Patient and caregiver involvement in the formulation of guideline questions

Article type
Authors
Giordano A1, Köpke S2, Veronese S3, Rahn AC4, Kleiter I5, Basedow-Rajwich B5, Fornari A6, Battaglia MA7, Drulovic J8, Kooij L9, Koops J9, Mens J9, Meza Murillo ER10, Milanov I11, Milo R12, Patti F13, Pekmezovic T14, Sastre-Garriga J10, Vosburgh J15, Voltz R16, Bay J17, Oliver DJ18, Solari A6
1Unit of Neuroepidemiology, Foundation IRCCS Neurological Institute C. Besta/Department of Psychology, University of Turin
2Institute of Social Medicine and Epidemiology, University of Lübeck
3FARO Charitable Foundation
4Institut für Neuroimmunologie und Multiple Sklerose (INIMS), Universitätsklinikum Hamburg-Eppendorf
5Kempfenhausen Centre for Treatment of Multiple Sclerosis, Marianne-Strauß-Klinik
6Unit of Neuroepidemiology, Foundation IRCCS Neurological Institute C. Besta
7Department of Life Sciences, University of Siena
8Clinic of Neurology, CSS, Faculty of Medicine, University Hospital of Belgrade
9Nieuw Unicum
10MS Centre of Catalonia (Cemcat), University Hospital Vall d'Hebron
11Neurology Clinic, Medical University of Sofia
12Department of Neurology, Barzilai Medical Center
13Neurology Clinic, Multiple Sclerosis Centre, University Hospital Policlinico Vittorio Emanuele
14Institute of Epidemiology, Faculty of Medicine, University of Belgrade
15Israel Multiple Sclerosis Society,
16Department of Palliative Medicine, University Hospital Cologne
17Multiple Sclerosis International Federation, People with MS Advisory Committee
18The Tizard Centre, University of Kent
Abstract
Background:
Patient and public involvement in clinical practice guideline development is recommended to increase the trustworthiness and relevance of guidelines.

Objective:
To engage multiple sclerosis (MS) patients and caregivers in the definition of key questions to be answered in the EAN Guideline on Palliative Care of People with Severe MS.

Methods:
We used a mixed-methods approach: an international, online survey launched by the national MS societies of eight countries (after pilot testing/debriefing on 20 MS patients and 18 caregivers) and focus group meetings (FGMs) of Italian and German MS patients and caregivers.

Results:
Of 1199 participants, 951 (79%) completed the whole online survey and we analysed the data of 934 participants from seven countries: 751 (80%) were MS patients (74% women, mean age 46.1) and 183 (20%) were caregivers (36% spouses/partners, 72% women, mean age 47.4). Participants agreed/strongly agreed on inclusion of the nine pre-specified topics (from 89% for 'advance care planning' to 98% for 'multidisciplinary rehabilitation'). Less than 5% replied "I prefer not to answer" to any topic. There were 569 free comments: 182 (32%) on the pre-specified topics, 227 (40%) on additional topics (16 guideline-pertinent) and 160 (28%) on outcomes. Five FGMs (three of MS patients, two of caregivers; overall 35 participants) corroborated the survey findings. In addition, they allowed the explanation of the guideline production process, and the exploration of patient-important outcomes and taxing issues.

Conclusions:
MS patient and caregiver involvement was resource and time-intensive, but rewarding. It was key for the formulation of the 10 guideline questions and for the identification of patient-important outcomes.

Patient or healthcare consumer involvement:
An initial and fundamental guideline phase is the formulation of the clinical questions and specifying the population, intervention, comparator and outcomes (PICO). For complex healthcare interventions, formulating PICO questions is more challenging than for questions about drug interventions and no guidance currently exists. The engagement of healthcare consumers is even more important to facilitate the operational definition of these questions.