Effect of Deep Propioceptive Stimulation in Prader-Willi Syndrome

Study Overview
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Brief Summary
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The main objective is to study the effect of deep propioceptive stimulation with a heavy blanket for 2 weeks compared with a placebo blanket on sleep quality and behavior in patients with PWS

Detailed Description
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Prader-Willi syndrome (PWS) is a genetic disease associated with sleep disorders whose etiology is partly central due to the inherent hypothalamic deficit. It is associated with low cognitive performance and worsening behavior with more impulsivity, tantrums, scratches and obsession for food. Deep propioceptive stimulation with a heavy blanket (HB) stimulates the parasympathetic system and improves the state of anxiety in persons with autism or other causes of intellectual disability and/or psychopathology. To date, there are no studies evaluating the effect of HB on sleep quality or brain activity in persons with PWS. The main objective is to study the effect of HB for 2 weeks compared to a placebo blanket on sleep quality and behavior in patients with SPW. Methodology: Actigraphy, questionnaires, crossover study in 60 patients with PWS, with HB/placebo blankets (15 days each). Effect of HB on brain connectivity with functional magnetic resonance imaging in 30 patients and 30 controls. Expected results: PWS group will show worse sleep quality and behavior that will improve after using HB. Brain connectivity will be different with/without HB. These favorable changes will help to justify the use of this ecological treatment and its inclusion in the NHS clinical practice guidelines for improving the quality of life of these patients and their caregivers.

Official Title
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Effect of Deep Propioceptive Stimulation on Sleep Disorders, Behavior and Brain Connectivity in Adults With Prader-Willi Syndrome

Conditions
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Sleep Disorder, Hyperphagia, Behavior Disorders

Intervention / Treatment
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* Device: Deep propioceptive stimulation


Participation Criteria
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Eligibility Criteria
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Inclusion Criteria: Patients with PWS genetically confirmed at least 16y old willing to participate and being able to be still in the MRI. Exclusion Criteria: Age < 16 years old Presence of uncontrolled Obstructive Sleep Apnea (Residual AHI ≥ 30). Narcolepsy or other sleep disorders with daily somnolence Intolerance to the heavy blanket in spite of previous adaptation sessions Active psychopathological disorder that may interfere with sleep Use of hypnotic or sleep regulator drugs (change of dose in the last 3 months) Contraindications for MRI Impossibility to stay still during MRI adquisition

Ages Eligible for Study
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Minimum Age: 16 Years

Sexes Eligible for Study
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All

Accepts Healthy Volunteers
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Accepts Healthy Volunteers

Study Plan
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How is the study designed?
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Design Details

Primary Purpose: Treatment
Allocation: Randomized
Intervention Model: Crossover Assignment
Masking: Single


Arms and Interventions

| Participant Group/Arm | Intervention/Treatment |
| --- | --- |
| Experimental: Heavy blanket<br>Patients will start with heavy blanket during 15 days | Device: Deep propioceptive stimulation<br>* Participants will start using the blanket (of the arm name), 30 days wash-out and 15d with the other blanket<br>|
| Placebo Comparator: Non-heavy blanket<br>Patients will start with non-heavy blanket during 15 days | Device: Deep propioceptive stimulation<br>* Participants will start using the blanket (of the arm name), 30 days wash-out and 15d with the other blanket<br>|

What is the study measuring?
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Primary Outcome Measures

| Outcome Measure | Measure Description | Time Frame |
| --- | --- | --- |
| Changes in number of awakenings | measured by actigraphy | 14 nights | 
| Change in duration of awakenings | minutes measured by actigraphy | 14 nights | 

Secondary Outcome Measures

| Outcome Measure | Measure Description | Time Frame |
| --- | --- | --- |
| Number of tantrums | caregivers report | 15 days | 
| number of skin injuries | physical examination by the principal investigator or collaborators | 15 days | 
| Interest for food | Hyperphagia questionnaire for clinical trials (9 items from 1-4, maximum score 36, more is worse) | 15 days | 
| Brain connectivity | functional magnetic resonance imaging | 1 day |