Controlled trial of lovastatin combined with an open-label treatment of a parent-implemented language intervention in youth with fragile X syndrome / Angela John THURMAN in Journal of Neurodevelopmental Disorders, 12 (2020)  

Public ISBD [article]  inJournal of Neurodevelopmental Disorders > 12 (2020) Titre : Controlled trial of lovastatin combined with an open-label treatment of a parent-implemented language intervention in youth with fragile X syndrome Type de document : texte imprimé Auteurs : Angela John THURMAN, Auteur ; Laura A. POTTER, Auteur ; Kyoungmi KIM, Auteur ; Flora TASSONE, Auteur ; Amy BANASIK, Auteur ; Sarah Nelson POTTER, Auteur ; Lauren BULLARD, Auteur ; Vivian NGUYEN, Auteur ; Andrea MCDUFFIE, Auteur ; Randi HAGERMAN, Auteur ; Leonard ABBEDUTO, Auteur Langues : Anglais (eng) Mots-clés : Adolescent  California  Child  Communication  Double-Blind Method  Female  Fragile X Syndrome/therapy  Humans  Language  Language Therapy/methods  Lovastatin/therapeutic use  Male  Mothers/education  Outcome Assessment, Health Care  Telecommunications  Distance teleconferencing  Expressive language sampling  Fragile X syndrome  Lovastatin  Narrative storytelling  Pili  Parent-implemented language intervention  outcome measures from Fulcrum Therapeutics and the Azrieli Foundation. FT has  received funding from the Azrieli Foundation, Zynerba, and Asuragen, Inc., for  studies in FXS. RH has received funding from Zynerba, Ovid, and the Azrieli  Foundation for treatment studies in children and adults with FXS. She has also  consulted with Zynerba and Fulcrum regarding treatment studies in FXS. LA has  received funding for the development and implementation of treatment outcome  measures from the F. Hoffmann-La Roche Ltd., Roche TCRC, Inc., Neuren  Pharmaceuticals Ltd., Fulcrum Therapeutics, Azrieli Foundation, and LuMind IDSC  Foundation. Index. décimale : PER Périodiques Résumé : BACKGROUND: The purpose of this study was to conduct a 20-week controlled trial of lovastatin (10 to 40 mg/day) in youth with fragile X syndrome (FXS) ages 10 to 17 years, combined with an open-label treatment of a parent-implemented language intervention (PILI), delivered via distance video teleconferencing to both treatment groups, lovastatin and placebo. METHOD: A randomized, double-blind trial was conducted at one site in the Sacramento, California, metropolitan area. Fourteen participants were assigned to the lovastatin group; two participants terminated early from the study. Sixteen participants were assigned to the placebo group. Lovastatin or placebo was administered orally in a capsule form, starting at 10 mg and increasing weekly or as tolerated by 10 mg increments, up to a maximum dose of 40 mg daily. A PILI was delivered to both groups for 12 weeks, with 4 activities per week, through video teleconferencing by an American Speech-Language Association-certified Speech-Language Pathologist, in collaboration with a Board-Certified Behavior Analyst. Parents were taught to use a set of language facilitation strategies while interacting with their children during a shared storytelling activity. The main outcome measures included absolute change from baseline to final visit in the means for youth total number of story-related utterances, youth number of different word roots, and parent total number of story-related utterances. RESULTS: Significant increases in all primary outcome measures were observed in both treatment groups. Significant improvements were also observed in parent reports of the severity of spoken language and social impairments in both treatment groups. In all cases, the amount of change observed did not differ across the two treatment groups. Although gains in parental use of the PILI-targeted intervention strategies were observed in both treatment groups, parental use of the PILI strategies was correlated with youth gains in the placebo group and not in the lovastatin group. CONCLUSION: Participants in both groups demonstrated significant changes in the primary outcome measures. The magnitude of change observed across the two groups was comparable, providing additional support for the efficacy of the use of PILI in youth with FXS. TRIAL REGISTRATION: US National Institutes of Health (ClinicalTrials.gov), NCT02642653. Registered 12/30/2015. En ligne : https://dx.doi.org/10.1186/s11689-020-09315-4 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=573 [article] Controlled trial of lovastatin combined with an open-label treatment of a parent-implemented language intervention in youth with fragile X syndrome [texte imprimé] / Angela John THURMAN, Auteur ; Laura A. POTTER, Auteur ; Kyoungmi KIM, Auteur ; Flora TASSONE, Auteur ; Amy BANASIK, Auteur ; Sarah Nelson POTTER, Auteur ; Lauren BULLARD, Auteur ; Vivian NGUYEN, Auteur ; Andrea MCDUFFIE, Auteur ; Randi HAGERMAN, Auteur ; Leonard ABBEDUTO, Auteur. Langues : Anglais (eng) in Journal of Neurodevelopmental Disorders > 12 (2020) Mots-clés : Adolescent  California  Child  Communication  Double-Blind Method  Female  Fragile X Syndrome/therapy  Humans  Language  Language Therapy/methods  Lovastatin/therapeutic use  Male  Mothers/education  Outcome Assessment, Health Care  Telecommunications  Distance teleconferencing  Expressive language sampling  Fragile X syndrome  Lovastatin  Narrative storytelling  Pili  Parent-implemented language intervention  outcome measures from Fulcrum Therapeutics and the Azrieli Foundation. FT has  received funding from the Azrieli Foundation, Zynerba, and Asuragen, Inc., for  studies in FXS. RH has received funding from Zynerba, Ovid, and the Azrieli  Foundation for treatment studies in children and adults with FXS. She has also  consulted with Zynerba and Fulcrum regarding treatment studies in FXS. LA has  received funding for the development and implementation of treatment outcome  measures from the F. Hoffmann-La Roche Ltd., Roche TCRC, Inc., Neuren  Pharmaceuticals Ltd., Fulcrum Therapeutics, Azrieli Foundation, and LuMind IDSC  Foundation. Index. décimale : PER Périodiques Résumé : BACKGROUND: The purpose of this study was to conduct a 20-week controlled trial of lovastatin (10 to 40 mg/day) in youth with fragile X syndrome (FXS) ages 10 to 17 years, combined with an open-label treatment of a parent-implemented language intervention (PILI), delivered via distance video teleconferencing to both treatment groups, lovastatin and placebo. METHOD: A randomized, double-blind trial was conducted at one site in the Sacramento, California, metropolitan area. Fourteen participants were assigned to the lovastatin group; two participants terminated early from the study. Sixteen participants were assigned to the placebo group. Lovastatin or placebo was administered orally in a capsule form, starting at 10 mg and increasing weekly or as tolerated by 10 mg increments, up to a maximum dose of 40 mg daily. A PILI was delivered to both groups for 12 weeks, with 4 activities per week, through video teleconferencing by an American Speech-Language Association-certified Speech-Language Pathologist, in collaboration with a Board-Certified Behavior Analyst. Parents were taught to use a set of language facilitation strategies while interacting with their children during a shared storytelling activity. The main outcome measures included absolute change from baseline to final visit in the means for youth total number of story-related utterances, youth number of different word roots, and parent total number of story-related utterances. RESULTS: Significant increases in all primary outcome measures were observed in both treatment groups. Significant improvements were also observed in parent reports of the severity of spoken language and social impairments in both treatment groups. In all cases, the amount of change observed did not differ across the two treatment groups. Although gains in parental use of the PILI-targeted intervention strategies were observed in both treatment groups, parental use of the PILI strategies was correlated with youth gains in the placebo group and not in the lovastatin group. CONCLUSION: Participants in both groups demonstrated significant changes in the primary outcome measures. The magnitude of change observed across the two groups was comparable, providing additional support for the efficacy of the use of PILI in youth with FXS. TRIAL REGISTRATION: US National Institutes of Health (ClinicalTrials.gov), NCT02642653. Registered 12/30/2015. En ligne : https://dx.doi.org/10.1186/s11689-020-09315-4 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=573

Distance delivery of a parent-implemented language intervention for young boys with fragile X syndrome / Lauren BULLARD in Autism & Developmental Language Impairments, 2 (January-December 2017)  

Public ISBD [article]  inAutism & Developmental Language Impairments > 2 (January-December 2017) Titre : Distance delivery of a parent-implemented language intervention for young boys with fragile X syndrome Type de document : texte imprimé Auteurs : Lauren BULLARD, Auteur ; Andrea MCDUFFIE, Auteur ; Leonard ABBEDUTO, Auteur Langues : Anglais (eng) Index. décimale : PER Périodiques Résumé : BackgroundIn addition to significant cognitive delays, boys with fragile X syndrome display phenotypic characteristics that include delays in language, inattention, social anxiety, and escape-maintained challenging behaviors. Despite these challenges, families affected by fragile X syndrome often have limited access to center-based intervention programs.MethodsThe present study utilized a multiple baseline design across participants to examine the preliminary effectiveness of a 12-week, parent-implemented spoken language intervention for three 5- to 7-year-old boys with fragile X syndrome. The goal of the intervention was to teach the biological mothers of each boy to use a set of verbally responsive language support strategies while participating in shared book reading activities with her child. All aspects of the intervention, including pre- and post-treatment measures, were delivered into the family?s home via distance video teleconferencing.ResultsResults from this study provide preliminary support for the efficacy of this parent-implemented language intervention approach by demonstrating generalized improvements in both targeted maternal strategy use and measures of child spoken language.ConclusionsThe present study expands upon prior language intervention research utilizing distance delivery of services for families affected by fragile X syndrome. Additionally, the study provides early support for the feasibility of collecting pre- and post-treatment assessments at a distance. En ligne : https://doi.org/10.1177/2396941517728690 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=386 [article] Distance delivery of a parent-implemented language intervention for young boys with fragile X syndrome [texte imprimé] / Lauren BULLARD, Auteur ; Andrea MCDUFFIE, Auteur ; Leonard ABBEDUTO, Auteur. Langues : Anglais (eng) in Autism & Developmental Language Impairments > 2 (January-December 2017) Index. décimale : PER Périodiques Résumé : BackgroundIn addition to significant cognitive delays, boys with fragile X syndrome display phenotypic characteristics that include delays in language, inattention, social anxiety, and escape-maintained challenging behaviors. Despite these challenges, families affected by fragile X syndrome often have limited access to center-based intervention programs.MethodsThe present study utilized a multiple baseline design across participants to examine the preliminary effectiveness of a 12-week, parent-implemented spoken language intervention for three 5- to 7-year-old boys with fragile X syndrome. The goal of the intervention was to teach the biological mothers of each boy to use a set of verbally responsive language support strategies while participating in shared book reading activities with her child. All aspects of the intervention, including pre- and post-treatment measures, were delivered into the family?s home via distance video teleconferencing.ResultsResults from this study provide preliminary support for the efficacy of this parent-implemented language intervention approach by demonstrating generalized improvements in both targeted maternal strategy use and measures of child spoken language.ConclusionsThe present study expands upon prior language intervention research utilizing distance delivery of services for families affected by fragile X syndrome. Additionally, the study provides early support for the feasibility of collecting pre- and post-treatment assessments at a distance. En ligne : https://doi.org/10.1177/2396941517728690 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=386

Patterns and predictors of adaptive skills in 2- to 7-year-old children with Down syndrome / Emily K. SCHWORER in Journal of Neurodevelopmental Disorders, 14 (2022)  

Public ISBD [article]  inJournal of Neurodevelopmental Disorders > 14 (2022) Titre : Patterns and predictors of adaptive skills in 2- to 7-year-old children with Down syndrome Type de document : texte imprimé Auteurs : Emily K. SCHWORER, Auteur ; Anna J. ESBENSEN, Auteur ; Vivian NGUYEN, Auteur ; Lauren BULLARD, Auteur ; Deborah J. FIDLER, Auteur ; Lisa A. DAUNHAUER, Auteur ; Carolyn B. MERVIS, Auteur ; Angela M. BECERRA, Auteur ; Leonard ABBEDUTO, Auteur ; Angela John THURMAN, Auteur Langues : Anglais (eng) Mots-clés : Adaptation, Psychological  Autism Spectrum Disorder/psychology  Child  Child, Preschool  Down Syndrome  Humans  Motor Skills  Socialization  ASD symptoms  Adaptive skills  Cognition  Down syndrome  Motor skills  Pharmaceuticals Ltd., Fulcrum Therapeutics, and Lumind to consult on and  implement outcome measures in clinical trials for FXS and DS. AJT has received  funding from Fulcrum Therapeutics to develop outcome measures for FXS. The other  authors declare that they have no competing interests. Index. décimale : PER Périodiques Résumé : BACKGROUND: There is substantial variability in adaptive skills among individuals with Down syndrome. Few studies, however, have focused on the early developmental period or on the potential sources of variability in adaptive skills. This study characterizes adaptive skills in young children with Down syndrome and investigates child characteristics associated with adaptive skills. METHODS: Participants were 44 children with Down syndrome ranging in age from 2.50 to 7.99 years (M = 4.66 years, SD = 1.46). The Vineland Adaptive Behavior Scales-3 (VABS-3) Comprehensive Interview Form was used to assess adaptive behavior in the three core domains: socialization, daily living, and communication skills. Caregivers also reported on motor skills and autism spectrum disorder symptoms. Child cognitive abilities were assessed. RESULTS: Analyses comparing mean standard score performance across the three VABS-3 core domains demonstrated significant differences between all pairs of domains, resulting in a group-level pattern of socialization > daily living > communication skills. At the individual level, 10 different patterns of relative strength and weakness were identified, with only 18% of participants evidencing significant differences between adaptive skill domain standard scores corresponding to the group-level pattern of significant differences. Child characteristics (cognitive abilities, motor skills, and autism spectrum disorder symptoms) were significantly associated with VABS-3 adaptive domain standard scores. CONCLUSION: These findings underscore the importance of individualizing intervention programs focused on improving the adaptive skills of young children with Down syndrome based on consideration of the child's relative adaptive strengths and weaknesses. En ligne : https://dx.doi.org/10.1186/s11689-022-09430-4 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=574 [article] Patterns and predictors of adaptive skills in 2- to 7-year-old children with Down syndrome [texte imprimé] / Emily K. SCHWORER, Auteur ; Anna J. ESBENSEN, Auteur ; Vivian NGUYEN, Auteur ; Lauren BULLARD, Auteur ; Deborah J. FIDLER, Auteur ; Lisa A. DAUNHAUER, Auteur ; Carolyn B. MERVIS, Auteur ; Angela M. BECERRA, Auteur ; Leonard ABBEDUTO, Auteur ; Angela John THURMAN, Auteur. Langues : Anglais (eng) in Journal of Neurodevelopmental Disorders > 14 (2022) Mots-clés : Adaptation, Psychological  Autism Spectrum Disorder/psychology  Child  Child, Preschool  Down Syndrome  Humans  Motor Skills  Socialization  ASD symptoms  Adaptive skills  Cognition  Down syndrome  Motor skills  Pharmaceuticals Ltd., Fulcrum Therapeutics, and Lumind to consult on and  implement outcome measures in clinical trials for FXS and DS. AJT has received  funding from Fulcrum Therapeutics to develop outcome measures for FXS. The other  authors declare that they have no competing interests. Index. décimale : PER Périodiques Résumé : BACKGROUND: There is substantial variability in adaptive skills among individuals with Down syndrome. Few studies, however, have focused on the early developmental period or on the potential sources of variability in adaptive skills. This study characterizes adaptive skills in young children with Down syndrome and investigates child characteristics associated with adaptive skills. METHODS: Participants were 44 children with Down syndrome ranging in age from 2.50 to 7.99 years (M = 4.66 years, SD = 1.46). The Vineland Adaptive Behavior Scales-3 (VABS-3) Comprehensive Interview Form was used to assess adaptive behavior in the three core domains: socialization, daily living, and communication skills. Caregivers also reported on motor skills and autism spectrum disorder symptoms. Child cognitive abilities were assessed. RESULTS: Analyses comparing mean standard score performance across the three VABS-3 core domains demonstrated significant differences between all pairs of domains, resulting in a group-level pattern of socialization > daily living > communication skills. At the individual level, 10 different patterns of relative strength and weakness were identified, with only 18% of participants evidencing significant differences between adaptive skill domain standard scores corresponding to the group-level pattern of significant differences. Child characteristics (cognitive abilities, motor skills, and autism spectrum disorder symptoms) were significantly associated with VABS-3 adaptive domain standard scores. CONCLUSION: These findings underscore the importance of individualizing intervention programs focused on improving the adaptive skills of young children with Down syndrome based on consideration of the child's relative adaptive strengths and weaknesses. En ligne : https://dx.doi.org/10.1186/s11689-022-09430-4 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=574

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