<clinical_study>
  <!-- This xml conforms to an XML Schema at:
    https://clinicaltrials.gov/ct2/html/images/info/public.xsd -->
  <required_header>
    <download_date>ClinicalTrials.gov processed this data on September 20, 2023</download_date>
    <link_text>Link to the current ClinicalTrials.gov record.</link_text>
    <url>https://clinicaltrials.gov/ct2/show/NCT03827525</url>
  </required_header>
  <id_info>
    <org_study_id>RECHMPL18_0375</org_study_id>
    <nct_id>NCT03827525</nct_id>
  </id_info>
  <brief_title>Cognitive and Behavioral Therapy of Anxiety in Williams Syndrome</brief_title>
  <acronym>WILL-COPE</acronym>
  <official_title>Cognitive and Behavioral Therapy of Anxiety in Williams Syndrome</official_title>
  <sponsors>
    <lead_sponsor>
      <agency>University Hospital, Montpellier</agency>
      <agency_class>Other</agency_class>
    </lead_sponsor>
    <collaborator>
      <agency>Fondation Jérôme Lejeune</agency>
      <agency_class>Other</agency_class>
    </collaborator>
    <collaborator>
      <agency>association autour des Williams</agency>
      <agency_class>Other</agency_class>
    </collaborator>
    <collaborator>
      <agency>réseau de santé Maladies Rares</agency>
      <agency_class>Other</agency_class>
    </collaborator>
  </sponsors>
  <source>University Hospital, Montpellier</source>
  <oversight_info>
    <has_dmc>No</has_dmc>
    <is_fda_regulated_drug>No</is_fda_regulated_drug>
    <is_fda_regulated_device>No</is_fda_regulated_device>
  </oversight_info>
  <brief_summary>
    <textblock>
      Patients with Williams-Beuren syndrome are eight times more likely to suffer from anxiety&#xD;
      compared to the general population. Few therapeutic solutions are proposed to these patients.&#xD;
      The objective of this research is to validate a cognitive and behavioral therapy anxiety&#xD;
      protocol for patients with this syndrome.&#xD;
    </textblock>
  </brief_summary>
  <detailed_description>
    <textblock>
      Most studies agree that anxiety disorders are more frequent in people with intellectual&#xD;
      disabilities that in the general population. Williams syndrome (WS) is a rare disease,&#xD;
      associated with an anxiety rate 8 times higher than in the general population. The&#xD;
      therapeutic solutions proposed in this population remain limited, in particular concerning&#xD;
      approaches without the use of medication. The objective of the study is to evaluate the&#xD;
      effectiveness of a program of Cognitive and Behavioral Therapies (CBT) in Williams Syndrome&#xD;
      (WS). For this, an ABA-type protocol will be used. It consists of evaluating the&#xD;
      effectiveness of an interventional program longitudinally, using a single case type study.&#xD;
      Adults patients with WS and suffering from anxiety will be recruited. They will be enrolled&#xD;
      in a program consisting in nine sessions of a psychotherapy program targeting anxiety. There&#xD;
      will be a pretherapy visit (day 0), nine sessions of psychotherapy (month 0 to month 5) and a&#xD;
      visit 3 month after the end of the therapy (at month 8) : the visit of the end of the&#xD;
      research (V10). The expected results will be to validate a psychotherapeutic program for&#xD;
      patients with WS suffering of anxiety. This program might be extended to other patients with&#xD;
      intellectual disability.&#xD;
    </textblock>
  </detailed_description>
  <overall_status>Unknown status</overall_status>
  <last_known_status>Recruiting</last_known_status>
  <start_date type="Actual">August 29, 2019</start_date>
  <completion_date type="Anticipated">April 29, 2021</completion_date>
  <primary_completion_date type="Anticipated">April 29, 2021</primary_completion_date>
  <study_type>Observational</study_type>
  <has_expanded_access>No</has_expanded_access>
  <study_design_info>
    <observational_model>Cohort</observational_model>
    <time_perspective>Prospective</time_perspective>
  </study_design_info>
  <primary_outcome>
    <measure>Likert anxiety Scale</measure>
    <time_frame>From day 0 to month 5.</time_frame>
    <description>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.</description>
  </primary_outcome>
  <secondary_outcome>
    <measure>Hamilton Anxiety Scale</measure>
    <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)</time_frame>
    <description>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.</description>
  </secondary_outcome>
  <secondary_outcome>
    <measure>Inhibition score</measure>
    <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)</time_frame>
    <description>the inhibition will be assessed with the Go/no Go test which last 3 minutes.</description>
  </secondary_outcome>
  <secondary_outcome>
    <measure>salivary cortisol</measure>
    <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).</time_frame>
    <description>The sample will be realised by a nurse. The level of cortisol is related to the level of stress.</description>
  </secondary_outcome>
  <secondary_outcome>
    <measure>WHOQOL-BREF score</measure>
    <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).</time_frame>
    <description>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.</description>
  </secondary_outcome>
  <number_of_groups>1</number_of_groups>
  <enrollment type="Anticipated">5</enrollment>
  <condition>Williams Syndrome</condition>
  <condition>Anxiety</condition>
  <arm_group>
    <arm_group_label>Cognitive and Behavioral Therapy</arm_group_label>
    <description>There is no group, the study will be based on single case method. The sudy concerns 5 patients with a Williams Syndrome</description>
  </arm_group>
  <intervention>
    <intervention_type>Behavioral</intervention_type>
    <intervention_name>Cognitive and Behavioral Therapy</intervention_name>
    <description>9 sessions of Cognitive and Behavioral Therapy will be realised with the patient by a clinical psychologist. They will last between 1h and 1h30.</description>
    <arm_group_label>Cognitive and Behavioral Therapy</arm_group_label>
  </intervention>
  <biospec_retention>Samples Without DNA</biospec_retention>
  <biospec_descr>
    <textblock>
      Salivary sample will be obtained to evaluate salivary cortisol. Then the sample will be&#xD;
      destroyed.&#xD;
    </textblock>
  </biospec_descr>
  <eligibility>
    <study_pop>
      <textblock>
        5 adults with a Williams Syndrome&#xD;
      </textblock>
    </study_pop>
    <sampling_method>Non-Probability Sample</sampling_method>
    <criteria>
      <textblock>
        Inclusion Criteria:&#xD;
&#xD;
          -  Diagnosis of Williams Syndrome&#xD;
&#xD;
          -  Complaint about anxiety&#xD;
&#xD;
          -  18 years old and more&#xD;
&#xD;
          -  Score of 7 or more at the CELF-4 (it is a scale assessing the language)&#xD;
&#xD;
          -  informed consent signed&#xD;
&#xD;
        Exclusion Criteria:&#xD;
&#xD;
          -  Scoring less than 7 at the CELF-4&#xD;
&#xD;
          -  Hearing impairment&#xD;
&#xD;
          -  Visual impairment&#xD;
      </textblock>
    </criteria>
    <gender>All</gender>
    <minimum_age>18 Years</minimum_age>
    <maximum_age>N/A</maximum_age>
    <healthy_volunteers>No</healthy_volunteers>
  </eligibility>
  <overall_official>
    <last_name>Natacha LEHMAN</last_name>
    <role>Principal Investigator</role>
    <affiliation>Department of Medical Genetics</affiliation>
  </overall_official>
  <overall_contact>
    <last_name>David GENEVIEVE, MH PD</last_name>
    <phone>04 67 33 61 04</phone>
    <email>d-genevieve@chu-montpellier.fr</email>
  </overall_contact>
  <overall_contact_backup>
    <last_name>Natacha LEHMAN, master 2</last_name>
    <phone>0467335972</phone>
    <email>n-lehman@chu-montpellier.fr</email>
  </overall_contact_backup>
  <location>
    <facility>
      <name>Arnaud de villeneuve Hospital</name>
      <address>
        <city>Montpellier</city>
        <state>Hérault</state>
        <zip>34295</zip>
        <country>France</country>
      </address>
    </facility>
    <status>Recruiting</status>
    <contact>
      <last_name>Natacha LEHMAN</last_name>
      <phone>00.33.467.33.59.72</phone>
      <email>n-lehman@gmail.com</email>
    </contact>
    <contact_backup>
      <last_name>David GENEVIEVE</last_name>
      <email>d-genevieve@chu-montpellier.fr</email>
    </contact_backup>
  </location>
  <location_countries>
    <country>France</country>
  </location_countries>
  <reference>
    <citation>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.</citation>
    <PMID>24051363</PMID>
  </reference>
  <reference>
    <citation>Royston 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.</citation>
    <PMID>27696186</PMID>
  </reference>
  <reference>
    <citation>Unwin 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.</citation>
    <PMID>26803286</PMID>
  </reference>
  <verification_date>January 2019</verification_date>
  <study_first_submitted>January 29, 2019</study_first_submitted>
  <study_first_submitted_qc>January 31, 2019</study_first_submitted_qc>
  <study_first_posted type="Actual">February 1, 2019</study_first_posted>
  <last_update_submitted>October 17, 2019</last_update_submitted>
  <last_update_submitted_qc>October 17, 2019</last_update_submitted_qc>
  <last_update_posted type="Actual">October 18, 2019</last_update_posted>
  <responsible_party>
    <responsible_party_type>Sponsor</responsible_party_type>
  </responsible_party>
  <keyword>Williams Syndrome</keyword>
  <keyword>Anxiety</keyword>
  <keyword>Cognitive and Behavioral Therapy</keyword>
  <keyword>Intellectual Disability</keyword>
  <condition_browse>
    <!-- CAUTION:  The following MeSH terms are assigned with an imperfect algorithm            -->
    <mesh_term>Williams Syndrome</mesh_term>
    <mesh_term>Syndrome</mesh_term>
    <mesh_term>Anxiety Disorders</mesh_term>
  </condition_browse>
  <!-- Results have not yet been posted for this study                                          -->
</clinical_study>


download date: ClinicalTrials.gov processed this data on September 20, 2023
link text: Link to the current ClinicalTrials.gov record.
url: https://clinicaltrials.gov/ct2/show/NCT03827525
org study id: RECHMPL18_0375
nct id: NCT03827525
lead sponsor: 
collaborator: 
collaborator: 
collaborator: 
has dmc: No
is fda regulated drug: No
is fda regulated device: No
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.
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.
observational model: Cohort
time perspective: Prospective
measure: Likert anxiety Scale
time frame: From day 0 to month 5.
description: 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.
measure: 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)
description: 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.
measure: 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)
description: the inhibition will be assessed with the Go/no Go test which last 3 minutes.
measure: 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).
description: The sample will be realised by a nurse. The level of cortisol is related to the level of stress.
measure: 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).
description: 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.
arm group label: Cognitive and Behavioral Therapy
description: There is no group, the study will be based on single case method. The sudy concerns 5 patients with a Williams Syndrome
intervention type: Behavioral
intervention name: Cognitive and Behavioral Therapy
description: 9 sessions of Cognitive and Behavioral Therapy will be realised with the patient by a clinical psychologist. They will last between 1h and 1h30.
arm group label: Cognitive and Behavioral Therapy
Salivary sample will be obtained to evaluate salivary cortisol. Then the sample will be
      destroyed.
study pop: 
sampling method: Non-Probability Sample
criteria: 
gender: All
minimum age: 18 Years
maximum age: N/A
healthy volunteers: No
last name: Natacha LEHMAN
role: Principal Investigator
affiliation: Department of Medical Genetics
last name: David GENEVIEVE, MH PD
phone: 04 67 33 61 04
email: d-genevieve@chu-montpellier.fr
last name: Natacha LEHMAN, master 2
phone: 0467335972
email: n-lehman@chu-montpellier.fr
facility: 
status: Recruiting
contact: 
contact backup: 
country: France
citation: 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.
PMID: 24051363
citation: Royston 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.
PMID: 27696186
citation: Unwin 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.
PMID: 26803286
responsible party type: Sponsor
mesh term: Williams Syndrome
mesh term: Syndrome
mesh term: Anxiety Disorders