SCOUT – Sarcoidosis Outcomes Taskforce. A systematic review of outcomes to inform the development of a core outcome set for pulmonary sarcoidosis
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Keywords
Pulmonary sarcoidosis, Systematic review, Core outcome set, outcomes
Abstract
Background
Clinical trials evaluating different management strategies for pulmonary sarcoidosis may measure different outcomes. This heterogeneity in outcomes can lead to waste in research due to the inability to compare and combine data. Core outcome sets (COS) have the potential to address this issue and here we describe a systematic review of outcomes as the first step in the development of a COS for pulmonary sarcoidosis research.
Methods: A search of clinical trial registries for phase II, III and IV trials of pulmonary sarcoidosis was undertaken along with a rapid review of the patient perspective literature. Each study was screened for eligibility and outcomes extracted verbatim from the registry entry or publication then reviewed, grouped and categorised using the COMET taxonomy.
Results: 36 trial registry entries and 6 studies on patients’ perspective of pulmonary sarcoidosis were included reporting 56 and 82 unique outcomes respectively across 23 domains. The most frequently reported outcome domain was “respiratory, thoracic and mediastinal outcomes”. However, the patients’ perspective literature identified outcomes in the “personal circumstances” and “societal/carer burden” domains that were not reported in any of the included trial registrations.
Conclusions: Using both clinical trial registry data and published literature on patients’ perspective has allowed rapid review of outcomes measured and reported in pulmonary sarcoidosis research. The use of multiple sources has led to the development of a comprehensive list of outcomes that represents the first step in the development of a COS for use in future pulmonary sarcoidosis research.
References
2. James WE, Koutroumpakis E, Saha B, Nathani A, Saavedra L, Yucel RM, Judson MA: Clinical Features of Extrapulmonary Sarcoidosis Without Lung Involvement. Chest 2018, 154:349-356.
3. Williamson PR, Altman DG, Bagley H, Barnes KL, Blazeby JM, Brookes ST, Clarke M, Gargon E, Gorst S, Harman N, et al: The COMET Handbook: version 1.0. Trials 2017, 18:280.
4. Kampstra NA, Grutters JC, van Beek FT, Culver DA, Baughman RP, Renzoni EA, Wuyts W, Kouranos V, Wijsenbeek MS, Biesma DH, et al: First patient-centred set of outcomes for pulmonary sarcoidosis: a multicentre initiative. BMJ Open Respiratory Research 2019, 6:e000394.
5. Baughman RP, Barriuso R, Beyer K, Boyd J, Hochreiter J, Knoet C, Martone F, Quadder B, Richardson J, Spitzer G, et al: Sarcoidosis: patient treatment priorities. ERJ Open Research 2018, 4:00141-02018.
6. Kirkham JJ, Davis K, Altman DG, Blazeby JM, Clarke M, Tunis S, Williamson PR: Core Outcome Set-STAndards for Development: The COS-STAD recommendations. PLOS Medicine 2017, 14:e1002447.
7. [www.clinicaltrials.gov]
8. Ganann R, Ciliska D, Thomas H: Expediting systematic reviews: methods and implications of rapid reviews. Implementation Science 2010, 5:56.
9. Gorst SL, Young B, Williamson PR, Wilding JPH, Harman NL: Incorporating patients’ perspectives into the initial stages of core outcome set development: a rapid review of qualitative studies of type 2 diabetes. BMJ Open Diabetes Research & Care 2019, 7:e000615.
10. Wong SS, Wilczynski NL, Haynes RB: Developing optimal search strategies for detecting clinically relevant qualitative studies in MEDLINE. Stud Health Technol Inform 2004, 107:311-316.
11. Dodd S, Clarke M, Becker L, Mavergames C, Fish R, Williamson PR: A taxonomy has been developed for outcomes in medical research to help improve knowledge discovery. J Clin Epidemiol 2018, 96:84-92.
12. van Helmondt SJ, Polish LB, Judson MA, Grutters JC: Patient perspectives in sarcoidosis. Curr Opin Pulm Med 2019, 25:478-483.
13. Thunold RF, Løkke A, Langballe Cohen A, Hilberg O, Bendstrup E: Patient Reported Outcome Measures (PROMs) in Sarcoidosis. Sarcoidosis Vasculitis and Diffuse Lung Disease 2017, 34:2-17.
14. Kirwan JR, Hewlett SE, Heiberg T, Hughes RA, Carr M, Hehir M, Kvien TK, Minnock P, Newman SP, Quest EM, et al: Incorporating the patient perspective into outcome assessment in rheumatoid arthritis--progress at OMERACT 7. J Rheumatol 2005, 32:2250-2256.
15. Patel AS, Siegert RJ, Creamer D, Larkin G, Maher TM, Renzoni EA, Wells AU, Higginson IJ, Birring SS: The development and validation of the King's Sarcoidosis Questionnaire for the assessment of health status. Thorax 2013, 68:57-65.
16. Prinsen CAC, Vohra S, Rose MR, Boers M, Tugwell P, Clarke M, Williamson PR, Terwee CB: How to select outcome measurement instruments for outcomes included in a “Core Outcome Set” – a practical guideline. Trials 2016, 17:449.
17. Gorst SL, Prinsen CAC, Salcher-Konrad M, Matvienko-Sikar K, Williamson PR, Terwee CB: Methods used in the selection of instruments for outcomes included in core outcome sets have improved since the publication of the COSMIN/COMET guideline. Journal of Clinical Epidemiology 2020, 125:64-75.
18. Swayze S: Helping them cope. J Psychosoc Nurs Ment Health Serv 1991, 29:35-37.
19. Victorson DE, Cella D, Grund H, Judson MA: A conceptual model of health-related quality of life in sarcoidosis. Qual Life Res 2014, 23:89-101.
20. Moor CC, van Manen MJG, van Hagen PM, Miedema JR, van den Toorn LM, Gür-Demirel Y, Berendse APC, van Laar JAM, Wijsenbeek MS: Needs, Perceptions and Education in Sarcoidosis: A Live Interactive Survey of Patients and Partners. Lung 2018, 196:569-575.
21. Voortman M, Hendriks CMR, Elfferich MDP, Bonella F, Møller J, De Vries J, Costabel U, Drent M: The Burden of Sarcoidosis Symptoms from a Patient Perspective. Lung 2019, 197:155-161.
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