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CBDV vs Placebo in Children and Adults up to Age 30 With Prader-Willi Syndrome (PWS)

Phase 2
Completed
Conditions
Prader-Willi Syndrome
Interventions
Drug: Placebo
Registration Number
NCT03848481
Lead Sponsor
Montefiore Medical Center
Brief Summary

This study aims to examine the feasibility and safety of cannabidivarin (CBDV) as a treatment for children and young adults with PWS.

Detailed Description

This clinical research trial aims to study the feasibility and safety of cannabidivarin (CBDV), in children and young adults with Prader-Willi Syndrome (PWS). CBDV has effects independent of CB1 and CB2 receptor activation and a good safety profile. This proposal addresses the Foundation for Prader Willi Research's PWS Research Plan: Program 1, Clinical Care Research: seeks to evaluate treatments that aim to reduce behavioral symptoms, such as irritability, in order to improve the quality of life of both the individual with PWS and their families. GW Pharmaceuticals will provide the CBDV drug and matching placebo.

Recruitment & Eligibility

Status
COMPLETED
Sex
All
Target Recruitment
14
Inclusion Criteria

Not provided

Exclusion Criteria

Not provided

Study & Design

Study Type
INTERVENTIONAL
Study Design
PARALLEL
Arm && Interventions
GroupInterventionDescription
Cannabidivarin (CBDV)CBDV CompoundWeight-based dosing of 10 mg/kg/day of CBDV for 12 weeks
Matched PlaceboPlaceboWeight-based dosing of 10 mg/kg/day of placebo for 12 weeks
Primary Outcome Measures
NameTimeMethod
Aberrant Behavior Checklist-Irritability Subscale (ABC-I)from Baseline to Week 12

Change in the ABC-I score. The ABC-I is a well-characterized outcome that is accepted by the FDA for the purpose of labeling, and is one of the best and most validated outcome measures in the developmental disabilities. An inclusion cutoff of 18 or higher on the ABC-I at screening was chosen based on multiple medication trials with irritability as the primary target.

Secondary Outcome Measures
NameTimeMethod
Repetative Behavior Scale- Revised (RBS-R)from Baseline to Week 12

Change in RBS-R score (a 44-item self-report questionnaire that is used to measure the breadth or repetitive behaviors)

Children's Yale-Brown Obsessive Compulsive Scale (CY-BOCS)from Baseline to Week 12

Change in the CY-BOCS score. The CY-BOCS is 10-item clinician measure designed to assess the severity of obsessive compulsive symptoms in children and adolescents over the previous week. It consists of four primary sections including Obsessions checklist, Severity items for Obsessions, Compulsions checklist and severity items for Compulsions and a set of investigational items. Improvement on this scale was associated with improvements in caregiver quality of life in our 8-week study of intranasal oxytocin.

Hyperphagia Questionnaire for Clinical Trials (HQ-CT)from Baseline to Week 12

Change in HQ-CT score. The HQ-CT is a 9-item caregiver-reported measure of food-seeking behaviors that was used in the phase 3 beloranib trial.

Clinical Global Impression Scale - Improvement (CGI-I)from Baseline to Week 12

The CGI-I will be used as a measure of improvement and contains a 7 point scale as follows: 1 = very much improved, 2 = much improved, 3 = minimally improved, 4 = no change, 5 = minimally worse, 6 = much worse, 7 = very much worse. The CGI-I is a clinician rated global measure of improvement and has been used as a measure in previous clinical psychopharmacology trials.

Caregiver Strain Questionnaire (CSQ)from Baseline to Week 12

The Caregiver Strain questionnaire is a 21-item self-report questionnaire that was developed to assess caregiver strain for families with a child living with an emotional or behavioral disorder.

Montefiore Einstein Rigidity Scale-Revised-PWS (MERS-R-PWS)from Baseline to Week 12

The Montefiore-Einstein Rigidity Scale-Revised-PWS (MERS-R-PWS) is designed to assess three domains of rigid behavior in individuals with PWS:

1. Behavioral Rigidity (e.g., Insistence on sameness, things must be done in his/her way, etc.)

2. Cognitive Rigidity (e.g., Special interests, inflexible adherence to rules, etc.)

3. Protest (in response to deviation from rigidity; e.g., tantrum, irritability, arguing) The MERS-R-PWS is a clinician-rated scale and takes about 20 minutes to complete. It will be completed only for subjects who display rigid behaviors at baseline, week 4, week 8 and week 12.

Aberrant Behavior Checklist (ABC) subscales in lethargy/social withdrawal, stereotypic behavior, hyperactivity/noncompliance, and inappropriate speechfrom Baseline to Week 12

The ABC is an informative rating instrument that was empirically derived by principal component analysis to measure behavior in those with developmental disability and ASD. It contains 58 items that resolve into 5 subscales. The subscales and the respective number of items are as follows: (a) irritability - 15 items, (b) lethargy/social withdrawal - 16 items, (c) stereotypic behavior - 7 items, (d) hyperactivity/noncompliance - 16 items, and (e) inappropriate speech - 4 items. The ABC was designed to be completed by any adult who knows the patient well, such as a parent/caregiver or teacher. This instrument measures behavior on a four-point severity scale where 0 = no problem at all, 1 = behavior is a problem but in a slight degree, 2 = problem is moderately serious, and 3 = problem is severe in degree.

ActiGraph GT9X-BT activity monitorsfrom Baseline to Week 12

Change in Sleep Behaviors. Actigraph activity monitors are a well validated activity sleep monitoring device that has been utilized widely in clinical trials and health research, measuring sleep latency, total sleep time, and sleep efficiency.

Trial Locations

Locations (1)

Montefiore Medical Center, Albert Einstein College of Medicine

🇺🇸

Bronx, New York, United States

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