21, 37–46.

    Article  PubMed  Google Scholar 

  • Baron-Cohen, S. (2000). Theory of mind and autism: A fifteen year review. In S. Baron-Cohen, H. Tager-Flusberg, & D. J. Cohen (Eds.), Understanding other minds: Perspectives from developmental cognitive neuroscience. Oxford: Oxford University Press.

    Google Scholar 

  • Belser, R. C., Sudhalter, V. (2001). Fragile X syndrome: repetitive speech. American Journal of Mental Retardation, 106, 28–38.

    Article  PubMed  Google Scholar 

  • Bennetto, L., & Pennington, B. F. (2002). Neuropsychology. In R. J. Hagerman & P. J. Hagerman (Eds.), Fragile X syndrome: Diagnosis, treatment, and research (3rd ed., pp. 206–248). Baltimore, MD: Johns Hopkins University Press.

    Google Scholar 

  • Bennetto, L., Pennington, B. F., Porter, D., Taylor, A. K., & Hagerman, R. J. (2001). Profile of cognitive functioning in women with the fragile X mutation. Neuropsychology, 15, 290–299.

    Article  PubMed  Google Scholar 

  • Berument, S. K., Rutter, M., Lord, C., Pickles, A., & Bailey, A. (1999). Autism screening questionnaire: Diagnostic validity. British Journal of Psychiatry, 175, 444–451.

    Article  PubMed  Google Scholar 

  • Carlson, S. M., & Moses, L. J. (2001). Individual differences in inhibitory control and children’s theory of mind. Child Development, 72, 1032–1053.

    Article  PubMed  Google Scholar 

  • Cornish, K., Burack, J., Rahman, A., Munir, F., Russo, N., & Grant, C. (2005). Theory of mind deficits in children with Fragile X syndrome. Journal of Intellectual Disability Research, 49, 372–378.

    Article  PubMed  Google Scholar 

  • Cornish, K. M., Munir, F., & Cross, G. (2001). Differential impact of the FMR-1 full mutation on memory and attention functioning: A neuropsychological perspective. Journal of Cognitive Neuroscience, 13, 144–150.

    Article  PubMed  Google Scholar 

  • Cornish, K., Sudhalter, V., & Turk, J. (2004). Attention and language in fragile X. Mental Retardation and Developmental Disabilities Research Reviews, 10, 11–16.

    Article  PubMed  Google Scholar 

  • de Villiers, J. G., & Pyers, J. E. (2002) Complements to cognition: A longitudinal study of the relationship between complex syntax and false-belief- understanding. Cognitive Development, 17, 1037–1060.

    Article  Google Scholar 

  • Dunn, L. M., Dunn, L. M., Whetton, C., & Burley, J. (1997). British picture vocabulary scale (2nd ed.) (BPVS-II). Windsor, Berks: NFER-Nelson.

    Google Scholar 

  • Dykens, E. M., & 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.). New York: Wiley.

    Google Scholar 

  • Frith, U., & Frith, C. D. (2003) Development and neurophysiology of mentalizing. Philosophical Transactions of the Royal Society of London B. Biological Sciences, 358, 459–473.

    Article  Google Scholar 

  • Garner, C., Callias, M., & Turk, J. (1999). Executive function and theory of mind performance of boys with fragile-X syndrome. Journal of Intellectual Disability Research, 43, 466–474.

    Article  PubMed  Google Scholar 

  • Hadwin, J., Baron-Cohen, S., Howlin, P., & Hill, K. (1997). Does teaching theory of mind have an effect on the ability to develop conversational ability in children with autism? Journal of autism and Developmental Disorders, 27, 519–537.

    Article  PubMed  Google Scholar 

  • Happé, F. G. E. (1995). The role of age and verbal ability in the theory of mind task performance of subjects with autism. Child Development, 66, 843–845.

    Article  PubMed  Google Scholar 

  • Howlin, P., & Karpf, J. (2004). Using the social communication questionnaire to identify ‘autistic spectrum’ disorders associated with other genetic conditions. Autism, 8, 175–182.

    Article  PubMed  Google Scholar 

  • Jarrold, C., Boucher, J., & Russell, J. (1997). Language profiles in children with autism. Autism, 1, 57–76.

    Google Scholar 

  • Loesch, D. Z., Bui, Q. M., Grigsby, J., Butler, E., Epstein, J., Huggins, R. M., Taylor, A. K., & Hagerman, R. J. (2003). Effect of fragile X status categories and the fragile X mental retardation protein levels on executive functioning in males and females with fragile X. Neuropsychology, 17, 646–657.

    Article  PubMed  Google Scholar 

  • Lord, C., Rutter, M., & Le Couteur, A. (1994). Autism diagnostic interview-revised: A revised version of a diagnostic interview for carers of individuals with possible pervasive developmental disorders. Journal of Autism and Developmental Disorders, 24, 659–685.

    Article  PubMed  Google Scholar 

  • Mazzocco, M. M. M., Pennington, B. F., & Hagerman, R. J. (1994). Social cognition skills among females with fragile X. Journal of Autism and Developmental Disorders, 24, 473–485.

    Article  PubMed  Google Scholar 

  • Ozonoff, S., Pennington, B. F., & Rogers, S. J. (1991). Executive function deficits in high-functioning autistic individuals: Relationship to theory of mind. Journal of Child Psychology and Psychiatry, 32, 1081–1105.

    Article  PubMed  Google Scholar 

  • Perner, J., Frith, U., Leslie, A. M., & Leekam, S. (1989). Exploration of the autistic child’s theory of mind: Knowledge, belief and communication. Child Development, 60, 689–700.

    Article  Google Scholar 

  • Perner, J., & Lang, B. (2000). Theory of mind and executive function: Is there a developmental relationship. In S. Baron-Cohen, T. Tager-Flusberg, & D. Cohen (Eds.), Understanding other minds: Perspectives from developmental cognitive neuroscience (pp. 150–181). Oxford: Oxford University Press.

    Google Scholar 

  • Raven, J. C. (1965). Guide to using the coloured progressive matrices. London: Lewis.

    Google Scholar 

  • Russell, J. (1997). How executive disorders can bring about an inadequate theory of mind. In J. Russell (Ed.), Autism as an executive disorder (pp. 256–304). Oxford: Oxford University Press.

    Google Scholar 

  • Samson, D., Apperly, I. A., Kathirgamanathan, U., & Humphreys, G. W. (2005). Seeing it my way: A case of selective deficit in inhibiting self-perspective. Brain, 128, 1102–1111.

    Article  PubMed  Google Scholar 

  • Saxe, R., Carey, S., & Kanwisher, N. (2004). Understanding other minds: Linking developmental psychology and functional neuroimaging. Annual Review of Psychology, 55, 87–124.

    Article  PubMed  Google Scholar 

  • Slade, L., & Ruffman, T. (2005). How language does (and does not) relate to theory of mind: A longitudinal study of syntax, semantics, working memory and false belief. British Journal of Developmental Psychology, 23, 1–26.

    Article  Google Scholar 

  • Smith, M., Apperly, I. A., & White, V. (2003) False belief reasoning and the acquisition of relative clause sentences. Child Development, 74(6), 1709–1719.

    Article  PubMed  Google Scholar 

  • Sperber, D. (Ed.). (2000). Metarepresentations: A multidisciplinary perspective. New York: Oxford University Press.

    Google Scholar 

  • Sudhalter, V., & Belser, R. C. (2001). Conversational characteristics of children with fragile X syndrome: tangential language. American Journal of Medical Genetics, Sep;106(5), 389–400.

    Google Scholar 

  • Wellman, H. M., Baron-Cohen, S., Caswell, R., Gomez, J. C., Swettenham, J., Toye, E., & Lagattuta, K. (2002). Thought-bubbles help children with autism acquire an alternative to a theory of mind. Autism, 6, 343–363.

    Article  PubMed  Google Scholar 

  • Wimmer, H., & Perner, J. (1983). Beliefs about beliefs: representation and the constraining function of wrong beliefs in young children’s understanding of deception. Cognition, 13, 103–128.

    Article  PubMed  Google Scholar 

  • Yirmiya, N., Erel, O., Shaked, M., & Solomonica-Levi, D. (1998). Meta-analysis comparing theory of mind abilities of individuals with autism, individuals with mental retardation, and normally developing individuals. Psychological Bulletin, 124, 283–307.

    Article  PubMed  Google Scholar 

Download references

Acknowledgements

We would like to thank Lynne Zwink for her invaluable support, the families recruited through the Fragile X Society and the staff and pupils at Foxwood School, who generously gave their time to take part in this research.

Author information

Authors and Affiliations

Authors

Appendix A

Appendix A

Standard location change false belief task. Based upon the Sally-Anne task (Baron-Cohen et al., 1985), the participant is introduced to two play-mobile figures and is invited to give them names (for example James and Carol). The experimenter then narrates the following story: “This is James and this is Carol. James has just come into the house from playing football outside. He takes his ball and puts in into his toy box and goes to play upstairs”. The participant sees the play-mobile figure put his ball into a box and exit the area. The narrative is then continued. “While James is playing upstairs, Carol decides to move the ball. Carol takes the ball out of the box and puts it in the cupboard”. The participant sees the second play-mobile figure remove the ball from the box and put it into the cupboard, where it is out of sight. This second figure then exits the area. The participant is then asked two control questions about the original and the current location of the ball: Memory Question: “Where did James put his ball in the beginning?” and the Reality Question “Where is the ball now?” The first play-mobile character is then brought back into the area, and the participant is told “James is going to play outside again now and he wants his ball”. Then the participant is asked the Test Question, “Where will James look for his ball?” The participant scores one for a correct response or zero for an incorrect response on the test question.

Standard Deceptive box false belief task. In this variant of the deceptive box task (Perner, Frith, Leslie, & Leekam, 1989), the participant is shown a sealed egg carton and asked, “What do you think is inside here?” The participant answers “eggs” or “those” (pointing to the picture of the eggs on the box). The box is then opened up and the participant is shown that the box actually contains a pen. The participant is asked to name the pen to check that they know what it is. The pen is put back in the box and the box is closed. The participant is then asked, “In a minute X is going to come and work with me. (S)he hasn’t seen this box yet, or what’s inside it. When (s)he comes in, I am going to show her/him this box, closed just like this (participant is shown box again). I am going to ask her/him “X, What’s in this box?” The participant is then asked the Test Question “What will (s)he say?” and a Reality Question, “What is really inside the box?” The participant scores one for a correct response or zero for an incorrect response on the test question.

Rights and permissions

Reprints and permissions

About this article

Cite this article

Grant, C.M., Apperly, I. & Oliver, C. Is Theory of Mind Understanding Impaired in Males with Fragile X Syndrome?. J Abnorm Child Psychol 35, 17–28 (2007). https://doi.org/10.1007/s10802-006-9077-0

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s10802-006-9077-0

Keywords

Navigation