Abstract
Rett syndrome is a severe neurodevelopmental disorder that results in both motor and language skill regression with a wide range of severity in symptom presentation. Communication intervention may be particularly challenging for this population due to the decline in speech, motor skills, and motor planning difficulties that characterize the disorder (Townend et al., 2020), often resulting in the need for augmentative and alternative communication (AAC) technology. Very limited research has evaluated communication interventions for individuals with Rett syndrome and even fewer have targeted expressive communication, an important skill required for improved autonomy and quality of life (Sigafoos et al., 2009; Townend et al., 2020). The current study sought to systematically replicate the Simacek et al. (2017) mand training procedures to teach three girls with Rett Syndrome to use AAC to make requests through caregiver coaching by researchers via telehealth. Results suggest that mand training was successful in increasing AAC use for all three participants. Barriers to intervention for this population and implications of results for future research and clinical practice are discussed.
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References
Akemoglu, Y., Muharib, R., & Meadan, H. (2020). A systematic and quality review of parent-implemented language and communication interventions conducted via telepractice. Journal of Behavioral Education, 29, 282–316. https://doi.org/10.1007/s10864-019-09356-3
Allen, A. A., Schlosser, R. W., Brock, K. L., & Shane, H. C. (2017). The effectiveness of aided augmented input techniques for persons with developmental disabilities: A systematic review. Augmentative and Alternative Communication, 33(3), 149–159. https://doi.org/10.1080/07434618.2017.1338752
American Speech-Language-Hearing Association (n.d.). Augmentative and Alternative Communication (Practice Portal). Retrieved July 2, 2022, from www.asha.org/Practice-Portal/Professional-Issues/Augmentative-and-Alternative-Communication/
Amoako, A. N., & Hare, D. J. (2020). Non-medical interventions for individuals with rett syndrome: A systematic review. Journal of Applied Research in Intellectual Disabilities, 33, 808–827. https://doi.org/10.1111/jar.12694
Anderson, C. M., & Long, E. S. (2002). Use of a structured descriptive assessment methodology to identify variables affecting prob;em behavior. Journal of Applied Behavior Analysis, 35(2), 137–154. https://doi.org/10.1901/jaba.2002.35-137
Andzik, N. R., Schaefer, J. M., Nichols, R. T., & Chung, Y. (2018). National survey describing and quantifying students with communication needs. Developmental Neurorehabilitation, 21(1), 40–47. https://doi.org/10.1080/17518423.2017.1339133
Bak, M. Y. S., Dueñas, A. D., Avendaño, G., & A. C., & Stanley, T. (2021). Tact instruction for children with autism spectrum disorder: A review. Autism and Developmental Language Impairments, 6, 1–6. https://doi.org/10.1177/2396941521999010
Bartolotta, T. E., & Remshifski, P. A. (2011). Communication skills in girls with rett syndrome. Focus on Autism and Other Developmental Disabilities, 26(1), 15–24. https://doi.org/10.1177/1088357610380042
Brady, N. C., Bruce, S., Goldman, A., Erickson, K., Mineo, B., Ogletree, B. T., Paul, D., Romski, M. A., Sevcik, R., Siegel, E., Schoonover, J., Snell, M., Sylvester, L., & Wilkison, K. (2016). Communication services and supports for individuals with severe disabilities: Guidance for assessment and intervention. American Journal on Intellectual and Developmental Disabilities, 121(2), 121–138. https://doi.org/10.1352/1944-7558-121.2.121
Byiers, B. J., Dimian, A., & Symons, F. J. (2014). Functional communication training in rett syndrome: A preliminary study. American Journal on Intellectual and Developmental Disabilities, 119(4), 340–350. https://doi.org/10.1352/1944-7558-119.4.340
Byiers, B. J., & Symons, F. J. (2012). Issues in estimating developmental level and cognitive function in Rett syndrome. In R. M. Hodapp (Ed.), International review of research in developmental disabilities (Vol. 43, pp. 147–185). Academic Press. https://doi.org/10.1016/B978-0-12-398261-2.00004-0
California Association for Behavior Analysis. (2017). Telehealth and the practice of applied behavior analysis (ABA). Retrieved January 6, 2023, from https://calaba.org/media/content/CalABA_TeleHealth_Practice_Brief_Final-2.pdf
Carnett, A., Raulston, T. J., & Charpentier, J. (2019). The application of skinner’s analysis of verbal behavior for teaching communication skills to persons with developmental disabilities. Current Developmental Disorders Reports, 6, 131–137. https://doi.org/10.1007/s40474-019-00170-0
Cianfaglione, R., Clarke, A., Kerr, M., Hastings, R. P., Oliver, C., & Felce, D. (2015). A national survey of rett syndrome: Age, clinical characteristics, current abilities, and health. American Journal of Medical Genetics, 167(A), 1493–1500. https://doi.org/10.1002/ajmg.a.37027
Dimian, A. F., Elmquist, M., Reichle, J., & Simacek, J. (2018). Teaching communicative responses with a speech-generating device via telehealth coaching. Advances in Neurodevelopmental Disorders, 2, 86–99. https://doi.org/10.1007/s41252-018-0055-7
Drew, C. M., Machalicek, W., Crowe, B., Glugatch, L., Wei, Q., & Erturk, B. (2022). Parent-implemented behavior interventions via telehealth for older children and adolescents. Journal of Behavioral Education, Advanced online publication. https://doi.org/10.1007/s10864-021-09464-z
Elmquist, M., Simacek, J., Dimian, A. F., & Reichle, J. (2019). Impact of aided aac interventions on speech comprehension of children with neurodevelopmental disabilities: A critically appraised topic. Evidence-Based Communication Assessment and Intervention, 13(1–2), 67–84. https://doi.org/10.1080/17489539.2019.1598011
Fabio, R. A., Semino, M., & Giannatiempo, S. (2022). The GAIRS checklist: a useful global assessment tool in patients with rett syndrome. Orphanet Journal of Rare Diseases, 17, 116–116. https://doi.org/10.1186/s13023-022-02259-z
Fisher, W., Piazza, C. C., Bowman, L. G., Hagopian, L. P., Owens, J. C., & Slevin, I. (1992). A comparison of two approaches for identifying reinforcers for persons with severe and profound disabilities. Journal of Applied Behavior Analysis, 25(2), 491–498. https://doi.org/10.1901/jaba.1992.25-491
Ganz, J. B. (2015). AAC interventions for individuals with autism spectrum disorders: State of the science and future research directions. Augmentative and Alternative Communication, 31(3), 203–214. https://doi.org/10.3109/07434618.2015.1047532
García, J. C., Díez, E., Wojcik, D. Z., & Santamaría, M. (2020). Communication support needs in adults with intellectual disabilities and its relation to quality of life. International Journal of Environmental Research and Public Health, 17, 7370. https://doi.org/10.3390/ijerph17207370
Greer, D. R., & Ross, D. E. (2008). Verbal behavior analysis: Inducing and expanding new verbal capabilities in children with language delays. Pearson.
Hagberg, B. (2002). Clinical manifestations and stages of Rett syndrome. Mental Retardation and Developmental Disabilities Research Reviews, 8(2), 61–65. https://doi.org/10.1002/mrdd.10020
Hagberg, B., & Witt-Engerström, I. (1986). Rett syndrome: A suggested staging system for describing impairment profile with increasing age toward adolescence. American Journal of Medical Genetics, 24, 47–59. https://doi.org/10.1002/ajmg.1320250506
Harris, M. D., & Reichle, J. (2004). The impact of aided language stimulation on symbol comprehension and production in children with moderate cognitive disabilities. American Journal of Speech-Language Pathology, 13, 155–167. https://doi.org/10.1044/1058-0360(2004/016)
Haywood, H., & Lidz, C. (2006). Dynamic assessment in practice: Clinician and educational applications. Cambridge University Press. https://doi.org/10.1017/CBO9780511607516
Howard, J. S., Sparkman, C. R., Cohen, H. G., Green, G., & Stanislaw, H. (2005). A comparison of intensive behavior analytic and eclectic treatments for young children with autism. Research in Developmental Disabilities, 26, 359–383. https://doi.org/10.1016/j.ridd.2004.09.005
Huer, M. (1983). The Nonspeech Test. Don Johnston Developmental Equipment Inc. Retrieved from https://www.proedinc.com/Products/12640/tecel-test-of-early-communication-and-emerging-language.aspx
Huer, M. B., & Miller, L. (2011). Examiner’s manual of the Test of Early Communication and Emerging Language (TECEL). Technical manual. Pro-Ed.
Iacono, T., Carter, M., & Hook, J. (1998). Identification of intentional communication in students with severe and multiple disabilities. Augmentative and Alternative Communication, 14, 102–114. https://doi.org/10.1080/07434619812331278246
Iacono, T., Trembath, D., & Erickson, S. (2016). The role of augmentative and alternative communication for children with autism: Current status and future trends. Neuropsychiatric Disease and Treatment, 12, 2349–2361. https://doi.org/10.2147/NDT.S95967
International Rett Syndrome Foundation. (2022). Find a Rett Clinic. Retrieved June 15, 2022, from https://www.rettsyndrome.org/for-families/rett-syndrome-clinics/
Jennings, A. M., Vladescu, J. C., Miguel, C. F., Reeve, K. F., & Sidener, T. M. (2021). A systematic review of empirical intraverbal research: 2015–2020. Behavioral Interventions, 37, 79–104. https://doi.org/10.1002/bin.1815
Kane, C. L., & DeBar, R. M. (2022). A Descriptive Review of Telehealth for Individuals With Autism Spectrum Disorder. Behavior Modification. https://doi.org/10.1177/01454455221121085
Larriba-Quest, K., Byiers, B. J., Beisang, A., Merbler, A. M., & Symons, F. J. (2020). Special education supports and services for rett syndrome: Parent perceptions and satisfaction. Intellectual and Developmental Disabilities, 58(1), 49–64. https://doi.org/10.1352/1934-9556-58.1.49
Lim, J., Greenspoon, D., Hunt, A., & Mcadam, L. (2020). Rehabilitation interventions in Rett syndrome: A scoping review. Developmental Medicine and Child Neurology, 62, 906–916. https://doi.org/10.1111/dmcn.14565
Lindgren, S., Wacker, D., Schieltz, K., Suess, A., Pelzel, K., Kopelman, T., Lee, J., Romani, P., & O’Brien, M. (2020). A randomized controlled trial of functional communication training via telehealth for young children with autism spectrum disorder. Journal Developmental Disorders, 50(12), 4449–4462. https://doi.org/10.1007/s10803-020-04451-1
Machalicek, W., Lequia, J., Pinkelman, S., Knowles, C., Raulston, T., Davis, T., & Alresheed, F. (2016). Behavioral telehealth consultation with families of children with autism spectrum disorder. Behavioral Interventions, 31(3), 223–250. https://doi.org/10.1002/bin.1450
Marino, F., Chilà, P., Failla, C., Crimi, I., Minutoli, R., Puglisi, A., Arnao, A. A., Tartarisco, G., Ruta, L., Vagni, D., & Pioggia, G. (2020). Tele-assisted behavioral intervention for families with children with autism spectrum disorders: A randomized control trial. Brain Sciences, 10, 649–661. https://www.mdpi.com/2076-3425/10/9/649
Markham, C., van Laar, D., Gibbard, D., & Dean, T. (2009). Children with speech, language and comunicaatino needs: Their perceptions of their quality of life. International Journal of Language and Communication Disorders, 44(5), 748–768. https://doi.org/10.1080/13682820802359892
National Institute of Neurological Disorders and Stroke (NINDS). (2021). Rett Syndrome Fact Sheet. Retrieved June 15, 2022, from https://www.ninds.nih.gov/Disorders/Patient-Caregiver-Education/Fact-Sheets/Rett-Syndrome-Fact-Sheet
Neely, L., MacNaul, H., Gregori, E., & Cantrell, K. (2021). Effects of telehealth‐mediated behavioral assessments and interventions on client outcomes: A quality review. Journal of Applied Behavior Analysis, 54, 484–510. https://doi.org/10.1002/jaba.818
Neely, L., Rispoli, M., Gerow, S., Hong, E. R., & Hagan-Burke, S. (2017). Fidelity outcomes for autism-focused interventionists coached via telepractice: A systematic literature review. Journal of Developmental & Physical Disabilities, 29, 849–874. https://doi.org/10.1007/s10882-017-9550-4
Reichle, J. (1997). Communication interventions with persons who have severe disabilities. The Journal of Special Education, 31(1), 110–134. https://doi.org/10.1177/002246699703100110
Reimers, T. M., Wacker, D. P., & Cooper, L. J. (1991). Evaluation of the acceptability of treatments for children’s behavioral difficulties: Ratings by parents receiving services in an outpatient clinic. Child & Family Behavior Therapy, 13(2), 53–71. https://doi.org/10.1300/J019v13n02_04
Rett Syndrome Research Trust. (2022). About Rett Syndrome. Retrieved June 15, 2022, from https://reverserett.org/about-rett-syndrome/
Ricceri, L., De Filippis, B., & Laviola, G. (2008). Mouse models of Rett syndrome: From behavioural phenotyping to preclinical evaluation of new therapeutic approaches. Behavioral Pharmacology, 19, 501–517. https://doi.org/10.1097/FBP.0b013e32830c3645
Sennott, S. C., Light, J. C., & McNaughton, D. (2016). AAC Modeling intervention research review. Research and Practice for Persons with Severe Disabilities, 41(2), 101–115. https://doi.org/10.1177/1540796916638822
Sigafoos, J., Green, V. A., Schlosser, R., O’Reilly, M. F., Lancioni, G. E., Rispoli, M., & Lang, R. (2009). Communication intervention in rett syndrome: A systematic review. Research in Autism Spectrum Disorders, 3, 304–318. https://doi.org/10.1016/j.rasd.2008.09.006
Sigafoos, J., Kagohara, D., van der Meer, L., Green, V. A., O’Reilly, M. F., Lancioni, G. E., Rispoli, M., & Zisimopoulos, D. (2011). Communication assessment for individuals with rett syndrome: A systematic review. Research in Autism Spectrum Disorders, 5, 692–700. https://doi.org/10.1016/j.rasd.2010.10.006
Sigafoos, J., Laurie, S., & Pennell, D. (1996). Teaching children with rett syndrome to request preferred objets using aided communication: Two preliminary studies. Augmentative and Alternative Communication, 12(2), 88–96. https://doi.org/10.1080/07434619612331277538
Sigafoos, J., Woodyatt, G., Keen, D., Tait, K., Tucker, M., Roberts-Pennell, D., & Pittendreigh, N. (2000). Identifying potential communicative acts in children with developmental and physical disabilities. Communication Disorders Quarterly, 21(2), 77–86. https://doi.org/10.1177/152574010002100202
Simacek, J., Reichle, J., & McComas, J. J. (2017). Communication interventions to teach requesting through aided AAC for two learners with rett syndrome. Journal of Physical and Developmental Disabilities, 28, 59–81. https://doi.org/10.1007/s10882-015-9423-7
Skinner, B. F. (1957). Verbal behavior. Appleton-Century-Crofts. https://doi.org/10.1037/11256-000
Smith, T., Groen, A. D., & Wynn, J. W. (2000). Randomized trial of intensive early intervention for children with pervasive developmental disorder. American Journal of Mental Retardation, 105(4), 269–285. https://doi.org/10.1352/0895-8017(2000)105%3c0269:RTOIEI%3e2.0.CO;2
Still, K., Rehfeldt, R. A., Whelan, R., May, R., & Dymond, S. (2014). Facilitating requesting skills using high-tech augmentative and alternative communication devices with individuals with autism spectrum disorders: A systematic review. Research in Autism Spectrum Disorders, 8, 1184–1199. https://doi.org/10.1016/j.rasd.2014.06.003
Suess, A. N., Wacker, D. P., Schwartz, J. E., Lustig, N., & Detrick, J. (2016). Preliminary evidence on the use of telehealth in an outpatient behavior clinic. Journal of Applied Behavior Analysis, 49(3), 1–7. https://doi.org/10.1002/jaba.305
Sundberg, M. L., & Michael, J. (2001). The benefits of skinner’s analysis of verbal behavior for children with autism. Behavior Modification, 25, 698–724. https://doi.org/10.4135/9781473978409
Tapp, M. C., Pennington, R. C., Clausen, A. M., & Carpenter, M. E. (2021). Systematic review of mand training parameters for students with intellectual and developmental disabilities in school settings. Education and Training in Autism and Developmental Disabilities, 56(4), 437–453.
Tomlinson, S. R. L., Gore, N., & McGill, P. (2018). Training individuals to implement applied behavior analytic procedures via telehealth: A systematic review of the literature. Journal of Behavioral Education, 27, 172–222. https://doi.org/10.1007/s10864-018-9292-0
Townend, G. S., Bartolotta, T. E., Urbanowicz, A., Wandin, H., & Curds, L. M. G. (2020). Rett syndrome communication guidelines: A handbook for therapists, educators and families. Rett Experience Center; Rettsyndrome.org.
Unholz-Bowden, E., McComas, J. J., McMaster, K. L., Girtler, S. N., Kolb, R. L., & Shipchandler, A. (2020). Caregiver training via telehealth on behavioral procedures: A systematic review. Journal of Behavioral Education. https://doi.org/10.1007/s10864-09381-7
Urbanowicz, A., Leonard, H., Girdler, S., Ciccone, N., & Downs, J. (2016). Parental perspectives on communication abilities of their daughters with rett syndrome. Developmental Neurorehabilitation, 19(1), 17–25. https://doi.org/10.3109/17518423.2013.879940
U.S. Department of Health and Human Services (Department). (2021, February 25). Health Resources & Services Administration. Retrieved January 5, 2023, from https://telehealth.hhs.gov/patients/understanding-telehealth/
Van Acker, R., & Grant, S. H. (1995). An effective computer-based requesting system for persons with rett syndrome. Journal of Childhood Communication Disorders, 16(2), 31–38. https://doi.org/10.1177/152574019501600205
Wandin, H., Lindberg, P., & Sonnander, K. (2021). Aided language modeling, responsive communication and eye-gaze technology as communication intervention for adults with rett syndrome: Three experimental single case studies. Disability and Rehabilitation: Assistive Technology, 16, 1–15. https://doi.org/10.1080/17483107.2021.1967469
Ward, C., Chiat, S., & Townend, G. S. (2021). A comparison of formal and informal methods for assessing language and cognition in children with rett syndrome. Research in Developmental Disabilities, 114, 103961. https://doi.org/10.1016/j.ridd.2021.103961
Wehmeyer, M., & Schwartz, M. (1997). Self-determination and positive adult outcomes: A follow-up study of youth with mental retardation or learning disabilities. Exceptional Children, 63(2), 245–255. https://doi.org/10.1177/001440299706300207
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This study was funded by National Institute of Health/National Institute on Deafness and Other Communication Disorders Grant No. 1R21DC015021.
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All procedures in this study involving human participants were approved by the Human Subjects Review Board at the University of Minnesota (IRB# 1404M49782 & 00008480). As such, procedures we’re conducted in accordance with the ethical standards of the institutional and national research committee and with the 1964 Helsinki Declaration and its later amendments or comparable ethical standards.
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Kolb, R.L., McComas, J.J., Girtler, S.N. et al. Teaching Requesting to Individuals with Rett Syndrome Using Alternative Augmentative Communication (AAC) Through Caregiver Coaching via Telehealth. J Dev Phys Disabil 35, 1063–1090 (2023). https://doi.org/10.1007/s10882-023-09894-9
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DOI: https://doi.org/10.1007/s10882-023-09894-9