Table 1 Top 10 research priorities from the PWS Mental Health Workshop

1. Obtain longitudinal and natural history data on mental health in PWS, including the behavioral and psychiatric components of the PWS phenotype. The Global PWS Registry will be a critical resource to advance mental health research since it will compile data on mental health over the lifespan.

2. Develop effective outcome measures for PWS mental health treatment studies, fully utilizing the Global PWS Registry to facilitate the development of such tools.

3. Determine the influence of weight management, hormones, and environment on mental well being over the life course, using validated measures of well-being.

4. Apply diverse research methods to advance mechanistic research on the neurobiology underlying mental health and behavioral issues in PWS.

5. Identify markers (neurobiological, genetic, psychological, environmental) of impending mental illness and characterize features of the prodromal phase (especially psychosis) in PWS, to allow mitigation and/or prevention of psychiatric episodes through environmental, behavioral or pharmacological interventions; and evaluate effectiveness of such interventions.

6. Facilitate further research on oxytocin and vasopressin in PWS in order to refine the timing and dosing of future oxytocin trials in PWS.

7. Further assess autonomic nervous system function in PWS to determine if the polyvagal theory and treatments help explain or ameliorate temper outbursts, anxiety and other behavioral problems.

8. Adapt or modify current state of the art behavioral interventions, including mindfulness, for anxiety, obsessive compulsive behaviors, and temper outbursts for PWS, and test their effectiveness over time.

9. Develop accessible methods and approaches to help reduce high levels of parental/caregiver stress, and determine effects of reducing caregiver distress/stress on child functioning.

10. Involve parents, families and caregivers (both family and professional) at all levels; Workshop attendees felt that their vast experience and engagement will be critical to advancing knowledge and treatment, with input as well from youth and adults with PWS.