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Accepted for/Published in: JMIR Research Protocols

Date Submitted: Nov 17, 2022
Open Peer Review Period: Nov 17, 2022 - Nov 24, 2022
Date Accepted: Feb 19, 2023
(closed for review but you can still tweet)

The final, peer-reviewed published version of this preprint can be found here:

Evaluation of the Effectiveness of Therapy for Anxiety in Williams Beuren Syndrome Using a Smartphone App: Protocol for a Single-Case Experiment

Lehman N, Trouillet R, Genevieve D

Evaluation of the Effectiveness of Therapy for Anxiety in Williams Beuren Syndrome Using a Smartphone App: Protocol for a Single-Case Experiment

JMIR Res Protoc 2023;12:e44393

DOI: 10.2196/44393

PMID: 37010888

PMCID: 10131632

Warning: This is an author submission that is not peer-reviewed or edited. Preprints - unless they show as "accepted" - should not be relied on to guide clinical practice or health-related behavior and should not be reported in news media as established information.

A single case experimental designed protocol to evaluate the effectiveness of anxiety therapy with a Smartphone application in Williams Beuren syndrome

  • Natacha Lehman; 
  • Raphael Trouillet; 
  • David Genevieve

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 eight times more likely to have anxiety disorders than the general population. Therapeutic solutions to treat the anxiety remain limited, particularly concerning non-pharmacological therapy. However, cognitive behavioural 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 for assessing the efficiency of a CBT program based on a digital support for people with Williams syndrome and anxiety based on a research methodology designed for rare diseases.

Methods:

Five individuals with Williams syndrome and anxiety will be recruited. They will undergo nine CBT sessions. Participants will perform daily self-assessments of anxiety by using a digital app, which will allow for ecological and repeated evaluation of the anxiety. This digital app will provide a support for each therapy session. Anxiety, and quality of life will be externally assessed before and after the program. Participants will be assessed at 3-month follow-up. This is a single-case intervention research design with multiple baselines implying repeated measures of judgement 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 it is projected that the study findings will be available for dissemination by spring 2023.

Conclusions:

This study will allow for assessing the efficiency of CBT program based on digital support to treat anxiety in people with Williams syndrome. Finally, the program could be used as an example of non-pharmacological therapy for rare diseases. Clinical Trial: ClinicalTrials.gov Protocol Identifier ID: NCT03827525


 Citation

Please cite as:

Lehman N, Trouillet R, Genevieve D

Evaluation of the Effectiveness of Therapy for Anxiety in Williams Beuren Syndrome Using a Smartphone App: Protocol for a Single-Case Experiment

JMIR Res Protoc 2023;12:e44393

DOI: 10.2196/44393

PMID: 37010888

PMCID: 10131632

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