Early Language Intervention Using Distance Video-Teleconferencing: A Pilot Study of Young Boys With Fragile X Syndrome and Their Mothers

McDuffie, Andrea; Oakes, Ashley; Machalicek, Wendy; Ma, Monica; Bullard, Lauren; Nelson, Sarah; Abbeduto, Leonard

doi:10.1044/2015_ajslp-14-0137

Cited by 61 publications

(41 citation statements)

References 86 publications

(106 reference statements)

Supporting

Mentioning

Contrasting

Order By: Relevance

“…Results of this double-blind placebo-controlled study should be available in 2017; however, open-label results of a FXS Cogmed feasibility study involving 17 males and females, mean age 12.49 years, were reported by Au and colleagues [94]. Learning paradigms focused on language have recently been shown to have efficacy for individuals with FXS over a wide age range (e.g., [95, 96]) and, like Cogmed, could be useful as outcome measures, with the added benefit of “boosting” the effects of pharmacological treatments.Novel adaptive behavior measures.Independent Living Scales (ILS; [97]). The ILS is a measure of adaptive behavior that assesses the ability to function independently and handle real-life situation.…”

Section: Cognitive Measuresmentioning

confidence: 99%

See 1 more Smart Citation

Updated report on tools to measure outcomes of clinical trials in fragile X syndrome

Budimirović

Berry‐Kravis

Erickson

et al. 2017

J Neurodevelop Disord

Self Cite

127

158

View full text Add to dashboard Cite

ObjectiveFragile X syndrome (FXS) has been the neurodevelopmental disorder with the most active translation of preclinical breakthroughs into clinical trials. This process has led to a critical assessment of outcome measures, which resulted in a comprehensive review published in 2013. Nevertheless, the disappointing outcome of several recent phase III drug trials in FXS, and parallel efforts at evaluating behavioral endpoints for trials in autism spectrum disorder (ASD), has emphasized the need for re-assessing outcome measures and revising recommendations for FXS.MethodsAfter performing an extensive database search (PubMed, Food and Drug Administration (FDA)/National Institutes of Health (NIH)’s www.ClinicalTrials.gov, etc.) to determine progress since 2013, members of the Working Groups who published the 2013 Report evaluated the available outcome measures for FXS and related neurodevelopmental disorders using the COSMIN grading system of levels of evidence. The latter has also been applied to a British survey of endpoints for ASD. In addition, we also generated an informal classification of outcome measures for use in FXS intervention studies as instruments appropriate to detect shorter- or longer-term changes.ResultsTo date, a total of 22 double-blind controlled clinical trials in FXS have been identified through www.ClinicalTrials.gov and an extensive literature search. The vast majority of these FDA/NIH-registered clinical trials has been completed between 2008 and 2015 and has targeted the core excitatory/inhibitory imbalance present in FXS and other neurodevelopmental disorders. Limited data exist on reliability and validity for most tools used to measure cognitive, behavioral, and other problems in FXS in these trials and other studies. Overall, evidence for most tools supports a moderate tool quality grading. Data on sensitivity to treatment, currently under evaluation, could improve ratings for some cognitive and behavioral tools. Some progress has also been made at identifying promising biomarkers, mainly on blood-based and neurophysiological measures.ConclusionDespite the tangible progress in implementing clinical trials in FXS, the increasing data on measurement properties of endpoints, and the ongoing process of new tool development, the vast majority of outcome measures are at the moderate quality level with limited information on reliability, validity, and sensitivity to treatment. This situation is not unique to FXS, since reviews of endpoints for ASD have arrived at similar conclusions. These findings, in conjunction with the predominance of parent-based measures particularly in the behavioral domain, indicate that endpoint development in FXS needs to continue with an emphasis on more objective measures (observational, direct testing, biomarkers) that reflect meaningful improvements in quality of life. A major continuous challenge is the development of measurement tools concurrently with testing drug safety and efficacy in clinical trials.

show abstract

Section: Cognitive Measuresmentioning

confidence: 99%

“…Learning paradigms focused on language have recently been shown to have efficacy for individuals with FXS over a wide age range (e.g., [95, 96]) and, like Cogmed, could be useful as outcome measures, with the added benefit of “boosting” the effects of pharmacological treatments.…”

Section: Cognitive Measuresmentioning

confidence: 99%

Updated report on tools to measure outcomes of clinical trials in fragile X syndrome

Budimirović

Berry‐Kravis

Erickson

et al. 2017

J Neurodevelop Disord

Self Cite

127

158

View full text Add to dashboard Cite

show abstract

“…follow child's lead, appropriate use of prompts, focus on child's object of interest, and linguistically map accordingly). Initial results for children with Fragile X syndrome suggest the feasibility and potential effectiveness of this approach (McDuffie et al 2015). As this strategy develops and is further refined, greater opportunities for establishing a true discourse framework will exist, allowing continuity of this and related relationship processes to emerge over time.…”

Section: Sensitive Responsiveness and Relationshipsmentioning

confidence: 99%

Early Intervention for Children with Intellectual Disabilities: An Update

Guralnick

2016

Research Intellect Disabil

126

View full text Add to dashboard Cite

This article provides an update on advances in both developmental and intervention science that have occurred in the field of early intervention for children with developmental delays in the past decade. From the perspective of developmental science, findings related to multiple and reciprocal pathways of influence on the development of children with delays that can inform early intervention practice are discussed. This is followed by a review of two prominent lines of early intervention research: promoting children's developm-ent by enhancing parent-sensitive responsiveness and improving children's cognitive and social outcomes in inclusive preschool settings. Merging policy initiati-ves with our knowledge of developmental processes is discussed as the key to accelerating progress in the development of comprehensive early intervention systems.

show abstract

“…A number of narrative reviews have concluded that therapy for childhood speech and language impairment via telepractice is feasible and valid (149,151,152,(159)(160)(161). However, there are no published systematic reviews describing the use of telepractice for paediatric speech and language screening.…”

Section: Efficacy and Adoption Of Speech Pathology Telepracticementioning

confidence: 99%

“…These studies report that SLPs are often reluctant to provide direct services due to concerns regarding the efficacy of assessment and therapy interventions delivered via telepractice. It is interesting that this perception continues despite a growing evidence base in the area of speech pathology telepractice (32,(147)(148)(149)(150)(151)(152)(153)(154)(155)(156)(157)(158)(159)(160)(161).…”

Section: Suitability Of Patient Groups and Services For Telepracticementioning

confidence: 99%

Speech and language screening for children with medical complexity: A comparison of telepractice and in-person methods

Taylor¹

View full text Add to dashboard Cite

Effective communication is vital for healthy development and successful educational and vocational outcomes. For children with medical complexity (CMC), the interaction between medical complexity and a speech and language impairment may put them at particular risk for adverse effects. It is important that these children are promptly assessed and diagnosed so that intervention can be started early. Medical complexity is rare in childhood, thus CMC usually access speech pathology services through tertiary facilities where staff have specialist experience and resources, and work as part of a coordinated tertiary team. Unfortunately these facilities are typically located in metropolitan centres, which may compromise service accessibility for CMC living in rural and remote locations.A potential solution to improve service accessibility is remote speech and language screening using telepractice. Telepractice screening allows the speech-language pathologist (SLP) to distinguish those children who need further assessment at the tertiary hospital from those with normal speech and language. This model is increasingly used for the delivery of tertiary medical services, however, there is little research to support the use of telepractice for paediatric speech and language screening, and very few studies have included CMC. Thus, the overall purpose of this research project was to investigate the role of telepractice for remote speech and language screening of CMC. To achieve this aim, two studies were carried out.The first study was an international survey of tertiary SLPs that aimed to describe access difficulties experienced by CMC, and to investigate the rate of telepractice adoption among SLPs who work with CMC. The survey was administered in 2011 (n = 46) and repeated in 2015 (n = 47). This survey identified widespread access difficulties experienced by CMC, highlighting a need for alternative service delivery models such as telepractice. Telepractice adoption among tertiary SLPs more than doubled between 2011 and 2015 (p < 0.05), and in 2015 the rates of telepractice adoption were higher (40%) than previously reported for the general speech pathology profession. Tertiary SLPs were willing to use telepractice with their CMC patients, and considered a wide range of patient groups and service types to be appropriate for telepractice delivery. However, some SLPs were reluctant to use telepractice for direct services such as screening, and a number of barriers to telepractice adoption were identified, including inability poor audio and image quality, lack of experience with telepractice, concerns about behaviour management and use of hands-on techniques, and concerns about the caregiver's inability to assist during telepractice sessions.ii The second study was an exploratory method comparison study that aimed to investigate the feasibility, validity, and acceptability of speech and language screening via telepractice for CMC.This study compared in-person speech and language screening with two telepractice screening ...

show abstract

Early Language Intervention Using Distance Video-Teleconferencing: A Pilot Study of Young Boys With Fragile X Syndrome and Their Mothers

Cited by 61 publications

References 86 publications

Updated report on tools to measure outcomes of clinical trials in fragile X syndrome

Updated report on tools to measure outcomes of clinical trials in fragile X syndrome

Early Intervention for Children with Intellectual Disabilities: An Update

Speech and language screening for children with medical complexity: A comparison of telepractice and in-person methods

Contact Info

Product

Resources

About