Evaluation of the Effectiveness of Therapy for Anxiety in Williams Beuren Syndrome Using a Smartphone App: Protocol for a Single-Case Experiment
Bibliographic record
Abstract
BACKGROUND: Williams syndrome (WS-OMIM 194050, orphaned number: Orpha 904) is a rare condition mostly associated with intellectual disability. People with Williams syndrome are 8 times more likely to have anxiety disorders than the general population. Therapeutic solutions to treat the anxiety remain limited, particularly nonpharmacological therapy. However, cognitive behavioral therapy (CBT) has been found efficacious in managing anxiety disorders and can be used for people with intellectual disability. OBJECTIVE: This paper describes a protocol to assess the efficiency of a CBT program based on digital support for people with Williams syndrome and anxiety based on a research methodology designed for rare diseases. METHODS: We will recruit 5 individuals with Williams syndrome and anxiety. They will participate in 9 CBT sessions. Participants will perform daily self-assessments of anxiety using a digital app, which will allow for ecological and repeated evaluation of their anxiety. This digital app will provide support for each therapy session. Anxiety and quality of life will be externally assessed before and after the program and at a 3-month follow-up. This is a single-case intervention research design with multiple baselines implying repeated measures of judgment criteria. The present protocol ensures high internal validity and will help identify encouraging contributions for later clinical trials. RESULTS: Participant recruitment and data collection began in September 2019, and we project that the study findings will be available for dissemination by spring 2023. CONCLUSIONS: This study will allow the assessment of the efficiency of a CBT program based on digital support to treat anxiety in people with Williams syndrome. Finally, the program could be used as an example of nonpharmacological therapy for rare diseases. TRIAL REGISTRATION: ClinicalTrials.gov ID: NCT03827525; https://clinicaltrials.gov/ct2/show/NCT03827525. INTERNATIONAL REGISTERED REPORT IDENTIFIER (IRRID): DERR1-10.2196/44393.
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How this classification was reachedexpand
Full frame machine prediction
Teacher imitationNot calibrated prevalence, not ground truth. Human validation pending. The Gemma side is a direct model label for every work in the frame, read from the title-only record. The Codex side is a classifier learned from the 10,348 direct Codex labels and calibrated to design-weighted sample rates; fields without enough sample support carry no Codex call. Candidate is the union of the two sides; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels.
Distilled classifier scores by category (both heads)
| Category | Codex | Gemma |
|---|---|---|
| Metaresearch | 0.033 | 0.029 |
| Meta-epidemiology (narrow) | 0.005 | 0.002 |
| Meta-epidemiology (broad) | 0.005 | 0.004 |
| Bibliometrics | 0.002 | 0.001 |
| Science and technology studies | 0.004 | 0.003 |
| Scholarly communication | 0.002 | 0.002 |
| Open science | 0.004 | 0.002 |
| Research integrity | 0.006 | 0.006 |
| Insufficient payload (model declined to judge) | 0.033 | 0.007 |
Machine scores (provisional)
The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.
Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.
score_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from itClassification
machine, unvalidatedMachine predicted; a candidate call from one source (direct Gemma or distilled Codex), not a consensus.
How this classification was reached, model by model and score by score, is at the end of the page under "How this classification was reached".