OBJECTIVES: To explore genetics professionals' and patients' views about which outcome domains are most appropriate to measure the patient benefits of using a clinical genetics service. METHODS: A postal Delphi survey was sent to: 115 consultant geneticists; 162 genetic counsellors; 156 support group representatives; 106 patients. The survey contained 19 outcome domains and respondents assessed the usefulness of each for clinical genetics services. RESULTS: The final professional panel comprised 115 genetics healthcare professionals and the patient panel comprised 72 patients. The outcome domains that achieved consensus (at least 75% of panel rated 'useful') for the patient and professional panels were: decision-making; knowledge of the genetic condition; perceived personal control; risk perception; satisfaction; meeting expectations; ability to cope; diagnosis accuracy; quality of life. Comparison of the ratings between the professional panel and the patient panel showed there was no statistical difference (chi(2), p<0.01) between the ratings ('useful' compared to 'not useful') for 14 of the 19 outcome domains but found differences for the perceived usefulness of: level of depression; health status; spiritual well-being; test accuracy; rate of termination. CONCLUSIONS: This Delphi survey identified nine outcome domains which are good starting points to develop a core set of outcome measures for evaluating clinical genetics services.
AimThe primary aim of this study was to explore which outcome domains patients of CGSs and genetics healthcare professionals believe are most appropriate to measure the patient benefits of using a CGS. A secondary aim was to compare the views of patients and genetics healthcare professionals.
Payne, Katherine Nicholls, Stuart G. McAllister, Marion MacLeod, Rhona Ellis, Ian Donnai, Dian Davies, Linda M.
Disease Category: Genetic disorders
Disease Name:
Age Range: 16 - 100
Sex: Either
Nature of Intervention: Any
- Clinical experts
- Consumers (patients)
- Patient/ support group representatives
- Patient perspectives
- Recommendations made
- Delphi process
A postal Delphi survey was used. This Delphi used a ‘professional’ and ‘patient’ panel.
The ‘professional panel’ comprised a purposive sample of 277 clinicians (consultants, genetic counsellors, or specialist nurses) working in clinical genetics departments. Professional panel members were identified and recruited from the 19 specialist clinical genetics departments in hospitals based in England using published information on websites and clinical colleagues. The ‘patient panel’ comprised a purposive sample of patients and patient support group representatives. This includes patients with direct CGS experience and 156 patient representatives that may have direct or indirect experience, through their support group members, of using a CGS.
The Delphi questionnaire was informed by the results of a systematic review of validated outcome measures used in CGS evaluation (see Payne et al 2007) . These 19 domains describing the core concepts from existing validated outcome measures were re-phrased using lay language. Extensive pre-pilot work with genetics healthcare professionals and representatives from patient support groups was used to develop these ‘lay descriptions’ of the outcomes to be rated
Respondents assessed the usefulness of each the 19 outcome domains as a measure of patient benefit on a rating-scale (1 = strongly disagree; 4 = neither agree nor disagree; 7 = strongly agree). Panel members gave qualitative views on each outcome domain, patients were asked about their experience of using a CGS and professionals their views about the how outcome measures could be used but these data are not reported here.
The first round of the survey was used in a pilot study administered by the means of face-to-face interviews with genetics professionals (n = 9) and patients (n = 5) or support group representatives (n = 13), to test the framing and phrasing of the questions. Qualitative comments on the survey design were used to improve the layout of the questions but the wording of questions remained similar.
This Delphi process involved two rounds. In round one, posted in November 2005, panel members rated each of the 19 outcome domains and provided qualitative comments on their usefulness. The second round survey, posted in February 2006, contained the same questions but included bar charts summarising round one results. The results from the patient and professional panels were analysed separately and reported back to the relevant panel. Panel members were given a summary of the answers they gave in the first round. Panel members were advised that the second survey gave them the opportunity to change their answers but they were not obliged to change their mind.
This study used a cut-off of 75% criterion for consensus.