Abstract
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.
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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.
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.
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. https://doi.org/10.1352/0895-8017(2004)109%3c237:Pwacim%3e2.0.Co;2.
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.
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.
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.
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.
Broussard, C. A. (2010). Research regarding low-income single mothers' mental and physical health: A decade in review. Journal of Poverty,14, 433–451.
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.
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.
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. https://doi.org/10.1002/mrdd.20008.
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. https://doi.org/10.1007/s12311-016-0809-6.
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.
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. https://doi.org/10.1037/0002-9432.74.1.14.
Hagerman, P. J., & Hagerman, R. J. (2004). The fragile-X premutation: A maturing perspective. American Journal of Human Genetics,74, 805–816.
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. https://doi.org/10.1016/S1474-4422(13)70125-X.
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.
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.
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. https://doi.org/10.1002/ajmg.10216.
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.
Hoffman, L. (2015). Longitudinal analysis: Modeling within-person fluctuation and change. New York, NY: Routledge Academic.
Hunter, J. E., Rohr, J. K., & Sherman, S. L. (2010). Co-occuring diagnoses amoing FMR1 premutation allele carriers. Clinical Genetics,77, 374–381.
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.
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. https://doi.org/10.1097/00004703-200308000-00008.
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. https://doi.org/10.1111/jir.12121.
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.
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.
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.
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.
McNair, D. M., Lorr, M., & Droppleman, L. F. (1971). Manual for the profile of mood States. San Diego, CA: Educational and Industrial Testing Services.
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.
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.
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.
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.
SAS Institute Inc. (2013). SAS 9.4. Cary, NC: SAS Institute Inc.
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. https://doi.org/10.1037/0882-7974.10.1.64.
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.
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.
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. https://doi.org/10.1093/humrep/deh635.
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.
U.S. Census Bureau. (2016). Current Population Survey Annual Social and Economic Supplement. Retrieved from https://www.census.gov/hhes/families.
U.S. Census Bureau. (2018). Decennial Census 1960 and Current Population Survey, Annual Social and Economic Supplements 1968 to 2018.
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.
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.
Wechsler, D. (1997). Wechsler adult intelligence scale-III (3rd ed.). San Antonio, TX: Harcourt Assessment.
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.
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.
Acknowledgments
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.
Funding
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).
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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.
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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.
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Informed consent was obtained from all individual participants included in the study. Mothers gave informed consent for themselves and their children.
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Fielding-Gebhardt, H., Warren, S.F. & Brady, N.C. Child Challenging Behavior Influences Maternal Mental Health and Relationship Quality Over Time in Fragile X Syndrome. J Autism Dev Disord 50, 779–797 (2020). https://doi.org/10.1007/s10803-019-04308-2
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DOI: https://doi.org/10.1007/s10803-019-04308-2