Differences in Cognition and Behaviour in Multiplex and Simplex Autism: Does Prior Experience Raising a Child with Autism Matter?
- 28 Downloads
Previous research has found multiplex (MPX) children have an advantage in cognition compared to simplex (SPX) children. However, MPX parent’s previous experience with older diagnosed siblings has not been considered. We used a large database sample to investigate the MPX advantage and contribution of birth order. Children from the Autism Genetic Resource Exchange (AGRE) were stratified into first- (MPX1, n = 152) and second-affected MPX (MPX2, n = 143), SPX (n = 111), and only-child SPX (SPXOC, n = 23) groups. Both MPX groups had higher cognitive scores compared to SPX groups, with no differences between MPX1 and MPX2 groups. No differences were found for autism symptoms or adaptive behaviour. These results suggest parent experience due to birth order is an unlikely contributor to the MPX cognitive advantage.
KeywordsAutism spectrum disorder Multiplex Simplex Parent experience Cognition
LPL is funded by the Cooperative Research Centre for Living with Autism (Autism CRC). The authors are also extremely grateful to all the study participants and their families for contributing to the project. We acknowledge support from the Autism Genetic Resource Exchange (AGRE) and Autism Speaks. We gratefully acknowledge the resources provided by AGRE and the participating AGRE families. The Autism Genetic Resource Exchange (AGRE) is a Program of Autism Speaks.
DB conceived and designed the study under the supervision of CD and LPL. DB cleaned and analysed the data. The manuscript was drafted by DB under the supervision of CD and LPL. LPL and CD critically reviewed the manuscript and provided feedback. All authors read and approved the final manuscript.
Compliance with Ethical Standards
Conflict of interest
The authors declare no conflict of interest.
This article does not contain any studies with human participants performed by any of the authors. Ethical approval for the analysis of de-identified human data was obtained from La Trobe University Human Research Ethics Committee.
- Autism Genetic Resource Exchange. (2017). Diagnostic Information. http://research.agre.org/program/diag.cfm.
- Campbell, D. B., Sutcliffe, J. S., Ebert, P. J., Militerni, R., Bravaccio, C., Trillo, S., et al. (2006). A genetic variant that disrupts MET transcription is associated with autism. Proceedings of the National Academy of Sciences of the United States, 103(45), 16834. https://doi.org/10.1073/pnas.0605296103.CrossRefGoogle Scholar
- Christensen, D. L., Baio, J., Van Naarden Braun, K., Bilder, D., Charles, J., Constantino, J. N., et al. (2016). Prevalence and characteristics of Autism Spectrum Disorder among children Aged 8 Years—Autism and Developmental Disabilities Monitoring Network, 11 sites, United States, 2012. MMWR Surveillance Summaries, 65(3), 1–23. https://doi.org/10.15585/mmwr.ss6503a1.CrossRefGoogle Scholar
- Cuccaro, M. L., Shao, Y., Bass, M. P., Abramson, R. K., Ravan, S. A., Wright, H. H., et al. (2003). Behavioral comparisons in Autistic individuals from multiplex and singleton families. Journal of Autism and Developmental Disorders, 33(1), 87–91. https://doi.org/10.1023/a:1022286622534.CrossRefGoogle Scholar
- Davis, J. M., Keeney, J. G., Sikela, J. M., & Hepburn, S. (2013). Mode of genetic inheritance modifies the association of head circumference and autism-related symptoms: A cross-sectional study (research article). PLoS ONE, 8(9), e74940. https://doi.org/10.1371/journal.pone.0074940.CrossRefGoogle Scholar
- De la Marche, W., Noens, I., Kuppens, S., Spilt, J., Boets, B., & Steyaert, J. (2015). Measuring quantitative autism traits in families: Informant effect or intergenerational transmission? European Child and Adolescent Psychiatry, 24(4), 385–395. https://doi.org/10.1007/s00787-014-0586-z.CrossRefGoogle Scholar
- Donaldson, C. K., Stauder, J. E. A., & Donkers, F. C. L. (2017). Increased sensory processing atypicalities in parents of multiplex ASD families versus typically developing and simplex ASD families. Journal of Autism and Developmental Disorders, 47(3), 535–548. https://doi.org/10.1007/s10803-016-2888-0.CrossRefGoogle Scholar
- Hu, V. W., Sarachana, T., Kim, K. S., Nguyen, A., Kulkarni, S., Steinberg, M. E., et al. (2009). Gene expression profiling differentiates autism case–controls and phenotypic variants of autism spectrum disorders: Evidence for circadian rhythm dysfunction in severe autism. Autism Research, 2(2), 78–97. https://doi.org/10.1002/aur.73.CrossRefGoogle Scholar
- Lord, C. (2012). Autism diagnostic observation schedule: ADOS-2. Los Angeles: Western Psychological Services.Google Scholar
- Lord, C., Risi, S., Lambrecht, L., Cook, E. H., Jr., Leventhal, B. L., DiLavore, P. C., et al. (2000). The autism diagnostic observation schedule-generic: A standard measure of social and communication deficits associated with the spectrum of autism. Journal of Autism and Developmental Disorders, 30(3), 205–223. https://doi.org/10.1023/a:1005592401947.CrossRefGoogle Scholar
- Lord, C., Rutter, M., & Couteur, A. (1994). Autism Diagnostic Interview-Revised: A revised version of a diagnostic interview for caregivers of individuals with possible pervasive developmental disorders. Journal of Autism and Developmental Disorders, 24(5), 659–685. https://doi.org/10.1007/bf02172145.CrossRefGoogle Scholar
- Lord, C., Rutter, M., DiLavore, P. C., & Risi, S. (1999). Autism diagnostic observation schedule: ADOS manual. Los Angeles: Western Psychological Services.Google Scholar
- Losh, M., Childress, D., Lam, K., & Piven, J. (2008). Defining key features of the broad autism phenotype: A comparison across parents of multiple- and single-incidence autism families. American Journal of Medical Genetics, Part B: Neuropsychiatric Genetics, 147B(4), 424–433. https://doi.org/10.1002/ajmg.b.30612.CrossRefGoogle Scholar
- Mullen, E. M. (1995). Mullen scales of early learning. Circle Pines, MN: American Guidance Service.Google Scholar
- Orsmond, G. I., Lin, L.-Y., & Seltzer, M. M. (2007). Mothers of adolescents and adults with autism: Parenting multiple children with disabilities. Intellectual and Developmental Disabilities, 45(4), 257–270. https://doi.org/10.1352/1934-9556(2007)45%5b257:moaaaw%5d2.0.co;2.CrossRefGoogle Scholar
- Pandey, J. (2008). The Modified Checklist for Autism in Toddlers (M-CHAT) sibling study: Are younger siblings representative of the general ASD population?. Storrs, CT: University of Connecticut.Google Scholar
- Perry, A., Flanagan, H. E., Dunn Geier, J., & Freeman, N. L. (2009). Brief Report: The Vineland Adaptive Behavior Scales in young children with Autism Spectrum Disorders at different cognitive levels. Journal of Autism and Developmental Disorders, 39(7), 1066–1078. https://doi.org/10.1007/s10803-009-0704-9.CrossRefGoogle Scholar
- Robinson, E., Samocha, K., Kosmicki, J., McGrath, L., Neale, B. M., Perlis, R. H., et al. (2014). Autism spectrum disorder severity reflects the average contribution of de novo and familial influences. Proceedings of the National Academy of Sciences of USA, 111(42), 15161–15165. https://doi.org/10.1073/pnas.1409204111.CrossRefGoogle Scholar
- Roid, G. H. (2003). Stanford–Binet intelligence scales, fifth edition: Technical manual. Itasca, IL: Riverside Publishing.Google Scholar
- Schwichtenberg, A. J., Young, G. S., Sigman, M., Hutman, T., & Ozonoff, S. (2010). Can family affectedness inform infant sibling outcomes of Autism Spectrum Disorders? Journal of Child Psychology and Psychiatry, 51(9), 1021–1030. https://doi.org/10.1111/j.1469-7610.2010.02267.x.CrossRefGoogle Scholar
- Sparrow, S. S., Balla, D. A., & Cicchetti, D. (1984). Vineland adaptive behaviour scales. Circle Pines, MN: American Guidance Service.Google Scholar
- Taylor, L. J., Maybery, M. T., Wray, J., Ravine, D., Hunt, A., & Whitehouse, A. J. O. (2015). Are there differences in the behavioural phenotypes of Autism Spectrum Disorder probands from simplex and multiplex families? Research in Autism Spectrum Disorders, 11, 56–62. https://doi.org/10.1016/j.rasd.2014.12.003.CrossRefGoogle Scholar
- Virkud, Y. V., Todd, R. D., Abbacchi, A. M., Zhang, Y., & Constantino, J. N. (2009). Familial aggregation of quantitative autistic traits in multiplex versus simplex autism. American Journal of Medical Genetics. Part B, Neuropsychiatric Genetics: The Official Publication of the International Society of Psychiatric Genetics, 150B(3), 328. https://doi.org/10.1002/ajmg.b.30810.CrossRefGoogle Scholar
- Weiner, D. J., Wigdor, E. M., Ripke, S., Walters, R. K., Kosmicki, J. A., Grove, J., et al. (2017). Polygenic transmission disequilibrium confirms that common and rare variation act additively to create risk for autism spectrum disorders (Analysis). Nature Genetics, 49(7), 978–985. https://doi.org/10.1038/ng.3863.CrossRefGoogle Scholar
- Zwaigenbaum, L., Thurm, A., Stone, W., Baranek, G., Bryson, S., Iverson, J., et al. (2007). Studying the emergence of Autism Spectrum Disorders in high-risk infants: Methodological and practical issues. Journal of Autism and Developmental Disorders, 37(3), 466–480. https://doi.org/10.1007/s10803-006-0179-x.CrossRefGoogle Scholar