scholarly journals Correction to: Expressive language sampling as a source of outcome measures for treatment studies in fragile X syndrome: feasibility, practice effects, test-retest reliability, and construct validity

2020 ◽  
Vol 12 (1) ◽  
Author(s):  
Leonard Abbeduto ◽  
Elizabeth Berry-Kravis ◽  
Audra Sterling ◽  
Stephanie Sherman ◽  
Jamie O. Edgin ◽  
...  
Keyword(s):  
Construct Validity ◽  
Fragile X Syndrome ◽  
Outcome Measures ◽  
Fragile X ◽  
Expressive Language ◽  
Practice Effects ◽  
Original Publication ◽  
Retest Reliability ◽  
Language Sampling ◽  
Test Retest Reliability

In the original publication of this article [1], the author name Leonard Abbeduto was misspelled as Leonardkk Abbeduto. The original article has been corrected.

scholarly journals Expressive language sampling as a source of outcome measures for treatment studies in fragile X syndrome: feasibility, practice effects, test-retest reliability, and construct validity

2020 ◽  
Vol 12 (1) ◽  
Author(s):  
Leonard Abbeduto ◽  
Elizabeth Berry-Kravis ◽  
Audra Sterling ◽  
Stephanie Sherman ◽  
Jamie O. Edgin ◽  
...  
Keyword(s):  
Construct Validity ◽  
Outcome Measures ◽  
Symptom Severity ◽  
Fragile X ◽  
Expressive Language ◽  
Practice Effects ◽  
Full Sample ◽  
Retest Reliability ◽  
Language Sampling ◽  
Test Retest Reliability

Abstract Background The evaluation of treatment efficacy for individuals with fragile X syndrome (FXS) or intellectual disability (ID) more generally has been hampered by the lack of adequate outcome measures. We evaluated expressive language sampling (ELS) as a procedure for generating outcome measures for treatment research in FXS. We addressed: (a) feasibility, (b) practice effects over two administrations, (c) test-retest reliability over the repeated administrations, and (d) construct validity. We addressed these issues for the full sample as well as for subgroups defined by age, IQ, and ASD status. Methods Participants were 106 individuals with FXS between ages 6 and 23 years who had IQs within the range of intellectual disability (IQ < 70). ELS procedures for collecting samples in conversation and narration were followed and analyzed separately. Five measures were derived from transcripts segmented into C-units (i.e., an independent clause and its modifiers): number of C-units per minute (talkativeness), number of different word roots (vocabulary), C-unit length in morphemes (syntax), percentage of C-units containing dysfluency (utterance planning), and percentage of C-units that were fully or partly unintelligible (articulatory quality). ELS procedures were administered twice at 4-week intervals for each participant. Standardized tests and informant reports were administered and provided measures for evaluating construct validity of ELS measures. Results We found low rates of noncompliance, suggesting the task can be completed meaningfully by most individuals with FXS, although noncompliance was higher for younger, lower IQ, and more autistic participants. Minimal practice effects and strong test-retest reliability over the 4-week interval were observed for the full sample and across the range of ages, IQs, and autism symptom severity. Evidence of convergent construct validity was observed for the measures of vocabulary, syntax, and unintelligibility for the full sample and across the range of IQ and autism symptom severity, but not for participants under age 12. Conversation and narration yielded largely similar results in all analyses. Conclusions The findings suggest that the ELS procedures are feasible and yield measures with adequate psychometric properties for a majority of 6 to 23 years with FXS who have ID. The procedures work equally well regardless of level of ID or degree of ASD severity. The procedures, however, are more challenging and have somewhat less adequate psychometric properties for individuals with FXS under age 12.

scholarly journals Spoken language outcome measures for treatment studies in Down syndrome: feasibility, practice effects, test-retest reliability, and construct validity of variables generated from expressive language sampling

2021 ◽  
Vol 13 (1) ◽  
Author(s):  
Angela John Thurman ◽  
Jamie O. Edgin ◽  
Stephanie L. Sherman ◽  
Audra Sterling ◽  
Andrea McDuffie ◽  
...  
Keyword(s):  
Construct Validity ◽  
Outcome Measures ◽  
Expressive Language ◽  
Spoken Language ◽  
Practice Effects ◽  
Short Term ◽  
Retest Reliability ◽  
Language Sampling ◽  
Language Outcome ◽  
Test Retest Reliability

Abstract Background The purpose of this study was to evaluate expressive language sampling (ELS) as a procedure for generating spoken language outcome measures for treatment research in Down syndrome (DS). We addressed (a) feasibility, (b) practice effects across two short-term administrations, (c) test-retest reliability across two short-term administrations, (d) convergent and discriminant construct validity, and (e) considered comparisons across the conversation and narration contexts. Method Participants were 107 individuals with DS between 6 and 23 years of age who presented with intellectual disability (IQ < 70). The utility of ELS procedures designed to collect samples of spoken language in conversation and narration were evaluated separately. Variables of talkativeness, vocabulary, syntax, utterance planning, and articulation quality, derived from transcripts segmented into C-units (i.e., an independent clause and its modifiers), were considered. A 4-week interval was used to assess practice effects and test-retest reliability. Standardized direct assessments and informant report measures were collected to evaluate construct validity of the ELS variables. Results Low rates of noncompliance were observed; youth who were under 12 years of age, had phrase-level speech or less, and had a 4-year-old developmental level or less were at particular risk for experiencing difficulty completing the ELS procedures. Minimal practice effects and strong test-retest reliability across the 4-week test-retest interval was observed. The vocabulary, syntax, and speech intelligibility variables demonstrated strong convergent and discriminant validity. Although significant correlations were found between the variables derived from both the conversation and narration contexts, some differences were noted. Conclusion The ELS procedures considered were feasible and yielded variables with adequate psychometric properties for most individuals with DS between 6 and 23 years old. That said, studies of outcome measures appropriate for individuals with DS with more limited spoken language skills are needed. Context differences were observed in ELS variables suggest that comprehensive evaluation of expressive language is likely best obtained when utilizing both contexts.

scholarly journals Using Telehealth-Delivered Procedures to Collect a Parent-Implemented Expressive Language Sampling Narrative Task in Monolingual and Bilingual Families With Autism Spectrum Disorder: A Pilot Study

2021 ◽  
Vol 2 ◽  
Author(s):  
Laura del Hoyo Soriano ◽  
Lauren Bullard ◽  
Cesar Hoyos Alvarez ◽  
Angela John Thurman ◽  
Leonard Abbeduto
Keyword(s):  
Autism Spectrum Disorder ◽  
Pilot Study ◽  
Outcome Measures ◽  
Expressive Language ◽  
Autism Spectrum ◽  
Spectrum Disorder ◽  
Practice Effects ◽  
Language Sampling ◽  
English Speaking ◽  
Test Retest Reliability

Language impairments are frequent, severe, and of prognostic value in autism spectrum disorder (ASD). Unfortunately, the evaluation of the efficacy of treatments targeting the language skills of those with ASD continues to be hindered by a lack of psychometrically sound outcome measures. Expressive Language Sampling (ELS) procedures offer a promising alternative to norm-referenced standardized tests for assessing expressive language in treatment studies. Until now, however, research on the validity and utility of ELS as outcome measures has been limited to administrations by a trained professional in a clinic setting and to use with English-speaking families. These limitations are a barrier for many families accessing the benefits of participation in treatment studies. The current study examines the feasibility of teaching native English-speaking parents (NESP) and native Spanish-speaking parents (NSSP) how to administer the ELS narrative task (ELS-N) to their sons and daughters with ASD (between ages 6 and 21) at home through telehealth-delivered procedures. The parent training was provided in the primary language of the participating parent (i.e., 11 NSSP and 11 NESP) and administered by the parent to the youth in the language that the parent reported to use to communicate with the youth at home (i.e., 9 Spanish and 13 English). Families were able to choose between using their own technology or be provided with the technology needed for participation. Of the 19 parents who completed the training, 16 learned to administer the ELS-N procedures. In addition, strong test-retest reliability and no practice effects over the 4-week interval were observed for ELS-N derived youth outcome measures (i.e., talkativeness, vocabulary, syntax, dysfluency, and intelligibility) for both NSSP and NESP. Results from this pilot study suggest that the home-based parent-implemented ELS-N procedures can be learned and administered at acceptable levels of fidelity by parents, with good test-retest reliability and limited practice effects observed in terms of outcome measures for youth with ASD. Implications for treatment studies and future directions are discussed.

Considerations for Evaluating and Recommending Worker Productivity Outcome Measures: An Update from the OMERACT Worker Productivity Group

2019 ◽  
Vol 46 (10) ◽  
pp. 1401-1405 ◽  
Author(s):  
Suzanne M.M. Verstappen ◽  
Diane Lacaille ◽  
Annelies Boonen ◽  
Reuben Escorpizo ◽  
Catherine Hofstetter ◽  
...  
Keyword(s):  
Construct Validity ◽  
Psychometric Properties ◽  
Outcome Measures ◽  
Worker Productivity ◽  
Single Item ◽  
Retest Reliability ◽  
Test Retest Reliability

Objective.The Outcome Measures in Rheumatology (OMERACT) Worker Productivity Group continues efforts to assess psychometric properties of measures of presenteeism.Methods.Psychometric properties of single-item and dual answer multiitem scales were assessed, as well as methods to evaluate thresholds of meaning.Results.Test-retest reliability and construct validity of single item global measures was moderate to good. The value of measuring both degree of difficulty and amount of time with difficulty in multiitems questionnaires was confirmed. Thresholds of meaning vary depending on methods and external anchors applied.Conclusion.We have advanced our understanding of the performance of presenteeism measures and have developed approaches to describing thresholds of meaning.

scholarly journals The Relationship between Expressive Language Sampling and Clinical Measures in Fragile X Syndrome and Typical Development

2020 ◽  
Vol 10 (2) ◽  
pp. 66 ◽  
Author(s):  
Rebecca C. Shaffer ◽  
Lauren Schmitt ◽  
Angela John Thurman ◽  
Leonard Abbeduto ◽  
Michael Hong ◽  
...  
Keyword(s):  
Fragile X Syndrome ◽  
Clinical Symptoms ◽  
Fragile X ◽  
Expressive Language ◽  
Autism Spectrum ◽  
Syntactic Complexity ◽  
Typical Development ◽  
Language Sampling ◽  
Wide Range ◽  
Clinical Measures

Language impairment is a core difficulty in fragile X syndrome (FXS), and yet standardized measures lack the sensitivity to assess developmental changes in the nature of these impairments. Expressive Language Sampling Narrative (ELS-N) has emerged as a promising new measure with research demonstrating its usefulness in a wide range of ages in developmental disabilities and typical development. We examined ELS-N results in FXS and age-matched typically developing (TD) controls along with cognitive, adaptive, and clinical measures. We found the groups differed significantly on all ELS-N variables. Cognitive abilities were related to lexical diversity, syntactic complexity, and unintelligibility for the FXS group, but only verbal abilities were related to syntactic complexity in TD. Autism spectrum disorder (ASD) symptomatology was related to less intelligibility in speech. Measures of hyperactivity were related to increased talkativeness and unintelligibility. In addition, FXS males in comparison to FXS females were more impaired in cognitive ability, ASD symptoms, hyperactivity, and anxiety. This study extends the previous ELS research, supporting its use in FXS research as a measure to characterize language abilities. It also demonstrates the relationships between ELS-N variables and measures of cognitive, adaptive, ASD symptoms, and clinical symptoms.

scholarly journals Social communication in fragile X syndrome: pilot examination of the Brief Observation of Social Communication Change (BOSCC)

2022 ◽  
Vol 14 (1) ◽  
Author(s):  
Rebecca Shaffer ◽  
Angela John Thurman ◽  
Lucienne Ronco ◽  
Diego Cadavid ◽  
Shane Raines ◽  
...  
Keyword(s):  
Fragile X Syndrome ◽  
Social Communication ◽  
Outcome Measure ◽  
Fragile X ◽  
Outcome Measurement ◽  
Autism Spectrum ◽  
Divergent Validity ◽  
Minimally Verbal ◽  
Retest Reliability ◽  
Test Retest Reliability

Abstract Background Social communication is a key area of difficulty in fragile X syndrome (FXS) and there are not yet adequate outcome measurement tools. Appropriate outcome measures for FXS have been identified as a key area of research interest in order to evaluate future therapeutic trials. The Brief Observation of Social Communication Change-Minimally Verbal (BOSCC-MV), an outcome measure with strong psychometrics developed for autism spectrum disorder, has promise as an outcome measure to assess social communication change with FXS participants. Methods We examined the BOSCC-MV via central coders in this multi-site-trial to assess its appropriateness for FXS. Eighteen minimally verbal males ages 3–12 years were enrolled and assessed on two consecutive days and 7 participants completed a third visit 6 months later. We examined test-retest reliability, inter-rater reliability, and both convergent and divergent validity with standard clinical measures including the Autism Diagnostic and Observation Schedule-2, Vineland 3, Social Responsiveness Scale, and the Aberrant Behavior Checklist. Results The BOSCC-MV in FXS demonstrated strong inter-rater and test-retest reliability, comparable to previous trials in idiopathic ASD. Strong convergent validity was found with Autism Diagnostic Observation Schedule-2 and Vineland-3. Divergent validity was demonstrated between BOSCC-MV and unrelated measures. Conclusions The BOSCC-MV shows promise as a FXS social communication outcome measure, warranting further large-scale evaluation.

scholarly journals Development of an Expressive Language Sampling Procedure in Fragile X Syndrome

2013 ◽  
Vol 34 (4) ◽  
pp. 245-251 ◽  
Author(s):  
Elizabeth Berry-Kravis ◽  
Emily Doll ◽  
Audra Sterling ◽  
Sara T. Kover ◽  
Susen M. Schroeder ◽  
...  
Keyword(s):  
Fragile X Syndrome ◽  
Fragile X ◽  
Expressive Language ◽  
Sampling Procedure ◽  
Language Sampling

Test-retest reliability and practice effects of a computerized neuropsychological battery: A solution-oriented approach.

2018 ◽  
Vol 30 (12) ◽  
pp. 1652-1662 ◽  
Author(s):  
Sophie J. M. Rijnen ◽  
Sophie D. van der Linden ◽  
Wilco H. M. Emons ◽  
Margriet M. Sitskoorn ◽  
Karin Gehring
Keyword(s):  
Practice Effects ◽  
Neuropsychological Battery ◽  
Retest Reliability ◽  
Test Retest Reliability ◽  
Oriented Approach

Reliability, Validity and Responsiveness of Physical Activity Monitors in Patients with Inflammatory Myopathy

Rheumatology ◽  
2021 ◽  
Author(s):  
Bonny Rockette-Wagner ◽  
Didem Saygin ◽  
Siamak Moghadam-Kia ◽  
Chester Oddis ◽  
Océane Landon-Cardinal ◽  
...  
Keyword(s):  
Physical Activity ◽  
Construct Validity ◽  
Disease Activity ◽  
Retest Reliability ◽  
Activity Monitors ◽  
Muscle Testing ◽  
Patient Reported ◽  
Physical Activity Monitors ◽  
Global Disease Activity ◽  
Test Retest Reliability

Abstract Objective Idiopathic inflammatory myopathies (IIM) cause proximal muscle weakness, which affect activities of daily living. Wearable physical activity monitors (PAMs) objectively assess continuous activity with potential clinical usefulness in IIM assessment. We examined the psychometric characteristics for PAM outcomes in IIM. Methods Adult IIM patients were prospectively evaluated (baseline, 3 and 6-months) in an observational study. A waist-worn PAM (ActiGraph GT3X-BT) assessed average step counts/min, peak 1-min cadence, and vector magnitude/min. Validated myositis core set measures (CSM) including manual muscle testing (MMT), physician global disease activity (MD global), patient global disease activity (Pt global), extra-muscular disease activity (Ex-muscular global), HAQ-DI, muscle enzymes, and patient-reported physical function were evaluated. Test-retest reliability, construct validity, and responsiveness were determined for PAM measures and CSM using Pearson correlations and other appropriate analyses. Results 50 adult IIM patients enrolled [mean (SD) age, 53.6 (±14.6); 60% female, 94% Caucasian]. PAM measures showed strong test-retest reliability, moderate-to-strong correlations at baseline with MD global (r=-0.37- -0.48), Pt-global (r=-0.43- -0.61), HAQ-DI (r=-0.47- -0.59) and MMT (r = 0.37–0.52), and strong discriminant validity for categorical MMT and HAQ-DI. Longitudinal association with MD global (r=-0.38- -0.44), MMT (r = 0.50–0.57), HAQ-DI (r=-0.45- -0.55), and functional tests (r = 0.30–0.65) were moderate-to-strong. PAM measures were responsive to MMT improvement (≥10%) and moderate-to-major improvement on ACR/EULAR myositis response criteria. Peak 1-min cadence had the largest effect size and Standardized Response Means (SRMs). Conclusion PAM measures showed promising construct validity, reliability, and longitudinal responsiveness; especially peak 1-min cadence. PAMs provide valid outcome measures for future use in IIM clinical trials.

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