The following are research studies where data collection is complete and we are currently working on manuscripts or have already published manuscripts:
Pilot Study of Play and Humor Infused Exposure Therapy for Young Children with Williams Syndrome
Many individuals with Williams syndrome have fears and anxieties. In this pilot study, we examined the acceptability and effectiveness of play- and humor-infused exposure therapy (based in Levine & Chedd’s “Replays” Approach). We found that families found the approach acceptable, and that even a small number of sessions led to some improvement in fear and anxiety for many of the children with Williams syndrome. We look forward to building on this work so further develop the usefulness of this approach, and also to disseminate guidance to community clinicians about how to implement the intervention with children with Williams syndrome. This work is conducted in collaboration with Dr. Karen Levine who has long worked clinically with children with Williams syndrome and their families.
This research was supported by a grant from the Williams Syndrome Association (WS #0110).
We now have 2 published papers from this work.
- In this manuscript, we describing outcomes of our study of the impact of the intervention in 9 children with Williams syndrome: Klein-Tasman, B. P., Young, B. N., Levine, K., Rivera, K., Miecielica, E. J., Yund, B. D., & French, S. E. (2022). Acceptability and effectiveness of humor-and play-Infused exposure therapy for fears in Williams syndrome. Evidence-Based Practice in Child and Adolescent Mental Health, 7(1), 94-111. [This is an Accepted Manuscript of an article published by Taylor & Francis in Evidence-Based Practice in Child and Adolescent Mental Health on October 12, 2021, available online: https://doi.org/10.1080/23794925.2021.1950080.
- In this manuscript, we dive more deeply into the effects of the intervention for the 4 children for whom it had the clearest positive impact, using observational coding of child behavior and therapist behavior: Young, B. N., Mohanty, E., Levine, K., & Klein-Tasman, B. P. (2023). Addressing fears of children with Williams syndrome: therapist and child behavior in the context of a novel play-and humor-infused exposure therapy approach. Frontiers in Psychology, 14, 1098449. https://doi.org/10.3389/fpsyg.2023.1098449
Response Inhibition Training for Children with Williams Syndrome
Many individuals with Williams syndrome have difficulty with inhibiting impulses. In this investigation we are trying out a cognitive retraining approach using computer games to improve inhibitory control in children and adolescents with Williams syndrome. This work is conducted in collaboration with Dr. Hanjoo Lee from the University of Wisconsin-Milwaukee. The primary goal of the current study is to test the effectiveness of a web-based response inhibition training program among individuals with Williams syndrome with a small randomized controlled trial.
This research was supported by a grant from the Williams Syndrome Association (WS #0110).
Brei, N. G., Raicu, A. M., Lee, H. J., & Klein-Tasman, B. P. (2020). Feasibility and acceptability of an online response inhibition cognitive training program for youth with Williams syndrome. International Review of Research in Developmental Disabilities.
School-Age Outcomes in NF1: Attention, Social, and Academic Functioning
As a follow-up to our past research examining early cognitive and behavior characteristics of young children with NF1, we examined attention, social, and academic functioning in 9-13 year olds with NF1. Research on children and adolescents with NF1 indicate that many youth experience significant academic and functional impairments related to attention difficulties. In addition, more recent research has suggested that children and adolescents with NF1 are at increased risk for difficulties with social skills, social-communication, reciprocal social interaction, and repetitive behaviors. However, the current literature is limited in its impact because published studies have been based either on the use of single measures or on case reports. As such, the goal of this study was twofold. First, we were aiming to characterize preschool-age predictors of school-age academic, attention, and psychosocial functioning. Second, we were aiming to characterize children and adolescents with NF1 with regard to core diagnostic features, associated features, and predictors of ASD symptomatology. We are currently in the process of data analysis.
This work was funded by NF Midwest and NF Northeast.
Glad, D., Casnar, C., Yund, B., Enderle, M., Siegel, D., Basel, D., & Klein-Tasman, B. P. (2020). Adaptive behavior and relations with executive functioning in children with neurofibromatosis type 1 using a mixed design. Journal of Developmental and Behavioral Pediatrics.
Payne, J., Walsh, K., Pride, N., Haebich, K., Maier, A., Chisolm, A., Glad, D. M., Casnar, C. L., North, K., & Klein-Tasman, B. P. (2020). Social skills and autism spectrum disorder symptoms in children with neurofibromatosis type 1: objective evidence for clinical trial outcomes. Developmental Medicine and Child Neurology, 62, 813-819. DOI: 10.1111/dmcn.14517
Early Cognitive and Behavior Characteristics in Neurofibromatosis-1
Previous research with older children with NF1 has shown that some children have learning, attention, and emotional difficulties. Given the lack of information available for younger children, and since NF1 is often diagnosed during the preschool years, we studied the functioning of 2 – 6 year-olds with NF1 and siblings without NF1 in collaboration with the Neurofibromatosis Clinic at the Genetics Center (Donald Basel, M. D., & Dawn Siegel, M.D.) and the Neurofibromatosis Program at the University of Chicago (Jim Tonsgard, M.D., and Scott Hunter, Ph.D.). We are no longer recruiting new participants for this study.
This research was supported by a UWM Research Growth Initiative Award, an Award from the University of Chicago Center for Translational Science CTSA (UL1 RR024999), NF Inc Midwest, NF Inc MidAtlantic, and private donations.
Casnar, C. L., & Klein-Tasman, B. P. (2017). Parent and teacher perspectives on emerging executive functioning in preschoolers with neurofibromatosis type 1: Comparison to unaffected children and lab-based measures. Journal of Pediatric Psychology, 42 (2): 198-207. https://doi.org/10.1093/jpepsy/jsw042.
Casnar, C. L., Janke, K., van der Fluit, F., Brei, N., & Klein-Tasman, B. P. (2014). Relations between fine motor skill and parental reports of attention in young children with neurofibromatosis-1. Journal of Clinical and Experimental Neuropsychology, 36(9): 930-943. DOI: 10.1080/13803395.2014.957166
Brei, N., Casnar, C. L., Schwarz, G. N. & Klein-Tasman, B. P. (2014). Language in young children with neurofibromatosis-1: Relations to functional communication, attention, and social functioning. Research in Developmental Disabilities, 35, 2495-2504.
Klein-Tasman, B. P., Janke, K. M., Luo, W., Casnar, C. L., Hunter, S. J., Tonsgard, J., Trapane, P., van der Fluit, F. & Kais, L. A. (2014). Cognitive and psychosocial phenotype of young children with neurofibromatosis-1. Journal of the International Neuropsychological Society, 20, 88-98. doi:10.1017/S1355617713001227
Klein-Tasman, B. P., Colon, A. M., Brei, N., van der Fluit, F., Casnar, C. L., Janke, K. M., Basel, D., Siegel, D. H. & Walker, J. A. (2013). Adaptive behavior in young children with neurofibromatosis – 1. International Journal of Pediatrics, Article ID 690432, 7 pages. doi:10.1155/2013/690432.
Evaluation of Measures of Attention for Young Children with NF1
Research suggests that children with NF1 experience an elevated rate of attention difficulties in comparison to same-aged peers; however, there is very little research about the appropriateness of the ways we measure attention for young children. Since attention difficulties during the preschool years can be subtle, it is important to know which attention measures are most likely to show difficulties in a consistent and meaningful way. Participants were children between the ages of 4 and 6 with NF1. The children completed two assessment visits approximately 2 months apart. At both visits, the researcher completed tasks with the child to look at cognitive, attention, and executive functioning skills. Caregivers completed questionnaires about their child’s behavior, emotions, temperament, and attention. We then evaluated the research-related properties of the measures used to gain a sense of which measures yielded consistent findings over time and which measures were most related to ratings of everyday functioning.
This work was funded by NF Midwest.
Pardej, S. K., Lee, K. M., Glad, D. M., & Klein-Tasman, B. P. (2022). Psychometric Properties of Attention Measures in Young Children with Neurofibromatosis Type 1: Preliminary Findings. Journal of pediatric neuropsychology, 8(3), 123–136. https://doi.org/10.1007/s40817-022-00129-x
Autism Spectrum Symptomatology among Children with Williams Syndrome
Children with Williams syndrome are very interested in interacting with others, which seems very different from how we think about autism spectrum disorders. However, there are overlaps of some of the characteristics of children with Williams syndrome with children with autism spectrum disorders. In our research, we have used tools developed for research and clinical purposes to assess social communication and restricted and repetitive behaviors to get a systematic sense of the degree of that overlap. We have found that many young children with Williams syndrome with limited language abilities show some of the same social communication challenges as typically seen for children with autism spectrum disorders. Once speaking more fluently, somewhat fewer children show these characteristics (around 20%) and we expect that these children may benefit from some of the same intervention approaches as are used with children with autism spectrum disorders.
This research was supported by a UWM Graduate School Research Committee Award and by a grant from the National Institutes of Mental Health (R03 MH69400).
Klein-Tasman, B. P., van der Fluit, F., & Mervis, C. B. (2018). Autism spectrum symptomatology in children with Williams syndrome who have phrase speech or fluent language. Journal of Autism and Developmental Disorders, 48: 3037-3050. https://doi.org/10.1007/s10803-018-3555-4
Klein-Tasman, B. P., Li-Barber, K. T., & Magargee, E. T. (2011). Honing in on the social phenotype in Williams syndrome using multiple measures and multiple raters. Journal of Autism and Developmental Disorders, 41 (3) 341-351.
van der Fluit, F., Gaffrey, M., & Klein-Tasman, B. P. (2012). Social cognition in Williams syndrome: Relations between performance on the social attribution task and cognitive and behavioral characteristics. Frontiers in Developmental Psychology, 3:197. doi: 10.3389/fpsyg.2012.00197
Klein-Tasman, B. P., Li-Barber, K. T., & Magargee, E. T. (2011). Honing in on the social phenotype in Williams syndrome using multiple measures and multiple raters. Journal of Autism and Developmental Disorders, 41 (3) 341-351.
Klein-Tasman, B. P., Phillips, K. D., Lord, C., Mervis, C. B., & Gallo, F. G. (2009). Overlap with the autism spectrum in young children with Williams syndrome. Journal of Developmental and Behavioral Pediatrics, 30, 289-299.
Klein-Tasman, B. P., Mervis, C. B., Lord, C. E., & Phillips, K. D. (2007). Socio-communicative deficits in young children with Williams syndrome: Performance on the Autism Diagnostic Observation Schedule. Child Neuropsychology, 13, 444-467.