Identifying a core outcome set for pulmonary sarcoidosis research - the Foundation for Sarcoidosis Research - Sarcoidosis Clinical OUtcomes Taskforce (SCOUT).

Core outcome set Outcomes Pulmonary sarcoidosis

Journal

Sarcoidosis, vasculitis, and diffuse lung diseases : official journal of WASOG
ISSN: 2532-179X
Titre abrégé: Sarcoidosis Vasc Diffuse Lung Dis
Pays: Italy
ID NLM: 9610928

Informations de publication

Date de publication:
2022
Historique:
received: 23 09 2021
accepted: 13 07 2022
entrez: 15 2 2023
pubmed: 16 2 2023
medline: 16 2 2023
Statut: ppublish

Résumé

Pulmonary sarcoidosis is a rare granulomatous disease of unknown aetiology. Heterogeneity in the outcomes measured in trials of treatment for pulmonary sarcoidosis has impacted on the ability to systematically compare findings, contributing to research inefficiency. The FSR-SCOUT study has aimed to address this heterogeneity by developing a core outcome set that represents a patient and health professional consensus on the most important outcomes to measure in future research for the treatment of pulmonary sarcoidosis. systematic review of trial registries, narrative synthesis of published qualitative literature on the patient experience and results of a patient survey contributed to the development of a comprehensive list of outcomes that were rated in a two round online Delphi survey. The Delphi survey was completed by patients/carers and health professionals and the results discussed and ratified at an online consensus meeting. 259 patients/carers and 51 health professionals completed both rounds of the Delphi survey. A pre-agreed definition of consensus was applied and the results discussed at an online consensus meeting attended by 17 patients and 7 health professionals). Fifteen outcomes, across five domains (physiological/clinical, treatment, resource use, quality of life, and death), reached the definition of consensus and were included in the core outcome set. The core outcome set represents a patient and health professional consensus on the most important outcomes for pulmonary sarcoidosis research. The use of the core outcome set in future trials, and efforts to validate its components, will enhance the relevance of trials to stakeholders and will increase the opportunity for the research to contribute to evidence synthesis.

Sections du résumé

Background UNASSIGNED
Pulmonary sarcoidosis is a rare granulomatous disease of unknown aetiology. Heterogeneity in the outcomes measured in trials of treatment for pulmonary sarcoidosis has impacted on the ability to systematically compare findings, contributing to research inefficiency. The FSR-SCOUT study has aimed to address this heterogeneity by developing a core outcome set that represents a patient and health professional consensus on the most important outcomes to measure in future research for the treatment of pulmonary sarcoidosis.
Research design and methods UNASSIGNED
systematic review of trial registries, narrative synthesis of published qualitative literature on the patient experience and results of a patient survey contributed to the development of a comprehensive list of outcomes that were rated in a two round online Delphi survey. The Delphi survey was completed by patients/carers and health professionals and the results discussed and ratified at an online consensus meeting.
Results UNASSIGNED
259 patients/carers and 51 health professionals completed both rounds of the Delphi survey. A pre-agreed definition of consensus was applied and the results discussed at an online consensus meeting attended by 17 patients and 7 health professionals). Fifteen outcomes, across five domains (physiological/clinical, treatment, resource use, quality of life, and death), reached the definition of consensus and were included in the core outcome set.
Conclusions UNASSIGNED
The core outcome set represents a patient and health professional consensus on the most important outcomes for pulmonary sarcoidosis research. The use of the core outcome set in future trials, and efforts to validate its components, will enhance the relevance of trials to stakeholders and will increase the opportunity for the research to contribute to evidence synthesis.

Identifiants

pubmed: 36791046
doi: 10.36141/svdld.v39i3.12319
pii: SVDLD-39-30
pmc: PMC9766848
doi:

Types de publication

Journal Article

Langues

eng

Pagination

e2022030

Informations de copyright

Copyright: © 2021 SARCOIDOSIS VASCULITIS AND DIFFUSE LUNG DISEASES.

Déclaration de conflit d'intérêts

NLH, SLG, PRW, RPB, MAJ, NAK, DEV, JCG, DAC have no competing interests. ESB is an employee of Janssen Research and Development, LLC, who has sponsored research studies in Sarcoidosis. EJS is an employee of Insmed Incorporated. MW employee of Janssen Research & Development. HJ is a sarcoidosis patient. TA-H, HN and NS were employed by the funder (The Foundation for Sarcoidosis Research) during the research.

Références

Lung. 2019 Apr;197(2):155-161
pubmed: 30778661
ERJ Open Res. 2018 Dec 21;4(4):
pubmed: 30588477
J Clin Epidemiol. 2011 Apr;64(4):395-400
pubmed: 21194891
Lung. 2018 Oct;196(5):569-575
pubmed: 30088094
Sarcoidosis Vasc Diffuse Lung Dis. 2021;38(3):e2021034
pubmed: 34744426
Lung. 2008;186 Suppl 1:S55-8
pubmed: 17939003
Cough. 2013 Mar 06;9(1):9
pubmed: 23497330
PLoS Med. 2016 Oct 18;13(10):e1002148
pubmed: 27755541
Cochrane Database Syst Rev. 2006 Jul 19;(3):CD003536
pubmed: 16856012
Sarcoidosis Vasc Diffuse Lung Dis. 2011 Jul;28(1):56-64
pubmed: 21796892
Chest. 2018 Aug;154(2):349-356
pubmed: 29453944
Am J Respir Crit Care Med. 2003 Aug 1;168(3):323-9
pubmed: 12738606
BMJ. 2019 Oct 22;367:l5553
pubmed: 31641045
PLoS Med. 2017 Nov 16;14(11):e1002447
pubmed: 29145404
Cochrane Database Syst Rev. 2005 Apr 18;(2):CD001114
pubmed: 15846612
Thorax. 2013 Jan;68(1):57-65
pubmed: 23065052
Trials. 2017 Jun 20;18(Suppl 3):280
pubmed: 28681707
Respiration. 2007;74(4):401-5
pubmed: 16612047
BMJ Open Respir Res. 2019 Feb 18;6(1):e000394
pubmed: 30956806
J Clin Epidemiol. 2018 Apr;96:84-92
pubmed: 29288712
Eur Respir J. 1999 Oct;14(4):735-7
pubmed: 10573213
Sarcoidosis Vasc Diffuse Lung Dis. 1996 Sep;13(2):159-66
pubmed: 8893386

Auteurs

Nicola L Harman (NL)

Department of Health Data Science, University of Liverpool, Liverpool, UK.

Sarah L Gorst (SL)

Department of Health Data Science, University of Liverpool, Liverpool, UK.

Paula R Williamson (PR)

Department of Health Data Science, University of Liverpool, Liverpool, UK.

Elliot S Barnathan (ES)

Janssen Research and Development, LLC.

Robert P Baughman (RP)

Department of Medicine, University of Cincinnati Medical Center, Cincinnati, OH, USA.

Marc A Judson (MA)

Department of Medicine, Division of Pulmonary and Critical Care Medicine, Albany Medical College, Albany, New York, USA.

Heidi Junk (H)

Patient Advocate - Foundation for Sarcoidosis Research.

Nynke A Kampstra (NA)

Dept of Value-Based Healthcare, St Antonius Hospital, Nieuwegein, The Netherlands; Radboud University Medical Center, Radboud Institute for Health Sciences, Scientific Center for Quality of Healthcare (IQ Healthcare), Nijmegen, The Netherlands.
Interstitial Lung Diseases Centre of Excellence, Department of Pulmonology, St Antonius Hospital, Nieuwegein, the Netherlands.

Eugene J Sullivan (EJ)

Insmed, Incorporated, Bridgewater NJ, USA.

David E Victorson (DE)

Northwestern University Feinberg School of Medicine, Evanston, IL, USA.

Marc K Walton (MK)

Janssen Research and Development, Titusville, NJ, USA.

Tamara Al-Hakim (T)

The Foundation for Sarcoidosis Research, Chicago, IL, USA.

Hana Nabulsi (H)

The Foundation for Sarcoidosis Research, Chicago, IL, USA.

Noopur Singh (N)

The Foundation for Sarcoidosis Research, Chicago, IL, USA.

Jan C Grutters (JC)

Interstitial Lung Diseases Centre of Excellence, Department of Pulmonology, St Antonius Hospital, Nieuwegein, the Netherlands.
Division of Heart & Lungs, University Medical Center Utrecht, Utrecht, the Netherlands.

Daniel A Culver (DA)

Department of Pulmonary, Medicine, Respiratory Institute, at Cleveland Clinic, Cleveland, OH, USA.

Classifications MeSH