Williams SyndromeWILL-COPENCT03827525
Cognitive and Behavioral Therapy of Anxiety in Williams Syndrome
An observational study of Williams Syndrome and Anxiety, run by University Hospital, Montpellier at 1 site in France.
Trial summary
Patients with Williams-Beuren syndrome are eight times more likely to suffer from anxiety compared to the general population. Few therapeutic solutions are proposed to these patients. The objective of this research is to validate a cognitive and behavioral therapy anxiety protocol for patients with this syndrome. Read more about this study
- Who is it for?
- Ages 18 years and older
- Conditions the study is for
- Williams Syndrome, Anxiety
- What is being tested?
- Cognitive and Behavioral Therapy
- Type of treatment
- Lifestyle or therapy programme
- How many people
- 5 participants
- Where
- 1 siteFrance
- Study dates
- 29 Aug 2019to 29 Apr 2021
- Phase
- Observational
- Are there results?
- None posted in the registry
Key requirements
Who can take part, as listed by the sponsor. Only the study team can confirm whether you are eligible, so take this list to your doctor.
- Ages 18 years and older
- All sexes
- No healthy volunteers
- Adult, Older adult
Study population: 5 adults with a Williams Syndrome
Who may be able to join
- Diagnosis of Williams Syndrome
- Complaint about anxiety
- 18 years old and more
- Score of 7 or more at the CELF-4 (it is a scale assessing the language)
- informed consent signed
Who cannot join
- Scoring less than 7 at the CELF-4
- Hearing impairment
- Visual impairment
Trial locations and contacts
1 site in 1 country.
France1 site, 1 recruiting
- Recruiting
Arnaud de villeneuve Hospital
Montpellier, Hérault, 34295
Natacha LEHMAN, 00.33.467.33.59.72, n-lehman@gmail.com
David GENEVIEVE, d-genevieve@chu-montpellier.fr
About this study
Patients with Williams-Beuren syndrome are eight times more likely to suffer from anxiety compared to the general population. Few therapeutic solutions are proposed to these patients. The objective of this research is to validate a cognitive and behavioral therapy anxiety protocol for patients with this syndrome.
Read the detailed description
Most studies agree that anxiety disorders are more frequent in people with intellectual disabilities that in the general population. Williams syndrome (WS) is a rare disease, associated with an anxiety rate 8 times higher than in the general population. The therapeutic solutions proposed in this population remain limited, in particular concerning approaches without the use of medication. The objective of the study is to evaluate the effectiveness of a program of Cognitive and Behavioral Therapies (CBT) in Williams Syndrome (WS). For this, an ABA-type protocol will be used. It consists of evaluating the effectiveness of an interventional program longitudinally, using a single case type study. Adults patients with WS and suffering from anxiety will be recruited. They will be enrolled in a program consisting in nine sessions of a psychotherapy program targeting anxiety. There will be a pretherapy visit (day 0), nine sessions of psychotherapy (month 0 to month 5) and a visit 3 month after the end of the therapy (at month 8) : the visit of the end of the research (V10). The expected results will be to validate a psychotherapeutic program for patients with WS suffering of anxiety. This program might be extended to other patients with intellectual disability.
- Conditions
- Williams Syndrome, Anxiety
- Being tested
- Cognitive and Behavioral Therapy
- Keywords
- Williams Syndrome, Anxiety, Cognitive and Behavioral Therapy, Intellectual Disability
- Medical subject headings
- Williams Syndrome, Anxiety Disorders, Intellectual Disability
What participants receive
What participants receive. In a randomised study each person is assigned to one group by chance.
Cognitive and Behavioral TherapyBehavioural
9 sessions of Cognitive and Behavioral Therapy will be realised with the patient by a clinical psychologist. They will last between 1h and 1h30.
| Study group | Role | What they receive |
|---|---|---|
| Cognitive and Behavioral Therapy | There is no group, the study will be based on single case method. The sudy concerns 5 patients with a Williams Syndrome Receives: Behavioral: Cognitive and Behavioral Therapy |
What is being measured
The primary outcome is the main question the study is designed to answer.
Likert anxiety Scale
Repeated evaluation of his anxiety by the patient with the Likert scale. Each day, the patient quantifies his anxiety with the scale with a score from 0 to 9.
Measured: From day 0 to month 5.
Secondary outcomes (4)
Hamilton Anxiety Scale
Hamilton anxiety scale will be realised with the patient by a clinical psychologist. It is an evaluation of the anxiety based on 14 questions and observations of the patient. The questions are rated according to the severity of the symptoms: anxious mood, depressed mood, somatic symptoms...Symptoms are evaluated using 5 degrees of severity, from absence of severity to disabling intensity. The overall score ranges from 0 to 60.
Measured: This score will be obtained at the pre therapy visit (day 0), at the visit of the end of the therapy (month 5), and at the visit of the end of the research (month 8)
Inhibition score
the inhibition will be assessed with the Go/no Go test which last 3 minutes.
Measured: This score will be obtained at the pre therapy visit (day 0), at the visit of the end of the therapy (month 5), and at the visit of the end of the research (month 8)
salivary cortisol
The sample will be realised by a nurse. The level of cortisol is related to the level of stress.
Measured: This sample will be obtained at the pretherapy visit (day 0), and at the visit of the end of the research (month 8).
WHOQOL-BREF score
this scale evaluates the quality of life with 26 questions. For each question, the quality of life is evaluated from very low to very high (5 levels). It will be completed by the person of trust.
Measured: This score will be obtained at the pre therapy visit (day 0), at the visit of the end of the therapy (month 5), and at the visit of the end of the research (month 8).
Timeline
From the start of the study to its completion; the mark is today. Estimated dates are the sponsor’s plan.
- First posted1 Feb 2019
- Study start29 Aug 2019
- Primary completion29 Apr 2021 (estimated)
- Study completion29 Apr 2021 (estimated)
Resources
- Official record on ClinicalTrials.govThe registry entry this page is built from, with its full change history.
- Publications (4)Papers the sponsor has linked to this study.
- Other trials for Williams SyndromeStudies of the same condition, open and under way.
- More trials run by University Hospital, MontpellierThe sponsor's other registered studies.
- What the trial phases meanHow a treatment moves from first tests in people to approval.
More details
For doctors and researchers: how the study is designed, who runs it, and its registry IDs.
Study design
- Study type
- Observational
- Observational model
- Cohort
- Time perspective
- Prospective
- Enrolment
- 5 participants (estimated)
Sponsor and investigators
- Lead sponsor
- University Hospital, Montpellier
- Sponsor type
- University, hospital or other
- Collaborators
- Fondation Jérôme Lejeune, association autour des Williams, réseau de santé Maladies Rares
- Responsible party
- Sponsor
- Investigators
- Natacha LEHMAN, Principal investigator, Department of Medical Genetics
IDs, dates and publications
- ClinicalTrials.gov ID
- NCT03827525
- Sponsor's protocol ID
- RECHMPL18_0375
- Acronym
- WILL-COPE
- Status last verified
- Jan 2019
- First submitted
- 29 Jan 2019
- Last updated
- 18 Oct 2019
- Results posted
- No
- Data monitoring committee
- No
- FDA-regulated drug
- No
- FDA-regulated device
- No
Publications
Vereenooghe L, Langdon PE. Psychological therapies for people with intellectual disabilities: a systematic review and meta-analysis. Res Dev Disabil. 2013 Nov;34(11):4085-102. doi: 10.1016/j.ridd.2013.08.030. Epub 2013 Sep 18.
PubMed 24051363Royston R, Howlin P, Waite J, Oliver C. Anxiety Disorders in Williams Syndrome Contrasted with Intellectual Disability and the General Population: A Systematic Review and Meta-Analysis. J Autism Dev Disord. 2017 Dec;47(12):3765-3777. doi: 10.1007/s10803-016-2909-z.
PubMed 27696186Unwin G, Tsimopoulou I, Kroese BS, Azmi S. Effectiveness of cognitive behavioural therapy (CBT) programmes for anxiety or depression in adults with intellectual disabilities: A review of the literature. Res Dev Disabil. 2016 Apr-May;51-52:60-75. doi: 10.1016/j.ridd.2015.12.010. Epub 2016 Jan 22.
PubMed 26803286Lehman 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 Apr 3;12:e44393. doi: 10.2196/44393.
PubMed 37010888
Study at a glance
From the registry record, last updated by the sponsor on 18 Oct 2019. Recruitment status is as the sponsor reported it: confirm with the study team.
- Official title
- Cognitive and Behavioral Therapy of Anxiety in Williams Syndrome
- ClinicalTrials.gov ID
- NCT03827525
- Acronym / sponsor ID
- WILL-COPE / RECHMPL18_0375
- Conditions
- Williams Syndrome, Anxiety
- Keywords
- Williams Syndrome, Anxiety, Cognitive and Behavioral Therapy, Intellectual Disability
- Study type
- Observational
- Recruitment status
- Unknown status
- Enrolment
- 5 participants (estimated)
- Lead sponsor
- University Hospital, MontpellierUniversity, hospital or other
- Collaborators
- Fondation Jérôme Lejeune, association autour des Williams, réseau de santé Maladies Rares
- Study start
- 29 Aug 2019 (actual)
- Primary completion
- 29 Apr 2021 (estimated)
- Study completion
- 29 Apr 2021 (estimated)
- First posted
- 1 Feb 2019 (actual)
- Results
- Not posted in the source record
Clinical summary
The research question as the sponsor registered it. This describes what the study plans to test, not what it found.
Patients with Williams-Beuren syndrome are eight times more likely to suffer from anxiety compared to the general population. Few therapeutic solutions are proposed to these patients. The objective of this research is to validate a cognitive and behavioral therapy anxiety protocol for patients with this syndrome.
Detailed description
Most studies agree that anxiety disorders are more frequent in people with intellectual disabilities that in the general population. Williams syndrome (WS) is a rare disease, associated with an anxiety rate 8 times higher than in the general population. The therapeutic solutions proposed in this population remain limited, in particular concerning approaches without the use of medication. The objective of the study is to evaluate the effectiveness of a program of Cognitive and Behavioral Therapies (CBT) in Williams Syndrome (WS). For this, an ABA-type protocol will be used. It consists of evaluating the effectiveness of an interventional program longitudinally, using a single case type study. Adults patients with WS and suffering from anxiety will be recruited. They will be enrolled in a program consisting in nine sessions of a psychotherapy program targeting anxiety. There will be a pretherapy visit (day 0), nine sessions of psychotherapy (month 0 to month 5) and a visit 3 month after the end of the therapy (at month 8) : the visit of the end of the research (V10). The expected results will be to validate a psychotherapeutic program for patients with WS suffering of anxiety. This program might be extended to other patients with intellectual disability.
Study design
- Observational model
- Cohort
- Time perspective
- Prospective
- Number of arms
- 1
| Arm | Type | Receives |
|---|---|---|
| Cognitive and Behavioral Therapy | – | Behavioral: Cognitive and Behavioral TherapyThere is no group, the study will be based on single case method. The sudy concerns 5 patients with a Williams Syndrome |
Who may qualify
- Ages
- 18 years and older
- Sex
- All
- Healthy volunteers
- Not accepted
- Age groups
- Adult, Older adult
- Study population
- 5 adults with a Williams Syndrome
Key inclusion criteria (5 listed)
- Diagnosis of Williams Syndrome
- Complaint about anxiety
- 18 years old and more
- Score of 7 or more at the CELF-4 (it is a scale assessing the language)
- informed consent signed
Key exclusion criteria (3 listed)
- Scoring less than 7 at the CELF-4
- Hearing impairment
- Visual impairment
Eligibility is decided by the study team after screening; this overview does not establish that a patient qualifies. Full inclusion and exclusion criteria.
Interventions
| Intervention | Type | Arms |
|---|---|---|
| Cognitive and Behavioral Therapy9 sessions of Cognitive and Behavioral Therapy will be realised with the patient by a clinical psychologist. They will last between 1h and 1h30. | Behavioural | Cognitive and Behavioral Therapy |
Primary outcome
Likert anxiety Scale
Time frame: From day 0 to month 5.
Repeated evaluation of his anxiety by the patient with the Likert scale. Each day, the patient quantifies his anxiety with the scale with a score from 0 to 9.
4 secondary outcomes
Hamilton Anxiety Scale
Time frame: This score will be obtained at the pre therapy visit (day 0), at the visit of the end of the therapy (month 5), and at the visit of the end of the research (month 8)
Inhibition score
Time frame: This score will be obtained at the pre therapy visit (day 0), at the visit of the end of the therapy (month 5), and at the visit of the end of the research (month 8)
salivary cortisol
Time frame: This sample will be obtained at the pretherapy visit (day 0), and at the visit of the end of the research (month 8).
WHOQOL-BREF score
Time frame: This score will be obtained at the pre therapy visit (day 0), at the visit of the end of the therapy (month 5), and at the visit of the end of the research (month 8).
Results and safety
Results are not available in the source record.
Research links
- Official record on ClinicalTrials.govLast updated by the sponsor 18 Oct 2019.
- Background: PubMed 24051363Vereenooghe L, Langdon PE. Psychological therapies for people with intellectual disabilities: a systematic review and meta-analysis. Res Dev Disabil. 2013 Nov;34(11):4085-102. doi: 10.1016/j.ridd.2013.08.030. Epub 2013 Sep 18.
- Background: PubMed 27696186Royston R, Howlin P, Waite J, Oliver C. Anxiety Disorders in Williams Syndrome Contrasted with Intellectual Disability and the General Population: A Systematic Review and Meta-Analysis. J Autism Dev Disord. 2017 Dec;47(12):3765-3777. doi: 10.1007/s10803-016-2909-z.
- Background: PubMed 26803286Unwin G, Tsimopoulou I, Kroese BS, Azmi S. Effectiveness of cognitive behavioural therapy (CBT) programmes for anxiety or depression in adults with intellectual disabilities: A review of the literature. Res Dev Disabil. 2016 Apr-May;51-52:60-75. doi: 10.1016/j.ridd.2015.12.010. Epub 2016 Jan 22.
- Derived from this study: PubMed 37010888Lehman 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 Apr 3;12:e44393. doi: 10.2196/44393.
Related trials for Williams Syndrome
RecruitingObservational study
Defining the Brain Phenotype of Children With Williams Syndrome
Williams Syndrome, Duplication
RecruitingObservational study
Rare Disease Patient Registry & Natural History Study - Coordination of Rare Diseases at Sanford
Rare Disorders, Undiagnosed Disorders
Recruiting
Characterization of the Natural History of Microduplication Syndrome 7q11.23
7q11.23 Microduplication Syndrome (7DUP), Autism Spectrum Disorder (ASD)
Enrolling by invitation
Parent and Infant Inter(X)Action Intervention (PIXI)
Fragile X Syndrome, Angelman Syndrome
RecruitingObservational study
Characterization and Natural History of Williams Syndrome and Other Chromosome 7q11.23 Variants
Williams Beuren Syndrome, Williams Syndrome
Referral and contact
AzTrials does not enrol participants or take referrals. Contact the study team or a site listed below; they decide who can take part. This study is not recruiting at the moment.
Natacha LEHMAN principal investigator
Department of Medical Genetics
1 site in 1 country: see every site and its status.
Questions about this trial
What is this clinical trial studying?
An observational study of Williams Syndrome and Anxiety, run by University Hospital, Montpellier at 1 site in France.
The full registered description is in the About this study section and the official record.
Who may be eligible to take part?
The sponsor lists 5 inclusion and 3 exclusion criteria. Eligibility is decided by the study team after screening, so read the full criteria and contact a listed site.
Is this clinical trial currently recruiting?
The record says: status not confirmed, as of the sponsor's last update on 18 Oct 2019. Status can change between updates, so confirm with the study contact before making plans.
Where is the study taking place?
The record lists 1 site in France. Whether a site is taking part right now should be confirmed with that site. See every site.
What are the possible benefits and risks?
They depend on the study and the treatment, and no study can promise a benefit. The study team explains the known risks during informed consent, before anyone joins; the record itself does not list them.
Are results available for this study?
Results are not available in the source record.
How can a doctor refer a patient or contact the research team?
Contact David GENEVIEVE, MH PD (04 67 33 61 04, d-genevieve@chu-montpellier.fr), or a site near the patient. AzTrials does not enrol participants or take referrals.
What does the primary outcome measure?
Likert anxiety Scale (time frame: From day 0 to month 5.). It is what the study was designed to measure, not a finding.
Where can I read the original study information?
On the ClinicalTrials.gov record for NCT03827525, which also lists 4 linked publications.
Does a listing mean the treatment is approved or proven to work?
No. A listing means the study is registered. It does not show that the treatment is safe, works, or is approved by a regulator.
How often is this information updated?
AzTrials copies changed records from ClinicalTrials.gov every night and reloads the whole registry every week. The sponsor last updated this record on 18 Oct 2019.