Reducing global health inequalities for a rare disorder: evaluating the international Prader-Willi Syndrome Organisation's Echo® programme

Tanzil Rujeedawa; Nora McNairney; Shelly Cordner; James O'Brien; Georgina Loughnan; Anthony Holland

doi:10.1186/s13023-022-02504-5

Reducing global health inequalities for a rare disorder: evaluating the international Prader-Willi Syndrome Organisation's Echo^® programme

Orphanet J Rare Dis. 2022 Oct 21;17(1):380. doi: 10.1186/s13023-022-02504-5.

Authors

Tanzil Rujeedawa¹, Nora McNairney², Shelly Cordner², James O'Brien³, Georgina Loughnan⁴, Anthony Holland⁵

Affiliations

¹ University of Cambridge School of Clinical Medicine, Addenbrookes Hospital, Hills Road, Cambridge, CB2 0SP, UK.
² International Prader-Willi Syndrome Association, Salisbury House, Station Road, Cambridge, CB1 2LA, UK.
³ International Prader-Willi Syndrome Organisation, Churchill Fellow, Parrera, Australia.
⁴ Metabolism and Obesity Services, Royal Prince Alfred Hospital, Sydney, NSW, Australia.
⁵ Department of Psychiatry Douglas House, 18b Trumpington Road, Cambridge, CB2 8AH, UK. ajh1008@medschl.cam.ac.uk.

Abstract

Background: People with rare disorders face significant global health inequalities; the challenge is how to raise awareness and develop a nucleus of experts in a country who are then able to provide guidance to others in that country. The International Prader-Willi Syndrome Organisation (IPWSO) established Project ECHO^® with the aim of facilitating the sharing of knowledge and the building of international partnerships to reduce global health inequalities for a particular rare genetically-determined neurodevelopmental disorder, Prader-Willi Syndrome (PWS). Four different ECHO programmes were established for the following groups: (a) Individuals (usually parents) who had taken on a leadership role in their country; (b) health professionals interested in PWS; (c) professional care providers supporting children and adults with PWS; and (d) a Latin American ECHO in Spanish. The programme started in 2020 and an evaluation was undertaken after one year to determine: the extent to which IPWSO had been able to recruit and retain individuals globally; the nature and extent of any benefits gained from the sessions; and examples of how individual involvement in the programme had led to local benefits. The methods included analysing routinely kept process indicators and survey data from the attendees of one component of the programme (the Leadership ECHO), together with a qualitative analysis of survey data and recorded interviews of attendees from countries of differing socio-economic status.

Results: We describe the IPWSO ECHO programme and report on the outcomes from the evaluation of one aspect of the programme, the Leadership ECHO. Attendance of the Leadership ECHO sessions was satisfactory, with a mean of 24.7 participants, with participants attending a mean of 5.67 sessions, i.e., 30% of sessions. There was also good global reach, with individuals attending from 34 countries, although there were notable geographic regions with very limited representation. Feedback and interviews demonstrated the positive impact of the programme with some early evidence of positive developments at national level.

Conclusions: Families and professionals from countries with a range of expertise and services offered to people with PWS remained engaged throughout the ECHO programme, established networks of support and fostered the development of good practice.

Keywords: Prader–Willi syndrome; Project ECHO; Rare disorder.

Publication types

Research Support, Non-U.S. Gov't

MeSH terms

Adult
Child
Global Health
Health Status Disparities
Humans
Prader-Willi Syndrome*
Rare Diseases
Surveys and Questionnaires