Skip to main content
Top
Gepubliceerd in:

16-08-2023 | Original Paper

Diversity of Participants in Williams Syndrome Intervention Studies

Auteurs: Eva Shin, Caitlin Ravichandran, Danielle Renzi, Barbara R. Pober, Christopher J. McDougle, Robyn P. Thom

Gepubliceerd in: Journal of Autism and Developmental Disorders | Uitgave 10/2024

Log in om toegang te krijgen
share
DELEN

Deel dit onderdeel of sectie (kopieer de link)

  • Optie A:
    Klik op de rechtermuisknop op de link en selecteer de optie “linkadres kopiëren”
  • Optie B:
    Deel de link per e-mail

Abstract

Purpose: This study describes participant diversity in Williams syndrome (WS) intervention studies. Methods: A literature search was conducted to identify prospective treatment studies including participants with WS. Data was extracted on the reporting of and information provided on age, sex, cognitive ability, socioeconomic status, race, and ethnicity. Results: Eleven eligible articles were identified. Reporting rates of demographic factors varied considerably, with the highest rates for age and sex (100%) and the lowest reporting rates for race (18%) and ethnicity (9%). Combining demographic data from the two studies that reported on race and/or ethnicity (n = 33), 88% of participants were White. The combined participant mean age was 20.9 years. Conclusion: There is a low frequency of reporting on several demographic factors including socioeconomic status, race, and ethnicity in WS intervention studies. There is a need for increased representation of racial and ethnic minority groups, older participants, and more cognitively impaired patients in WS research.
Literatuur
go back to reference Alfieri, P., Scibelli, F., Casula, L., Piga, S., Napoli, E., Valeri, G., & Vicari, S. (2022). Cooperative parent-mediated therapy in children with fragile x syndrome and williams beuren syndrome: A pilot RCT study of a transdiagnostic intervention-preliminary data. Brain Sciences, 12(1), https://doi.org/10.3390/brainsci12010008. Alfieri, P., Scibelli, F., Casula, L., Piga, S., Napoli, E., Valeri, G., & Vicari, S. (2022). Cooperative parent-mediated therapy in children with fragile x syndrome and williams beuren syndrome: A pilot RCT study of a transdiagnostic intervention-preliminary data. Brain Sciences, 12(1), https://​doi.​org/​10.​3390/​brainsci12010008​.
go back to reference Bellugi, U., Lichtenberger, L., Jones, W., Lai, Z., & George, S., M (2000). I. The neurocognitive profile of Williams syndrome: A complex pattern of strengths and weaknesses. Journal of Cognitive Neuroscience. MIT Press Journals. https://doi.org/10.1162/089892900561959. Bellugi, U., Lichtenberger, L., Jones, W., Lai, Z., & George, S., M (2000). I. The neurocognitive profile of Williams syndrome: A complex pattern of strengths and weaknesses. Journal of Cognitive Neuroscience. MIT Press Journals. https://​doi.​org/​10.​1162/​089892900561959.
go back to reference 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. In International Review of Research in Developmental Disabilities (Vol. 59, pp. 107–134). Academic Press Inc. https://doi.org/10.1016/bs.irrdd.2020.09.002. 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. In International Review of Research in Developmental Disabilities (Vol. 59, pp. 107–134). Academic Press Inc. https://​doi.​org/​10.​1016/​bs.​irrdd.​2020.​09.​002.
go back to reference Chen, M. S., Lara, P. N., Dang, J. H. T., Paterniti, D. A., & Kelly, K. (2014). Twenty years post-NIH revitalization act: Enhancing minority participation in clinical trials (EMPaCT): Laying the groundwork for improving minority clinical trial accrual. Cancer, 120, 1091–1096. https://doi.org/10.1002/CNCR.28575.CrossRefPubMed Chen, M. S., Lara, P. N., Dang, J. H. T., Paterniti, D. A., & Kelly, K. (2014). Twenty years post-NIH revitalization act: Enhancing minority participation in clinical trials (EMPaCT): Laying the groundwork for improving minority clinical trial accrual. Cancer, 120, 1091–1096. https://​doi.​org/​10.​1002/​CNCR.​28575.CrossRefPubMed
go back to reference Domínguez-García, C. M., Serrano-Juárez, C. A., Rodríguez-Camacho, M., Moreno-Villagómez, J., Araujo Solís, M. A., & Prieto-Corona, B. (2022). Neuropsychological intervention in attention and visuospatial skills in two patients with Williams syndrome with different types of genetic deletion. Applied Neuropsychology: Child. https://doi.org/10.1080/21622965.2022.2063723.CrossRefPubMed Domínguez-García, C. M., Serrano-Juárez, C. A., Rodríguez-Camacho, M., Moreno-Villagómez, J., Araujo Solís, M. A., & Prieto-Corona, B. (2022). Neuropsychological intervention in attention and visuospatial skills in two patients with Williams syndrome with different types of genetic deletion. Applied Neuropsychology: Child. https://​doi.​org/​10.​1080/​21622965.​2022.​2063723.CrossRefPubMed
go back to reference Fletcher, S. A., Bivalacqua, T. J., Brawley, O. W., & Kates, M. (2022). Race, ethnicity, and gender reporting in north american clinical trials for BCG-unresponsive non-muscle invasive bladder cancer. Urologic Oncology, 40(5), 195. .e13-195.e18.CrossRef Fletcher, S. A., Bivalacqua, T. J., Brawley, O. W., & Kates, M. (2022). Race, ethnicity, and gender reporting in north american clinical trials for BCG-unresponsive non-muscle invasive bladder cancer. Urologic Oncology, 40(5), 195. .e13-195.e18.CrossRef
go back to reference Jurado, L., Peoples, R., Kaplan, P., Hamel, B., & Francke, U. (1996). Molecular definition of the chromosome 7 deletion in Williams syndrome and parent-of-origin effects on growth. Am J Human Genet, 59(4), 781–792. Jurado, L., Peoples, R., Kaplan, P., Hamel, B., & Francke, U. (1996). Molecular definition of the chromosome 7 deletion in Williams syndrome and parent-of-origin effects on growth. Am J Human Genet, 59(4), 781–792.
go back to reference Kassai, B., Bouyé, P., Gilbert-Dussardier, B., Godart, F., Thambo, J. B., Rossi, M., et al. (2019). Minoxidil versus placebo in the treatment of arterial wall hypertrophy in children with Williams Beuren Syndrome: A randomized controlled trial. BMC Pediatrics, 19(1), https://doi.org/10.1186/s12887-019-1544-1. Kassai, B., Bouyé, P., Gilbert-Dussardier, B., Godart, F., Thambo, J. B., Rossi, M., et al. (2019). Minoxidil versus placebo in the treatment of arterial wall hypertrophy in children with Williams Beuren Syndrome: A randomized controlled trial. BMC Pediatrics, 19(1), https://​doi.​org/​10.​1186/​s12887-019-1544-1.
go back to reference 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. https://doi.org/10.1080/23794925.2021.1950080.CrossRef 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. https://​doi.​org/​10.​1080/​23794925.​2021.​1950080.CrossRef
go back to reference Schubert, C. (2008). The genomic basis of the Williams-Beuren syndrome. Cellular And Molecular Life Sciences, 66(7), 1178–1197.CrossRefPubMedCentral Schubert, C. (2008). The genomic basis of the Williams-Beuren syndrome. Cellular And Molecular Life Sciences, 66(7), 1178–1197.CrossRefPubMedCentral
go back to reference Sheikh, A., Netuveli, G., Kai, J., & Panesar, S. S. (2004). Comparison of reporting of ethnicity in US and European randomised controlled trials. Bmj, 87–88. Sheikh, A., Netuveli, G., Kai, J., & Panesar, S. S. (2004). Comparison of reporting of ethnicity in US and European randomised controlled trials. Bmj, 87–88.
go back to reference Siegel, M., McGuire, K., Veenstra-VanderWeele, J., Stratigos, K., & King, B. (2019). Practice parameter for the assessment and treatment of psychiatric disorders in children and adolescents with intellectual disability (Intellectual Developmental disorder)) Committee on Quality Issues (CQI). Journal Of The American Academy Of Child And Adolescent Psychiatry, 59(4), 468–496. https://doi.org/10.1016/j.jaac.2019.11.018.CrossRefPubMed Siegel, M., McGuire, K., Veenstra-VanderWeele, J., Stratigos, K., & King, B. (2019). Practice parameter for the assessment and treatment of psychiatric disorders in children and adolescents with intellectual disability (Intellectual Developmental disorder)) Committee on Quality Issues (CQI). Journal Of The American Academy Of Child And Adolescent Psychiatry, 59(4), 468–496. https://​doi.​org/​10.​1016/​j.​jaac.​2019.​11.​018.CrossRefPubMed
go back to reference Steinbrenner, J. R., McIntyre, N., Rentschler, L. F., Pearson, J. N., Luelmo, P., Jaramillo, M. E., et al. (2022). Patterns in reporting and participant inclusion related to race and ethnicity in autism intervention literature: Data from a large-scale systematic review of evidence-based practices. Autism: The International Journal of Research and Practice, 26(8), 2026–2040. https://doi.org/10.1177/13623613211072593.CrossRefPubMed Steinbrenner, J. R., McIntyre, N., Rentschler, L. F., Pearson, J. N., Luelmo, P., Jaramillo, M. E., et al. (2022). Patterns in reporting and participant inclusion related to race and ethnicity in autism intervention literature: Data from a large-scale systematic review of evidence-based practices. Autism: The International Journal of Research and Practice, 26(8), 2026–2040. https://​doi.​org/​10.​1177/​1362361321107259​3.CrossRefPubMed
go back to reference Thom, R., Pereira, J., Sipsock, D., & McDougle, C. (2021a). Recent updates in psychopharmacology for the core and associated symptoms of autism spectrum disorder. Current Psychiatry Reports, 23(79). Thom, R., Pereira, J., Sipsock, D., & McDougle, C. (2021a). Recent updates in psychopharmacology for the core and associated symptoms of autism spectrum disorder. Current Psychiatry Reports, 23(79).
go back to reference Thom, R. P., Pober, B. R., & McDougle, C. J. (2021b). Psychopharmacology of Williams syndrome: Safety, tolerability, and effectiveness. Expert Opinion on Drug Safety. Taylor and Francis Ltd. https://doi.org/10.1080/14740338.2021a.1867535. Thom, R. P., Pober, B. R., & McDougle, C. J. (2021b). Psychopharmacology of Williams syndrome: Safety, tolerability, and effectiveness. Expert Opinion on Drug Safety. Taylor and Francis Ltd. https://​doi.​org/​10.​1080/​14740338.​2021a.​1867535.​
Metagegevens
Titel
Diversity of Participants in Williams Syndrome Intervention Studies
Auteurs
Eva Shin
Caitlin Ravichandran
Danielle Renzi
Barbara R. Pober
Christopher J. McDougle
Robyn P. Thom
Publicatiedatum
16-08-2023
Uitgeverij
Springer US
Gepubliceerd in
Journal of Autism and Developmental Disorders / Uitgave 10/2024
Print ISSN: 0162-3257
Elektronisch ISSN: 1573-3432
DOI
https://doi.org/10.1007/s10803-023-06088-2