Evaluating Sensory Processing in Fragile X Syndrome: Psychometric Analysis of the Brain Body Center Sensory Scales (BBCSS)
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Individuals with fragile X syndrome (FXS), especially those co-diagnosed with autism spectrum disorder (ASD), face many sensory processing challenges. However, sensory processing measures informed by neurophysiology are lacking. This paper describes the development and psychometric properties of a parent/caregiver report, the Brain-Body Center Sensory Scales (BBCSS), based on Polyvagal Theory. Parents/guardians reported on 333 individuals with FXS, 41% with ASD features. Factor structure using a split-sample exploratory-confirmatory design conformed to neurophysiological predictions. Internal consistency, test–retest, and inter-rater reliability were good to excellent. BBCSS subscales converged with the Sensory Profile and Sensory Experiences Questionnaire. However, data also suggest that BBCSS subscales reflect unique features related to sensory processing. Individuals with FXS and ASD features displayed more sensory challenges on most subscales.
KeywordsFragile X Autism spectrum disorders Polyvagal theory Autonomic nervous system Psychometrics Sensory processing
We wish to thank all families who dedicated their time to make this study possible.
JK, MR, KJH, and SWP contributed to the conception and design of the study; JK conducted analyses and wrote the first draft of the manuscript; JK, MR, KJH, and SWP contributed to manuscript revision and approved the submitted version.
This study was funded by the North Carolina Translational & Clinical Sciences Institute Grant # 550KR111516.
Compliance with Ethical Standards
Conflict of interest
Jacek Kolacz declares that he has no conflict of interest. Melissa Raspa declares that she has no conflict of interest. Keri J. Heilman declares that she has no conflict of interest. Stephen W. Porges declares that he has no conflict of interest.
All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.
Informed consent was obtained from all individual participants included in the study.
- Ayres, A. J. (1972). Sensory integration and the child. Los Angeles, CA: Westem Psychological Services.Google Scholar
- Baranek, G. T. (1999). Sensory Experiences Questionnaire Version 2.1.Google Scholar
- Baranek, G. T., David, F. J., Poe, M. D., Stone, W. L., & Watson, L. R. (2006). Sensory experiences questionnaire: Discriminating sensory features in young children with autism, developmental delays, and typical development. Journal of Child Psychology and Psychiatry, 47(6), 591–601.CrossRefPubMedGoogle Scholar
- Baranek, G. T., Reinhartsen, D., & Wannamaker, S. (2001). Play: Engaging children with autism. In R. Heubner (Ed.), Autism: A sensorimotor approach to management (pp. 311–351). Philadelphia: F. A. Davis.Google Scholar
- Baranek, G. T., Roberts, J. E., David, F. J., Sideris, J., Mirrett, P. L., Hatton, D. D., & Bailey, D. B. (2008). Developmental trajectories and correlates of sensory processing in young boys with fragile X syndrome. Physical and Occupational Therapy in Pediatrics, 28(1), 79–98.CrossRefPubMedGoogle Scholar
- Belser, R. C., & Sudhalter, V. (1995). Arousal difficulties in males with fragile X syndrome: A preliminary report. Developmental Brain Dysfunction, 8(4–6), 270–279.Google Scholar
- Brown, C., & Dunn, W. (2002). Adolescent-adult sensory profile: user’s manual. San Antonio: Therapy Skill Builders.Google Scholar
- Christian, L. M., Dillman, D. A., & Smyth, J. D. (2008). The effects of mode and format on answers to scalar questions in telephone and web surveys. Advances in Telephone Survey Methodology, 12, 250–275.Google Scholar
- Dunn, W. (2014). Sensory Profile 2 manual. San Antonio, TX: Pearson.Google Scholar
- Fleiss, J. L. (1981). Statistical methods for rates and proportions (2nd ed.). New York: John Wiley.Google Scholar
- Hall, S. S., Frank, M. C., Pusiol, G. T., Farzin, F., Lightbody, A. A., & Reiss, A. L. (2015). Quantifying naturalistic social gaze in fragile X syndrome using a novel eye tracking paradigm. American Journal of Medical Genetics Part B: Neuropsychiatric Genetics, 168(7), 564–572.CrossRefPubMedCentralGoogle Scholar
- Herman, M. A., Cruz, M. T., Sahibzada, N., Verbalis, J., & Gillis, R. A. (2009). GABA signaling in the nucleus tractus solitarius sets the level of activity in dorsal motor nucleus of the vagus cholinergic neurons in the vagovagal circuit. American Journal of Physiology-Gastrointestinal and Liver Physiology, 296(1), G101–G111.CrossRefPubMedGoogle Scholar
- Kolacz, J., Lewis, G. F., & Porges, S. W. (in press). The integration of vocal communication and biobehavioral state regulation in mammals: A polyvagal hypothesis. In S. M. Brudzynski (Ed.), Handbook of ultrasonic vocalization. Boston: ElsevierGoogle Scholar
- McIntosh, D. N., Miller, L. J., & Shyu, V. (1999). Development and validation of the Short Sensory Profile. In W. Dunn (Ed.), Sensory profile manual (pp. 59–73). San Antonio, TX: Psychological Corporation.Google Scholar
- McNeish, D. (2017). Thanks coefficient alpha, we’ll take it from here. Psychological Methods. Advance online publication.Google Scholar
- Muthén, L. K., & Muthén, B. O. (1998–2015). Mplus user’s guide. (7th Ed.), Los Angeles, CA: Muthén & Muthén.Google Scholar
- Porges, S. W. (2011). The polyvagal theory: Neurophysiological foundations of emotions, attachment, communication, and self-regulation. New York: WW Norton.Google Scholar
- Porges, S. W. (2012). The Brain-Body Center Sensory Scales (BBCSS). The Brain-Body Center. Chicago: University of Illinois at Chicago.Google Scholar
- Porges, S. W., & Lewis, G. F. (2009). The polyvagal hypothesis: Common mechanisms mediating autonomic regulation, vocalizations, and listening. In S. M. Brudzynski (Ed.), Handbook of mammalian vocalizations: An integrative neuroscience approach (pp. 255–264). Amsterdam: Academic Press.Google Scholar
- Quinn, H. O. (2014). Bifactor models, explained common variance (ECV), and the usefulness of scores from unidimensional item response theory analyses. Unpublished Master’s thesis, The University of North Carolina at Chapel Hill, Chapel Hill, NC.Google Scholar
- R Core Team. (2017). R: A language and environment for statistical computing. Vienna: R Foundation for Statistical Computing. http://www.R-project.org/.
- Rogmann, J. J. (2013). Ordinal dominance statistics (orddom): An R project for statistical computing package to compute ordinal, nonparametric alternatives to mean comparison (version 3.1). Available online from the CRAN website http://cran.r-project.org/.
- semTools Contributors. (2016). semTools: Useful tools for structural equation modeling. R package version 0.4–14. Retrived from http://cran.r-project.org/web/packages/semTools/index.html.
- Stackhouse, T. M., Scharfenaker, S. K., Lachiewicz, A. M., Burgess, D., Hessl, D., Blitz, R., Burgess, K., Rohlik, D., Hess, L. G., Kidd, S. A., & Berry-Kravis, E. (2014). Sensory processing and integration issues in individuals with fragile X syndrome. Retrieved June 14, 2014, from https://fragilex.org/wp-content/uploads/2012/08/Sensory-Integration-Issues-In-Fragile-X-Syndrome-2014-May.pdf.
- Steiger, J. H., & Lind, J. C. (1980). Statistically-based tests for the number of common factors. Paper presented at the annual spring meeting of the Psychometric Society, Iowa City, IA.Google Scholar
- Wheeler, A. C., Mussey, J., Villagomez, A., Bishop, E., Raspa, M., Edwards, A., Bodfish, J., Bann, C., & Bailey, D. B. (2014). DSM-5 changes and the prevalence of parent-reported autism spectrum symptoms in fragile X syndrome. Journal of Autism and Developmental Disorders, 45(3), 816–829.Google Scholar
- Yates, A. (1987). Multivariate exploratory data analysis: A perspective on exploratory factor analysis. Albany: State University of New York Press.Google Scholar
- Zhu, J., Chang, L., Xie, J., & Ai, H. (2016). Arginine vasopressin injected into the dorsal motor nucleus of the vagus inhibits gastric motility in rats. Gastroenterology Research and Practice, 2016, 4618672.Google Scholar