Child Challenging Behavior Influences Maternal Mental Health and Relationship Quality Over Time in Fragile X Syndrome

  • Heather Fielding-GebhardtEmail author
  • Steven F. Warren
  • Nancy C. Brady
Original Paper


Parenting children with neurodevelopmental disabilities is often challenging. Biological mothers of children with Fragile X Syndrome (FXS) may be susceptible to increased risk of mental health problems. This study examined the longitudinal relationships between maternal mental health, child challenging behaviors, and mother–child relationship quality in children and adolescents with FXS. Fifty-five mother–child dyads were followed from childhood into adolescence. The findings suggest that child challenging behaviors, maternal mental health, and mother–child relationship quality were stable during that period. Additionally, elevated levels of child challenging behaviors negatively impacted maternal mental health. Finally, child challenging behaviors, in combination with maternal mental health, influenced mother–child relationship quality. Clinical implications are discussed.


Fragile X Mental health Challenging behaviors Relationship quality 



The authors would like to thank Shelley Bredin-Oja from the Fragile X Lab at the University of Kansas for her wonderful data management, Katie Schneider for assistance with an earlier version of this study, and Lesa Hoffman for her invaluable knowledge of statistical analyses. Finally, we thank the families for their continued participation and support in our ongoing study.

Author Contributions

All authors contributed to the study conception and design. Material preparation, data collection and analysis were performed by Heather Fielding-Gebhardt. The first draft of the manuscript was written by Heather Fielding-Gebhardt, and all authors commented on previous versions of the manuscript. All authors read and approved the final manuscript.


This study is supported by the following NICHD Grants: P30 HD00310, P30 HD02538, and R01 HD084563 (PIs REDACTED). The first author is supported by NICDC T32 DC000052 (PI: Mabel Rice).

Compliance with Ethical Standards

Conflicts of interest

The authors declare that they have no conflicts of interest.

Ethical Approval

All procedures performed in this study involving human participants were in accordance with the ethical standards of the institutional review board (University of Kansas IRB, study reference number 17261) and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.

Informed Consent

Informed consent was obtained from all individual participants included in the study. Mothers gave informed consent for themselves and their children.


  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.CrossRefGoogle Scholar
  2. Abbeduto, L., McDuffie, A., & Thurman, A. J. (2014). The fragile X syndrome-autism comorbidity: What do we really know? Frontiers in Genetics,5, 3–10.CrossRefGoogle Scholar
  3. Abbeduto, L., Seltzer, M. M., Shattuck, P., Krauss, M. W., Orsmond, G. I., & Murphy, M. M. (2004). Psychological well-being and coping in mothers of youths with autism, down syndrome, or fragile X syndrome. American Journal on Mental Retardation,109(3), 237–254.;2.CrossRefPubMedGoogle Scholar
  4. Achenbach, T. M., & Rescorla, L. A. (2001). Manial for the ASEBA school-age forms and profiles. Burlington, VT: University of Vermont, Research Center for Children, Youth, and Families.Google Scholar
  5. Bailey, D. B., Sideris, J., Roberts, J. E., & Hatton, D. D. (2008). Child and genetic variables associated with maternal adaptation to fragile X syndrome: A multidimensional analysis. American Journal of Medical Genetics Part A,146A, 720–729.CrossRefGoogle Scholar
  6. Bengtson, V. L., & Schrader, S. (1982). Parent-child relations. In D. Mangen & W. Peterson (Eds.), Research instruments in social gerontology (Vol. 2, pp. 115–186). Minneapolis, MN: University of Minnesota Press.Google Scholar
  7. Brady, N., Warren, S. F., Fleming, K., Keller, J., & Sterling, A. (2014). Effect of sustained maternal responsivity on later vocabulary development in children with fragile X syndrome. Journal of Speech, Language, and Hearing Research,57(1), 212–226.CrossRefGoogle Scholar
  8. Broussard, C. A. (2010). Research regarding low-income single mothers' mental and physical health: A decade in review. Journal of Poverty,14, 433–451.CrossRefGoogle Scholar
  9. Cohen, D., Pichard, N., Tordjman, S., Baumann, C., Burglen, L., Excoffier, E., et al. (2005). Specific genetic disorders and autism: Clinical contribution towards their identification. Journal of Autism and Developmental Disorders,35(1), 103–116.CrossRefGoogle Scholar
  10. Curran, S. L., Andrykowski, M. A., & Studts, J. L. (1995). Short form of the profile of mood states (POMS-SF): Psychometric information. Psychological Assessment,7(1), 80–83.CrossRefGoogle Scholar
  11. Darnell, J. C., Warren, S. T., & Darnell, R. B. (2004). The fragile X mental retardation protein, FMRP, recognizes G-quartets. Mental Retardation and Developmental Disabilities Research Reviews,10(1), 49–52. Scholar
  12. Debrey, S. M., Leehey, M. A., Klepitskaya, O., Filley, C. M., Shah, R. C., Kluger, B., et al. (2016). Clinical phenotype of adult fragile x gray zone allele carriers: A case series. The Cerebellum,15(5), 623–631. Scholar
  13. Franke, P., Maier, W., Hautzinger, M., Weiffenbach, O., Gansicke, M., Iwers, B., et al. (1996). Fragile-X carrier females: evidence for a distinct psychopathological phenotype? American Journal of Medical Genetics,64, 334–339.CrossRefGoogle Scholar
  14. Greenberg, J. S., Seltzer, M. M., Krauss, M. W., Chou, R. J.-A., & Hong, J. (2004). The effect of quality of relationship between mothers and adult children with schizophrenia, autism, or down syndrome on maternal well-being: the mediating role of optimism. American Journal of Orthopsychiatry,74(1), 14–25. Scholar
  15. Hagerman, P. J., & Hagerman, R. J. (2004). The fragile-X premutation: A maturing perspective. American Journal of Human Genetics,74, 805–816.CrossRefGoogle Scholar
  16. Hagerman, R. J., & Hagerman, P. J. (2013). Advances in clinical and molecular understanding of theFMR1premutation and fragile X-associated tremor/ataxia syndrome. Lancet Neurology,12(8), 786–798. Scholar
  17. Hardiman, R. L., & McGill, P. (2018). How common are challenging behaviors amongst individuals with fragile X syndrome? A systematic review. Research in Developmental Disabilities,76, 99–109.CrossRefGoogle Scholar
  18. Hartley, S. L., Barker, E. T., Baker, J. K., Seltzer, M. M., & Greenberg, J. S. (2012). Marital satisfaction and life circumstances of grown children with autism across 7 years. Journal of Family Psychology,26(5), 688–697.CrossRefGoogle Scholar
  19. Hatton, D. D., Hooper, S. R., Bailey, D. B., Skinner, M. L., Sulliva, K. M., & Wheeler, A. (2002). Problem behavior in boys with fragile X syndrome. American Journal of Medical Genetics,108, 105–116. Scholar
  20. Hauser, C. T., Kover, S. T., & Abbeduto, L. (2014). Maternal well-being and child behavior in families with fragile X syndrome. Research in Developmental Disabilities,35, 2477–2486.CrossRefGoogle Scholar
  21. Hoffman, L. (2015). Longitudinal analysis: Modeling within-person fluctuation and change. New York, NY: Routledge Academic.Google Scholar
  22. Hunter, J. E., Rohr, J. K., & Sherman, S. L. (2010). Co-occuring diagnoses amoing FMR1 premutation allele carriers. Clinical Genetics,77, 374–381.CrossRefGoogle Scholar
  23. Jess, M., Hastings, R. P., & Totsika, V. (2017). The construct of maternal positivity in mothers of children with intellectual disability. Journal of Intellectual Disability Research,61(10), 928–938.CrossRefGoogle Scholar
  24. Johnston, L. C., Hessl, L. D., Blasey, L. C., Eliez, L. S., Erba, L. H., Dyer-Friedman, L. J., et al. (2003). Factors associated with parenting stress in mothers of children with fragile X syndrome. Journal of Developmental and Behavioral Pediatrics,24(4), 267–275. Scholar
  25. 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,58(10), 940–952. Scholar
  26. Lewinsohn, P. M., Seeley, J. R., Roberts, R. E., & Allen, N. B. (1997). Center for edipemiologic studies depression scale (CES-D) as a screening instrument for depression among community-residing older adults. Psychology and Aging,12(2), 277–287.CrossRefGoogle Scholar
  27. Lewis, P., Abbeduto, L., Murphy, M. M., Richmond, E., Giles, N., Bruno, L., et al. (2006a). Psychological well-being of mothers of youth with fragile X syndrome: syndrome specificity and within-syndrome variability. Journal of Intellectual Disability Research,50(2), 894–904.CrossRefGoogle Scholar
  28. Lewis, P., Abbeduto, L., Murphy, M. M., Richmond, E., Giles, N., Bruno, L., et al. (2006b). Cognitive, language and social-cognitive skills of individuals with fragile X syndrome with and without autism. Journal of Intellectual Disability Research,50(7), 532–545.CrossRefGoogle Scholar
  29. Mazzocco, M. M. M., Kates, W. R., Baumgardner, T. L., Freund, L. S., & Reiss, A. L. (1997). Autistic behaviors among girls with fragile X syndrome. Journal of Autism and Developmental Disorders,27(4), 415–435.CrossRefGoogle Scholar
  30. McNair, D. M., Lorr, M., & Droppleman, L. F. (1971). Manual for the profile of mood States. San Diego, CA: Educational and Industrial Testing Services.Google Scholar
  31. Orsmond, G. I., Seltzer, M. M., Greenberg, J. S., & Krauss, M. W. (2006). Mother-child relationship quality among adolescents and adults with autism. American Journal on Mental Retardation,111(2), 121–137.CrossRefGoogle Scholar
  32. Radloff, L. S. (1977). The CES-D Scale: A self report depression scale for research in the general population. Applied Psychological Measurements,1, 385–401.CrossRefGoogle Scholar
  33. Rinehart, N. J., Cornish, K. M., & Tonge, B. J. (2010). Gender differences in neurodevelopmental disorders: Autism and fragile X syndrome. In J. C. Neill & J. Kulkarni (Eds.), Biological basis of sex differences in psychopharmacology. Berlin: Springer.Google Scholar
  34. Roberts, J. E., Bailey, D. B., Mankowski, J., Ford, A., Sideris, J., Weisenfeld, L. A., et al. (2008). Mood and anxiety disorders in females with the FMR1 premutation. American Journal of Medical Genetics Part B,150B, 130–139.CrossRefGoogle Scholar
  35. SAS Institute Inc. (2013). SAS 9.4. Cary, NC: SAS Institute Inc.Google Scholar
  36. Seltzer, M. M., Greenberg, J. S., & Krauss, M. W. (1995). A comparison of coping strategies of aging mothers of adults with mental illness or mental retardation. Psychology and Aging,10(1), 64–75. Scholar
  37. Smith, L. E., Greenberg, J. S., Seltzer, M. M., & Hong, J. (2008). Symptoms and behavior problems of adolescents and adults with autism: Effects of mother-child relationship quality, warmth, and praise. American Journal on Mental Retardation,113(5), 387–402.CrossRefGoogle Scholar
  38. Stack, R. J., & Meredith, A. (2018). The impact of financial hardship on single parents: An exploration of the journey from social distress to seeking help. Journal of Family and Economic Issues,39(2), 233–242.CrossRefGoogle Scholar
  39. Sullivan, A. K., Marcus, M., Epstein, M. P., Allen, E. G., Anido, A. E., Paquin, J. J., et al. (2005). Association of FMR1 repeat size with ovarian dysfunction. Human Reproduction,20(2), 402–412. Scholar
  40. Taylor, J. L., & Seltzer, M. M. (2011). Changes in the mother-child relationship during the transition to adulthood for youth with autism spectrum disorders. Journal of Autism and Developmental Disorders,41(10), 1397–1410.CrossRefGoogle Scholar
  41. U.S. Census Bureau. (2016). Current Population Survey Annual Social and Economic Supplement. Retrieved from
  42. U.S. Census Bureau. (2018). Decennial Census 1960 and Current Population Survey, Annual Social and Economic Supplements 1968 to 2018.Google Scholar
  43. 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(5), 905–914.CrossRefGoogle Scholar
  44. Warren, S. F., Brady, N., Sterling, A., Fleming, K., & Marquis, J. (2010). Maternal responsivity predicts language development in young children with fragile X syndrome. American Journal on Intellectual and Developmental Disabilities,115(1), 54–75.CrossRefGoogle Scholar
  45. Wechsler, D. (1997). Wechsler adult intelligence scale-III (3rd ed.). San Antonio, TX: Harcourt Assessment.Google Scholar
  46. Wheeler, A., Bailey, D. B., Berry-Kravis, E., Greenberg, J. S., Losh, M., Mailick, M., et al. (2014). Associated features in females with an FMR1 premutation. Journal of Neurodevelopmental Disorders,6(1), 30.CrossRefGoogle Scholar
  47. Zeedyk, S. M., & Blacher, J. (2017). Longitudinal correlates of maternal depression among mothers of children with or without intellectual disability. American Journal of Intellectual and Developmental Disabilities,122(5), 374–391.CrossRefGoogle Scholar

Copyright information

© Springer Science+Business Media, LLC, part of Springer Nature 2019

Authors and Affiliations

  • Heather Fielding-Gebhardt
    • 1
    Email author
  • Steven F. Warren
    • 2
  • Nancy C. Brady
    • 2
  1. 1.Child Language Doctoral ProgramUniversity of KansasLawrenceUSA
  2. 2.Department of Speech-Language-Hearing: Sciences and DisordersUniversity of KansasLawrenceUSA

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