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 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.

The Italian version of the Unité Rhumatologique des Affections de la Main (URAM) for Dupuytren’s disease: The URAM-I(10)

Hand Therapy ◽  
2021 ◽  
pp. 175899832110345
Author(s):  
E Lanfranchi ◽  
T Fairplay ◽  
P Arcuri ◽  
M Lando ◽  
F Marinelli ◽  
...  
Keyword(s):  
Factor Analysis ◽  
Internal Consistency ◽  
Cultural Adaptation ◽  
Dupuytren’S Disease ◽  
Divergent Validity ◽  
Dupuytren's Disease ◽  
Retest Reliability ◽  
Sf 36 ◽  
High Internal Consistency ◽  
Test Retest Reliability

Introduction Several general hand functional assessment tools for Dupuytren’s disease have been reported, but none of the patient-reported-outcome measures specific to Dupuytren’s disease-associated disabilities are available in the Italian language. The purpose of this study was to culturally adapt the Unité Rhumatologique des Affections de la Main (URAM) into Italian (URAM-I) and determine its measurement properties. Methods Cross-cultural adaptation was performed according to the current guidelines. Construct validity (convergent and divergent validity) was measured by comparing the URAM-I with the Pain-Rated Wrist/Hand Evaluation (PRWHE-I), Short-Form 36 (SF-36-I) scale and finger range of motion, respectively. Factor analysis was used to investigate the URAM-I’s internal structure. Reliability was assessed by internal consistency (Cronbach’s alpha) and test-retest reliability by Intra-Class Correlation Coefficient (ICC). Results This study included 96 patients (males = 85%, age = 66.8 ± 9.3). Due to the cultural adaptation, we divided the original item #1 into two separate items, thus generating the URAM-I(10). Convergent validity analysis showed a strong positive (r = 0.67), significant (p < 0.01) Pearson’s correlation with the PRWHE-I. Divergent validity analysis showed a weak, negative (r < 0.3) and not significant correlation with the SF-36-I subscales, except for the physical pain subscale (r = −0.21, p < 0.05). Factor analysis revealed a 2-factor, 4-item solution that explained 76% of the total variance. The URAM-I(10) demonstrated high internal consistency (α = 0.94) and high test-retest reliability (ICC = 0.97). Conclusion The URAM-I(10) demonstrates moderate construct validity, high internal consistency and test-retest reliability, and showed a 2-factor internal structure. Its evaluative use can be suggested for the Italian Dupuytren’s population.

scholarly journals Tandem Gait Test-Retest Reliability Among Healthy Child and Adolescent Athletes

2019 ◽  
Vol 54 (12) ◽  
pp. 1254-1259 ◽  
Author(s):  
David R. Howell ◽  
Anna N. Brilliant ◽  
William P. Meehan
Keyword(s):  
Dual Task ◽  
Healthy Child ◽  
Outcome Measure ◽  
Child And Adolescent ◽  
Adolescent Athletes ◽  
Single Task ◽  
Retest Reliability ◽  
Task Conditions ◽  
Test Retest Reliability ◽  
Tandem Gait

Context The tandem gait test is a method for assessing dynamic postural control and part of the Sport Concussion Assessment Tool, versions 3 and 5. However, its reliability among child and adolescent athletes has yet to be established. Objective To examine the test-retest reliability of the single-task and dual-task tandem gait test among healthy child and adolescent athletes. Design Descriptive laboratory study. Setting Sports injury-prevention center. Patients or Other Participants Uninjured and healthy athletes between the ages of 9 and 18 years. Intervention(s) Tandem gait measures repeated 3 times across the period of approximately 1 month. Main Outcome Measure(s) Participants completed the tandem gait test under single-task and dual-task (ie, while simultaneously executing a cognitive task) conditions. Our primary outcome measure was completion time during the single-task and dual-task conditions. We also assessed cognitive accuracy and response rate while participants completed the dual-task tandem gait test. Results Thirty-two child and adolescent athletes completed the study (mean age = 14.3 ± 2.4 years; females = 16). Single-task tandem gait times were similar across the 3 testing sessions (14.4 ± 4.8, 13.5 ± 4.2, and 13.8 ± 4.8 seconds; P = .45). Dual-task tandem gait times steadily improved across the test timeline (18.6 ± 6.9, 16.6 ± 4.5, and 15.8 ± 4.7 seconds; P = .02). Bivariate correlations indicated moderately high to high agreement from test 1 to test 2 (single-task r = .627; dual-task r = 0.655) and from test 2 to test 3 (single-task r = 0.852; dual-task r = 0.775). Both the single-task (intraclass correlation coefficient; ICC [3,1] = 0.86; 95% confidence interval [CI] = 0.73, 0.93) and dual-task (ICC [3,1] = 0.84; 95% CI = 0.69, 0.92) conditions demonstrated high reliability across testing sessions. Conclusions Tandem gait outcome measures demonstrated high test-retest reliability in both the single- and dual-task conditions. The overall reliability was within the acceptable range for clinical practice, but improvements across tests suggested a moderate practice effect. Tandem gait represents a reliable, dynamic, postural-control test that requires minimal space, cost, and time.

scholarly journals Impaired perceptual learning in Fragile X syndrome is mediated by parvalbumin neuron dysfunction in V1 and is reversible

2017 ◽  
Author(s):  
Anubhuti Goel ◽  
Daniel A. Cantu ◽  
Janna Guilfoyle ◽  
Gunvant R. Chaudhari ◽  
Aditi Newadkar ◽  
...  
Keyword(s):  
Perceptual Learning ◽  
Fragile X Syndrome ◽  
Visual Discrimination ◽  
Fragile X ◽  
Human Subjects ◽  
Autistic Traits ◽  
Autism Spectrum ◽  
Orientation Tuning ◽  
Mechanistic Basis

Atypical sensory processing is a core characteristic in autism spectrum disorders1 that negatively impacts virtually all activities of daily living. Sensory symptoms are predictive of the subsequent appearance of impaired social behavior and other autistic traits2, 3. Thus, a better understanding of the changes in neural circuitry that disrupt perceptual learning in autism could shed light into the mechanistic basis and potential therapeutic avenues for a range of autistic symptoms2. Likewise, the lack of directly comparable behavioral paradigms in both humans and animal models currently limits the translational potential of discoveries in the latter. We adopted a symptom-to-circuit approach to uncover the circuit-level alterations in the Fmr1-/- mouse model of Fragile X syndrome (FXS) that underlie atypical visual discrimination in this disorder4, 5. Using a go/no-go task and in vivo 2-photon calcium imaging in primary visual cortex (V1), we find that impaired discrimination in Fmr1-/- mice correlates with marked deficits in orientation tuning of principal neurons, and a decrease in the activity of parvalbumin (PV) interneurons in V1. Restoring visually evoked activity in PV cells in Fmr1-/- mice with a chemogenetic (DREADD) strategy was sufficient to rescue their behavioral performance. Finally, we found that human subjects with FXS exhibit strikingly similar impairments in visual discrimination as Fmr1-/- mice. We conclude that manipulating orientation tuning in autism could improve visually guided behaviors that are critical for playing sports, driving or judging emotions.

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