1. Health status and Impact of living with Idiopathic Pulmonary Fibrosis (IPF): UK & Ireland Delphi Survey
Anne-Marie Russell1,2,3, G. Jones4, R. Sabella5, N. Cassidy6, G. Burge7, S. Lines8, L. Stanley9, S Fletcher9, M. Weallans10 M. Bray11, S.Fleming2, TM. Maher1,2, P. Cullinan1,2
1Respiratory Epidemiology Occupational Medicine and Public Health National Heart & Lung Institute Imperial College, London/UK; 2 Respiratory Biomedical Research unit Royal Brompton Hospital London/UK;
3 National Institute Health Research Trainees Co-ordinating Centre, Leeds/UK; 4University Sheffield / UK, 5British Lung Foundation / UK, 6Irish Lung Fibrosis Association / Ireland, 7Heart of England NHS Foundation Trust / UK, 8Royal Devon & Exeter NHS Foundation Trust / UK, 9University Hospital Southampton NHS Foundation Trust 10The Pulmonary Fibrosis Trust /UK 11Action for Pulmonary fibrosis / UK,
Table 2: Complete Responses Age and Gender
Background:
In England and Wales the DH led programme supported by the Health and Social Care Information Centre (HSCIC) is expanding the clinical use of PROM’s to a range of chronic conditions, including diabetes, asthma, stroke, (COPD) and others¹. Concurrently, there has been an increase in clinical trials; growing research interest in patient experiences of living with IPF and the approval of two treatments. There is a growing need to reliably and consistently measure health status in this population and a need for an IPF Specific PRoM that is concordant with the European Medicines Agency (EMA) and US FDA criteria2 and UK NICE guidelines3.
Aim:
To develop a robust, patient centred methodological approach underpinning a patient reported outcome measure for IPF, a rare condition
To identify concepts & descriptors of IPF attributes which are important to patients
Results:
R1: Participants (n=120) rated 329 statements on a Likert scale 1-7 (‘very’ to ‘not at all’ important)
Consensus determined using criteria in Table 1.
98 items were retained for R2 and 18 additional items added from analysis of comments in R2 Repeating the process 12 items were eliminated after R2. Response rate >93%.
A greater number of items than anticipated remained resulting in too high a ratio of respondents per item
Development of a Round 3:
Aim: to recruit 300 participants
Participants were recruited through IPF charities & support groups.
An electronic link to the questionnaire was hosted on the charity websites / facebook.
Self-selecting participants, confirming they had a diagnosis of IPF, anonymously recorded their level of agreement / experience with 104 statements about IPF on 4 point Likert scales.
Questions regarding diagnosis were incorporated to maintain authenticity of disease group
IP addresses were checked to detect duplicate responses.
Results of Round 3:
Participants accessing survey n=510; Partially completing survey n=70;
Completing survey n=281; Completing hard copy n= 20;
Not required to complete survey due to a diagnosis other than IPF n=41
108 participants accessed the survey but did not proceed to the questions
Years
N (%)
≤59y
55 (19.5%)
60-69
109(39%)
70-79
93 (33%)
range ≥ 80y
24(8.5%).
Male n=181 (65%)
Female n=100 (35%)
Table 3: Geographical Distribution in R3
Region  N %
UK: South East 78
27.8
UK: Midlands 61
21.7
UK: South West 32
11.4
UK: North West 24
8.5
UK: North East 20
7.1
Ireland 14
5.0
UK: Scotland
UK: Yorkshire & Humber 13
4.6
UK: NI; Wales & other 26 9
Methodology:
Patient Centeredness (Integrated Research support Group (RSG) with defined mandate)
Literature Review (identification and deconstruction of existing measures to generate items)
Qualitative: Focus groups – x5 - London; Manchester and Bristol
Consensus: Nominal Group of ILD experts (South East)
Modified NG - Expert Interdisciplinary, Patient & Carer Group
Survey: Delphi Method x2 rounds – Patients; HCP’s and carers
Items generated from the literature; qualitative and consensus approaches were incorporated into a Delphi survey
Psychometrics: Item reduction; Factor Analysis
TRIANGULATION
OF METHODS
Discussion:
Taking a rigorous patient centred approach to item generation and reduction to inform the development of the IPF-PRoM is multifaceted. Working with IPF charities provided a feasible, reliable and an efficient way to gather a large volume of responses across the UK and Ireland in a short period of time (4 months) from patients diagnosed with IPF. This approach enables the application of appropriate psychometric analysis to these data and offers one approach for generating rich data in rare conditions
Table 1: Consensus Criteria
Statement result
Threshold to apply
Definitely include
>=70% of participants rate statement as >=6 OR Median rating of >=5
Maybe include
>=70% of participants rate statement as >=5 OR Median rating of >=5
Definitely exclude
<70% of participants rate statement as <=4 AND 100% participants understand statement OR Median <=4 AND 100% panel understand statement
Review
<70% of panel rate statement as >=6 AND <100% panel understand statement
References
Devlin N & Appleby J Getting the most out of proms: Putting health outcomes at the heart of NHS decision-making The King’s Fund 2010 Guidance on the routine collection of Patient Reported Outcome Measures (PROMs) DH:
U.S. Department of Health and Human Services Food and Drug Administration (FDA). Guidance for Industry: Patient-Reported Outcome Measures: Use in Medical Product Development to Support Labelling Claims. FDA: Maryland;
NICE Idiopathic pulmonary fibrosis in adults: diagnosis and management clinical guideline 2013 [CG163]
Acknowledgment:
This work is supported by a NIHR CDRF (Russell UK)