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Journal of Autism and Developmental Disorders

, Volume 45, Issue 4, pp 1042–1061 | Cite as

Use of Emotional Cues for Lexical Learning: A Comparison of Autism Spectrum Disorder and Fragile X Syndrome

  • Angela John Thurman
  • Andrea McDuffie
  • Sara T. Kover
  • Randi Hagerman
  • Marie Moore Channell
  • Ann Mastergeorge
  • Leonard Abbeduto
Original Paper

Abstract

The present study evaluated the ability of males with fragile X syndrome (FXS), nonsyndromic autism spectrum disorder (ASD), or typical development to learn new words by using as a cue to the intended referent an emotional reaction indicating a successful (excitement) or unsuccessful (disappointment) search for a novel object. Performance for all groups exceeded chance-levels in both search conditions. In the Successful Search condition, participants with nonsyndromic ASD performed similarly to participants with FXS after controlling for severity of ASD. In the Unsuccessful Search condition, participants with FXS performed significantly worse than participants with nonsyndromic ASD, after controlling for severity of ASD. Predictors of performance in both search conditions differed between the three groups. Theoretical and clinical implications are discussed.

Keywords

Fragile X syndrome Autism spectrum disorder Lexical learning Fast mapping Emotion 

Notes

Acknowledgments

This research was supported by Grant R01 HD054764 from the National Institute of Child Health and Human Development. We wish to thank the children and their families for their participation in this study. We also thank David Benjamin, Susan Harris, Beth Goodlin-Jones, Claire Hauser, Sara Lifson, Eileen Haebig, Ashley Oakes, and Cecilia Compton for assisting with data collection and Susen Schroeder for coordinating all study visits. Leonard Abbeduto has received financial support to develop and implement outcome measures for fragile X syndrome clinical trials from F. Hoffman-LaRoche, Ltd., Roche TCRC, Inc. and Neuren Pharmaceuticals Limited. Randi J. Hagerman has received funding from Novartis, Roche Pharmaceuticals, Curemark, Forest, and Seaside Therapeutics to carry out treatment studies in fragile X syndrome and autism. She has also consulted with Roche/Genetech and Novartis regarding treatment studies in fragile X syndrome. No other authors have financial disclosures to make.

References

  1. Abbeduto, L., Brady, N., & Kover, S. T. (2007). Language development and fragile X syndrome: Profiles, syndrome-specificity, and within-syndrome differences. Mental Retardation and Developmental Disabilities Research Reviews, 13, 36–46.CrossRefPubMedGoogle Scholar
  2. Abbeduto, L., Murphy, M. M., Cawthon, S. W., Richmond, E. K., Weissman, M. D., Karadottir, S., et al. (2003). Receptive language skills of adolescents and young adults with Down syndrome or fragile X syndrome. American Journal of Mental Retardation, 108, 146–160.CrossRefGoogle Scholar
  3. American Psychiatric Association. (2013). Diagnostic and statistical manual of mental disorders (5th ed.). Arlington, VA: American Psychiatric Publishing.CrossRefGoogle Scholar
  4. Bagni, C., & Oostra, B. A. (2013). Fragile X syndrome: From protein function to therapy. American Journal of Medical Genetics, Part A, 161, 2809–2821.CrossRefGoogle Scholar
  5. Baldwin, D. (1991). Infants’ contribution to the achievement of joint reference. Child Development, 62, 875–890.CrossRefPubMedGoogle Scholar
  6. Baldwin, D. (1993). Infants ability to consult the speaker for clues to word reference. Journal of Child Language, 20, 395–418.CrossRefPubMedGoogle Scholar
  7. Baldwin, D. A. (1995). Understanding the link between joint attention and language. In C. Moore & P. J. Dunham (Eds.), Joint attention: Its origins and role in development (pp. 130–158). Hillsdale, NJ: Lawrence Erlbaum Associates.Google Scholar
  8. Baldwin, D. A. (2000). Interpersonal understanding fuels knowledge acquisition. Current Directions in Psychological Science, 9, 40–45.CrossRefGoogle Scholar
  9. Bani Hani, H., Gonzalez-Barrero, A. M., & Nadig, A. S. (2013). Children’s referential understanding of novel words and parent labeling behaviors: Similarities across children with and without autism spectrum disorders. Journal of Child Language, 40, 971–1002.CrossRefGoogle Scholar
  10. Baron-Cohen, S., Baldwin, D., & Crowson, M. (1997). Do children with autism use the speaker’s direction of gaze strategy to crack the code of language? Child Development, 68, 48–57.CrossRefPubMedGoogle Scholar
  11. Bassell, G. J., & Warren, S. T. (2008). Fragile X syndrome: Loss of local mRNA regulation alters synaptic development and function. Neuron, 60, 201–214.CrossRefPubMedCentralPubMedGoogle Scholar
  12. Belmonte, M. K., & Bourgeron, T. (2006). Fragile X syndrome and autism at the intersection of genetic and neural networks. Nature Neuroscience, 9, 1221–1225.Google Scholar
  13. Berry-Kravis, E. M., Hessl, D., Rathmell, B., Zarevics, P., Cherubini, M., Walton-Bowen, K., et al. (2012). Effects of STX209 (arbaclofen) on neurobehavioral function in children and adults with fragile X syndrome: A randomized, controlled phase 2 trial. Science Translational Medicine, 4, 152ra127.Google Scholar
  14. Bishop, S. L., Hus, V., Duncan, A., Huerta, M., Gotham, K., Pickles, A., et al. (2013). Subcategories of restricted and repetitive behaviors in children with autism spectrum disorders. Journal of Autism and Developmental Disorders, 43, 1287–1297.CrossRefPubMedCentralPubMedGoogle Scholar
  15. Carey, S., & Bartlett, E. (1978). Acquiring a single new word. In Parents and reports on child language development (vol. 15, pp. 17–29). Stanford Univerity.Google Scholar
  16. Centers for Disease Control and Prevention. (2012). Prevalence of autism spectrum disorders—Autism and developmental disabilities monitoring network, 14 sites, United States, 2008. Morbidity and Mortality Weekly Report Surveillance Summary, 61, 1–19.Google Scholar
  17. Cordeiro, L., Ballinger, E., Hagerman, R., & Hessl, D. (2011). Clinical assessment of DSM-IV anxiety disorders in fragile X syndrome: Prevalence and characterization. Journal of Neurodevelopmental Disorders, 3, 57–67.CrossRefPubMedCentralPubMedGoogle Scholar
  18. 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(1), 144–150.CrossRefPubMedGoogle Scholar
  19. Cornish, K., Scerif, G., & Karmiloff-Smith, A. (2007). Tracing syndrome-specific trajectories of attention across the lifespan. Cortex, 43, 672–685.CrossRefPubMedGoogle Scholar
  20. Cornish, K. M., Turk, J., Wilding, J., Sudhalter, V., Munir, F., Kooy, F., et al. (2004). Annotation: Deconstructing the attention deficit in fragile X syndrome: A developmental neuropsychological approach. Journal of Child Psychology and Psychiatry, 45(6), 1042–1053.CrossRefPubMedGoogle Scholar
  21. Crawford, D. C., Acuña, J. M., & Sherman, S. L. (2001). FMR1 and the fragile X syndrome: Human genome epidemiology review. Genetics in Medicine, 3, 359–371.CrossRefPubMedGoogle Scholar
  22. DeGiacomo, A., & Fombonne, E. (1998). Parental recognition of developmental disabilities in autism. European Child and Adolescent Psychiatry, 7, 131–136.CrossRefGoogle Scholar
  23. Dykens, E. M., Hodapp, R. M., & Leckman, J. F. (1987). Strengths and weaknesses in the intellectual functioning of males with fragile X syndrome. American Journal of Mental Deficiency, 92, 234–236.PubMedGoogle Scholar
  24. Ellis Weismer, S., Lord, C., & Esler, A. (2010). Early language patterns of toddlers on the autism spectrum compared to toddlers with developmental delay. Journal of Autism and Developmental Disorders, 40, 1259–1273.CrossRefPubMedGoogle Scholar
  25. Esbensen, A. J., Rojahn, J., Aman, M. G., & Ruedrich, S. (2003). Reliability and validity of an assessment instrument for anxiety, depression, and mood among individuals with mental retardation. Journal of Autism and Developmental Disorders, 33, 617–629.CrossRefPubMedGoogle Scholar
  26. Frick, R. W. (1995). Accepting the null hypothesis. Memory & Cognition, 23, 132–138.CrossRefGoogle Scholar
  27. Gliga, T., Elsabbagh, M., Hudry, K., Charman, T., & Johnson, M. H. (2012). Gaze following, gaze reading, and word learning in children at risk for autism. Child Development, 83, 926–938.CrossRefPubMedGoogle Scholar
  28. Gotham, K., Pickles, A., & Lord, C. (2009). Standardizing ADOS scores of a measure of severity in autism spectrum disorders. Journal of Autism and Developmental Disorders, 39, 693–705.CrossRefPubMedCentralPubMedGoogle Scholar
  29. Gross, A. L., & Ballif, B. (1991). Children’s understanding of emotion from facial expressions and situations: A review. Developmental Review, 11, 368–398.CrossRefGoogle Scholar
  30. Hagerman, R. J., Rivera, S. M., & Hagerman, P. J. (2008). The fragile X family of disorders: A model for autism and targeted treatments. Current Pediatric Reviews, 4, 40–52.Google Scholar
  31. Harris, S., Hessl, D., Goodlin-Jones, B., Ferranti, J., Bacalman, S., Barbato, I., et al. (2008). Autism profiles of males with fragile X syndrome. American Journal on Intellectual and Developmental Disabilities, 113, 427–438.CrossRefGoogle Scholar
  32. Harris, M., Jones, D., Brookes, S., & Grant, J. (1986). Relations between the non-verbal context of maternal speech and rate of language development. British Journal of Developmental Psychology, 4, 261–268.CrossRefGoogle Scholar
  33. Hessl, D., Nguyen, D. V., Green, C., Chavez, A., Tassone, F., Hagerman, R. J., et al. (2009). A solution to limitations of cognitive testing in children with intellectual disabilities: The case of fragile X syndrome. Journal of Neurodevelopmental Disorders, 1, 33–45.CrossRefPubMedCentralPubMedGoogle Scholar
  34. Hodapp, R. M., Leckman, J. F., Dykens, E. M., Sparrow, S. S., Zelinsky, D. G., & Ort, S. I. (1992). K-ABC profiles in children with fragile X syndrome, Down syndrome, and nonspecific mental retardation. American Journal on Mental Retardation, 97, 39–46.PubMedGoogle Scholar
  35. Hollich, G. J., Hirsh-Pasek, K., & Golinkoff, R. M. (2000). Monographs of the Society for Research in Child Development, 65, 1–135.CrossRefGoogle Scholar
  36. Horst, J. S., & Samuelson, L. K. (2008). Fast mapping but poor retention by 24-month-old infants. Infancy, 13, 128–157.Google Scholar
  37. Hus, V., Gotham, K., & Lord, C. (in press). Standardizing ADOS domains scores: Separating severity of social affect and restricted and repetitive behaviors. Journal of Autism and Developmental Disorders. Google Scholar
  38. Jang, J., Dixon, D. R., Tarbox, J., & Granpeesheh, D. (2011). Symptom severity and challenging behavior in children with ASD. Research in Autism Spectrum Disorders, 5, 1028–1032.CrossRefGoogle Scholar
  39. Kau, A. S. M., Reider, E. E., Payne, L., Meyer, W. A., & Freund, L. (2000). Early behavioral signs of psychiatric phenotypes in fragile X syndrome. American Journal on Mental Retardation, 105, 266–299.CrossRefGoogle Scholar
  40. Kemper, M. B., Hagerman, R. J., & Altshul-Stark, D. (1988). Cognitive profiles of boys with fragile X syndrome. American Journal of Medical Genetics, 30, 191–200.CrossRefPubMedGoogle Scholar
  41. Klusek, J., Martin, G. E., & Losh, M. (2014). Consistency between research and clinical diagnoses of autism among boys and girls with fragile X syndrome. Journal of Intellectual Disability Research, 10, 940–952.Google Scholar
  42. Kover, S. T., & Atwood, A. K. (2013). Establishing equivalence: Methodological progress in group-matching design and analysis. American Journal on Intellectual and Developmental Disabilities, 118, 3–15.CrossRefPubMedGoogle Scholar
  43. Kover, S. T., McDuffie, A. S., Hagerman, R. J., & Abbeduto, L. (2013). Receptive vocabulary in boys with autism spectrum disorder: Cross-sectional developmental trajectories. Journal of Autism and Developmental Disorders, 43, 2696–2709.CrossRefPubMedCentralPubMedGoogle Scholar
  44. Lanfranchi, S., Cornoldi, C., Drigo, S., & Vianello, R. (2009). Working memory in individuals with fragile X syndrome. Child Neuropsychology, 15(2), 105–119.CrossRefPubMedGoogle Scholar
  45. 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.CrossRefPubMedGoogle Scholar
  46. Lord, C., Rutter, M., DiLavore, P. C., & Risi, S. (2007). Autism diagnostic observation schedule. Los Angeles: Western Psychological Services.Google Scholar
  47. Luyster, R., & Lord, C. (2009). Word learning in children with autism spectrum disorders. Developmental Psychology, 45, 1774–1786.CrossRefPubMedCentralPubMedGoogle Scholar
  48. Marshall, C. R., Noor, A., Vincent, J. B., Lionel, A. C., Feuk, L., Skaug, J., et al. (2008). Structural variation of chromosomes in autism spectrum disorder. American Journal of Human Genetics, 82, 477–488.Google Scholar
  49. Mazzocco, M. M. M. (2000). Advances in research on the fragile X syndrome. Mental Retardation and Developmental Disabilities Research Review, 6, 96–106.CrossRefGoogle Scholar
  50. McDuffie, A., Kover, S. T., Hagerman, R., & Abbeduto, L. (2013). Investigating word learning in fragile X syndrome: A fast-mapping study. Journal of Autism ad Developmental Disorders, 43, 1676–1691.CrossRefGoogle Scholar
  51. McDuffie, A., Thurman, A. J., Hagerman, R. J., & Abbeduto, L. (in press). Symptoms of autism in males with fragile X syndrome: A comparison to nonsyndromic ASD using current ADI-R scores. Journal of Autism and Developmental Disorders. Google Scholar
  52. McDuffie, A., Yoder, P., & Stone, W. (2006). Fast-mapping in young children with autism spectrum disorders. First Language, 26, 421–436.CrossRefGoogle Scholar
  53. Mervis, C. B., & John, A. E. (2008). Vocabulary abilities of children with Williams syndrome: Strengths, weaknesses, and relation to visuospatial construction ability. Journal of Speech, Language, and Hearing Research, 51, 967–982.CrossRefPubMedCentralPubMedGoogle Scholar
  54. Mervis, C. B., & Klein-Tasman, B. P. (2004). Methodological issues in group-matching design: α levels for control variable comparisons and measurement characteristics of control and target variables. Journal of Autism and Developmental Disorders, 34, 7–17.CrossRefPubMedGoogle Scholar
  55. Mundy, P., & Neal, R. (2001). Neural plasticity, joint attention, and a transactional social-orienting model of autism. International Review of Mental Retardation, 23, 139–168.CrossRefGoogle Scholar
  56. Munir, F., Cornish, K. M., & Wilding, J. (2000). A neuropsychological profile of attention deficits in young males with fragile X syndrome. Neuropsychologia, 38, 1261–1270.CrossRefPubMedGoogle Scholar
  57. Oostra, B. A., & Willemson, R. (2003). A fragile X balance: FMR1 expression levels. Human Molecular Genetics, 12, 249–257.CrossRefGoogle Scholar
  58. 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.CrossRefPubMedGoogle Scholar
  59. Pierpont, E. I., Richmond, E. K., Abbeduto, L., Kover, S. T., & Brown, W. T. (2011). Contributions of phonological and verbal working memory to language in adolescents with fragile X syndrome. Journal of Neurodevelopmental Disorders, 3, 335–347.CrossRefPubMedCentralPubMedGoogle Scholar
  60. Preissler, M. A., & Carey, S. (2005). The role of inferences about referential intent in word learning: Evidence from autism. Cognition, 97, 13–23.CrossRefGoogle Scholar
  61. Price, J., Roberts, J., Vandergrift, N., & Martin, G. (2007). Language comprehension in boys with fragile X syndrome. Journal of Intellectual Disability Research, 51, 318–326.CrossRefPubMedGoogle Scholar
  62. Remington, A., Swettenham, J., Campbell, R., & Coleman, M. (2009). Selective attention and perceptual load in autism spectrum disorder. Psychological Science, 20, 1388–1393.CrossRefPubMedGoogle Scholar
  63. Rice, M. L., Buhr, J. C., & Nemeth, M. (1990). Fast-mapping word-learning abilities of language-delayed preschoolers. Journal of Speech and Hearing Disorders, 55(1), 33–42.Google Scholar
  64. Risi, S., Lord, C., Gotham, K., Corsello, C., Chrysler, C., Szatmari, P., et al. (2006). Combining information from multiple sources in the diagnosis of autism spectrum disorders. Journal of the American Academy of Child and Adolescent Psychiatry, 45, 1094–1103.CrossRefPubMedGoogle Scholar
  65. Roberts, J. E., Mirrett, P., & Burchinal, M. (2001). Receptive and expressive communication development of young males with fragile X syndrome. American Journal on Mental Retardation, 106, 216–230.CrossRefPubMedGoogle Scholar
  66. Roid, G. H., & Miller, L. J. (1997). Leiter International Performance Scale-revised. Wood Dale, IL: Stoelting Company.Google Scholar
  67. Rutter, M., Bailey, A., Berument, S. K., Lord, C., & Pickles, A. (2003a). Social Communication Questionnaire (SCQ). Los Angeles: Western Psychological Services.Google Scholar
  68. Rutter, M., Le Couteur, A., & Lord, C. (2003b). Autism Diagnostic Interview-revised. Los Angeles: Western Psychological Services.Google Scholar
  69. Ryland, H. K., Hysing, M., Posserud, M. B., Gillberg, C., & Lundervold, A. J. (2012). Autistic features in school age children: IQ and gender effects in a population-based cohort. Research in Autism Spectrum Disorders, 8, 266–274.CrossRefGoogle Scholar
  70. Scerif, G., Longhi, E., Cole, V., Karmiloff-Smith, A., & Cornish, K. (2012). Attention across modalities as a longitudinal predictor of early outcomes: The case of fragile X syndrome. Journal of Child Psychology and Psychiatry, 53, 614–650.CrossRefGoogle Scholar
  71. Seltzer, M. M., Abbeduto, L., Krauss, M. W., Greenburg, J., & Swe, A. (2004). Comparison groups in autism family research: Down syndrome, fragile X syndrome, and schizophrenia. Journal of Autism and Developmental Disorders, 34, 41–48.CrossRefPubMedGoogle Scholar
  72. Serlin, R. C., & Harwell, M. R. (2004). More powerful tests of predictor subtests in regression analysis under nonnormality. Psychological Methods, 9, 492–509.CrossRefPubMedGoogle Scholar
  73. Spence, S. J., & Schneider, M. T. (2009). The role of epilepsy and epileptiform EEGs in autism spectrum disorders. Pediatric Research, 65, 599–606.CrossRefPubMedCentralPubMedGoogle Scholar
  74. Tager-Flusberg, H., Rogers, S., Cooper, J., Landa, R., Lord, C., Paul, R., et al. (2009). Defining spoken language benchmarks and selecting measures of expressive language development for young children with autism spectrum disorders. Journal of Speech, Language, and Hearing Research, 52, 643–652.CrossRefPubMedCentralPubMedGoogle Scholar
  75. Thurman, A. J., McDuffie, A., Hagerman, R. J., & Abbeduto, L. (2014). Psychiatric symptoms in boys with fragile X syndrome: A comparison with nonsyndromic autism spectrum disorder. Research in Developmental Disabilities, 35, 1072–1086.Google Scholar
  76. Tomasello, M. (2001). Cultural transmission: A view from chimpanzees and human infants. Journal of Cross-Cultural Psychology, 32, 135–146.CrossRefGoogle Scholar
  77. Tomasello, M., & Barton, M. E. (1994). Learning words in nonostensive contexts. Developmental Psychology, 30, 639–650.CrossRefGoogle Scholar
  78. Tomasello, M., & Farrer, M. J. (1986). Joint attention and early language. Child Development, 57, 1454–1463.CrossRefPubMedGoogle Scholar
  79. Tomasello, M., Strosberg, R., & Akhtar, N. (1996). Eighteen-month-old children learn words in non-ostensive contexts. Journal of Child Language, 23, 157–176.CrossRefPubMedGoogle Scholar
  80. Volden, J., Smith, I. M., Szatmari, P., Bryson, S., Fombonne, E., Mirenda, P., et al. (2011). Using the preschool language scale, fourth edition to characterize language in preschoolers with ASD. American Journal of Speech-Language Pathology, 20, 200–208.CrossRefPubMedGoogle Scholar
  81. Wolff, J. J., Bodfish, J. W., Hazlett, H. C., Lightbody, A. A., Reiss, A. L., & Piven, J. (2012). Evidence of a distinct behavioral phenotype in young boys with fragile X syndrome and autism. Journal of the American Academy of Child and Adolescent Psychiatry, 51, 1324–1332.CrossRefPubMedCentralPubMedGoogle Scholar
  82. Woodward, A. L., Markman, E. M., & Fitzsimmons, C. M. (1994). Rapid word learning in 13- and 18-month-olds. Developmental Psychology, 30, 553–566.CrossRefGoogle Scholar

Copyright information

© Springer Science+Business Media New York 2014

Authors and Affiliations

  • Angela John Thurman
    • 1
    • 2
  • Andrea McDuffie
    • 1
    • 2
  • Sara T. Kover
    • 3
  • Randi Hagerman
    • 2
    • 4
  • Marie Moore Channell
    • 1
    • 2
  • Ann Mastergeorge
    • 5
  • Leonard Abbeduto
    • 1
    • 2
  1. 1.MIND InstituteUniversity of California DavisSacramentoUSA
  2. 2.Department of Psychiatry and Behavioral SciencesUniversity of California DavisDavisUSA
  3. 3.Waisman CenterUniversity of Wisconsin-MadisonMadisonUSA
  4. 4.Department of PediatricsUniversity of California DavisDavisUSA
  5. 5.University of ArizonaTucsonUSA

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