Q uality of life and experiences of sarcoma trajectories (the QUEST study): protocol for an international observational cohort study on diagnostic pathways of sarcoma patients

Vicky Soomers, Ingrid Me Desar, Lonneke V van de Poll-Franse, Olga Husson, Winette Ta van der Graaf, Vicky Soomers, Ingrid Me Desar, Lonneke V van de Poll-Franse, Olga Husson, Winette Ta van der Graaf

Abstract

Introduction: Sarcomas are rare tumours with considerable heterogeneity. Early and accurate diagnosis is important to optimise patient outcomes in terms of local disease control, overall survival (OS) and health-related quality of life (HRQoL). Time to diagnosis is variable in bone as well as soft tissue sarcoma. Possible factors for a long time from first symptom to diagnosis (the total interval) include patient, tumour and healthcare characteristics, but until now the most relevant risk factors and its association with outcomes remain unknown. Our study aims to (1) quantify total interval, the time interval from first symptom until (histological) diagnosis; (2) identify factors associated with interval length and (3) determine the association between total interval and HRQoL, stage and tumour size at diagnosis, progression-free survival (PFS) and OS.

Methods and analysis: We will conduct a longitudinal, prospective, international, multicentre cohort study among patients aged ≥18 years with newly diagnosed bone or soft tissue sarcoma at eight centres (three in UK, five in The Netherlands). Patients will be asked to complete questionnaires at five points in time; one at diagnosis and at follow-up points of 3, 6, 12 and 24 months. Questionnaire data is collected within the Patient Reported Outcomes Following Initial treatment and Long term Evaluation of Survivorship (PROFILES) registry: an international data management system for collection of patient-reported outcomes. Clinical data will be extracted from patient records. The primary endpoint is HRQoL at diagnosis, measured with the EORTC QLQ-C30. Secondary endpoints are stage and tumour size at diagnosis, PFS, OS, additional patient-reported outcomes, such as quality-adjusted life years and psychological distress.

Ethics and dissemination: Ethical approval was given by the Health Research Authority and Research Ethics Committee for the United Kingdom (18/WA/0096) and medical ethical committee of Radboudumc for The Netherlands (2017-3881). Results will be presented in peer-reviewed journals and presented at meetings.

Trial registration number: NCT03441906.

Keywords: bone diseases; health policy; oncology; sarcoma.

Conflict of interest statement

Competing interests: None declared.

© Author(s) (or their employer(s)) 2020. Re-use permitted under CC BY. Published by BMJ.

Figures

Figure 1
Figure 1
Conceptual model.

References

    1. Fletcher C, et al. WHO classification of tumours of soft tissue and bone. 4th edition Geneva: WHO, 2013: 5.
    1. Stiller CA, Trama A, Serraino D, et al. . Descriptive epidemiology of sarcomas in Europe: report from the RARECARE project. Eur J Cancer 2013;49:684–95. 10.1016/j.ejca.2012.09.011
    1. Burningham Z, Hashibe M, Spector L, et al. . The epidemiology of sarcoma. Clin Sarcoma Res 2012;2:14. 10.1186/2045-3329-2-14
    1. Blay J-Y, Coindre J-M, Ducimetière F, et al. . The value of research collaborations and consortia in rare cancers. Lancet Oncol 2016;17:e62–9. 10.1016/S1470-2045(15)00388-5
    1. DeSantis CE, Kramer JL, Jemal A. The burden of rare cancers in the United States. CA Cancer J Clin 2017;67:261–72. 10.3322/caac.21400
    1. McPhail S, Johnson S, Greenberg D, et al. . Stage at diagnosis and early mortality from cancer in England. Br J Cancer 2015;112 Suppl 1:S108–15. 10.1038/bjc.2015.49
    1. Brouns F, Stas M, De Wever I. Delay in diagnosis of soft tissue sarcomas. Eur J Surg Oncol 2003;29:440–5. 10.1016/S0748-7983(03)00006-4
    1. Olesen F, Hansen RP, Vedsted P. Delay in diagnosis: the experience in Denmark. Br J Cancer 2009;101 Suppl 2:S5–8. 10.1038/sj.bjc.6605383
    1. Weller D, Vedsted P, Rubin G, et al. . The Aarhus statement: improving design and reporting of studies on early cancer diagnosis. Br J Cancer 2012;106:1262–7. 10.1038/bjc.2012.68
    1. Soomers V, Husson O, Young R, et al. . The sarcoma diagnostic interval: a systematic review on length, contributing factors and patient outcomes. ESMO Open 2020;5:e000592. 10.1136/esmoopen-2019-000592
    1. Zhou Y, Abel GA, Hamilton W, et al. . Diagnosis of cancer as an emergency: a critical review of current evidence. Nat Rev Clin Oncol 2017;14:45–56. 10.1038/nrclinonc.2016.155
    1. Grimer RJ. Size matters for sarcomas! Ann R Coll Surg Engl 2006;88:519–24. 10.1308/003588406X130651
    1. George A, Grimer R. Early symptoms of bone and soft tissue sarcomas: could they be diagnosed earlier? Ann R Coll Surg Engl 2012;94:261–6. 10.1308/003588412X13171221590016
    1. Bottomley A, Pe M, Sloan J, et al. . Analysing data from patient-reported outcome and quality of life endpoints for cancer clinical trials: a start in setting international standards. Lancet Oncol 2016;17:e510–4. 10.1016/S1470-2045(16)30510-1
    1. Winnette R, Hess LM, Nicol SJ, et al. . The patient experience with soft tissue sarcoma: a systematic review of the literature. Patient 2017;10:153–62. 10.1007/s40271-016-0200-1
    1. McDonough J, Eliott J, Neuhaus S, et al. . Health-Related quality of life, psychosocial functioning, and unmet health needs in patients with sarcoma: a systematic review. Psychooncology 2019;28:653–64. 10.1002/pon.5007
    1. Storey L, Fern LA, Martins A, et al. . A critical review of the impact of sarcoma on psychosocial wellbeing. Sarcoma 2019;2019:1–18. 10.1155/2019/9730867
    1. Walming S, Block M, Bock D, et al. . Timely access to care in the treatment of rectal cancer and the effect on quality of life. Colorectal Dis 2018;20:126–33. 10.1111/codi.13836
    1. Labetoulle M, Rolando M, Baudouin C, et al. . Patients’ perception of DED and its relation with time to diagnosis and quality of life: an international and multilingual survey. Br J Ophthalmol 2017;101:1100–5. 10.1136/bjophthalmol-2016-309193
    1. Wilson R. The challenge of sarcomas: the patient advocacy group perspective. Clin Sarcoma Res 2019;9:11. 10.1186/s13569-019-0121-6
    1. van de Poll-Franse LV, Horevoorts N, Eenbergen Mvan, et al. . The patient reported outcomes following initial treatment and long term evaluation of survivorship registry: scope, rationale and design of an infrastructure for the study of physical and psychosocial outcomes in cancer survivorship cohorts. Eur J Cancer 2011;47:2188–94. 10.1016/j.ejca.2011.04.034
    1. Sangha O, Stucki G, Liang MH, et al. . The self-administered comorbidity questionnaire: a new method to assess comorbidity for clinical and health services research. Arthritis Rheum 2003;49:156–63. 10.1002/art.10993
    1. Fransen MP, Van Schaik TM, Twickler TB, et al. . Applicability of internationally available health literacy measures in the Netherlands. J Health Commun 2011;16 Suppl 3:134–49. 10.1080/10810730.2011.604383
    1. Verkissen MN, Ezendam NPM, Fransen MP, et al. . The role of health literacy in perceived information provision and satisfaction among women with ovarian tumors: a study from the population-based profiles registry. Patient Educ Couns 2014;95:421–8. 10.1016/j.pec.2014.03.008
    1. Chew LD, Griffin JM, Partin MR, et al. . Validation of screening questions for limited health literacy in a large VA outpatient population. J Gen Intern Med 2008;23:561–6. 10.1007/s11606-008-0520-5
    1. Ferrell BR, Dow KH, Grant M. Measurement of the quality of life in cancer survivors. Qual Life Res 1995;4:523–31. 10.1007/BF00634747
    1. van Dis FW, Mols F, Vingerhoets AJJM, et al. . A validation study of the dutch version of the quality of life-cancer survivor (QOL-CS) questionnaire in a group of prostate cancer survivors. Qual Life Res 2006;15:1607–12. 10.1007/s11136-006-0015-y
    1. Schwarzer R, Jerusalem M. Generalized aelf-efficacy scale : Weinman J, Wright S, Johnston M, Measures in health psychology: a user’s portfolio. Causal and controle beliefs. Windsor: UK:NFER-NELSON, 1995: 35–7.
    1. Carver CS. You want to measure coping but your protocol’s too long: consider the brief cope. Int J Behav Med 1997;4:92–100. 10.1207/s15327558ijbm0401_6
    1. Smith BW, Dalen J, Wiggins K, et al. . The brief resilience scale: assessing the ability to bounce back. Int J Behav Med 2008;15:194–200. 10.1080/10705500802222972
    1. Aaronson NK, Ahmedzai S, Bergman B, et al. . The European organization for research and treatment of cancer QLQ-C30: a quality-of-life instrument for use in international clinical trials in oncology. J Natl Cancer Inst 1993;85:365–76. 10.1093/jnci/85.5.365
    1. Herdman M, Gudex C, Lloyd A, et al. . Development and preliminary testing of the new five-level version of EQ-5D (EQ-5D-5L). Qual Life Res 2011;20:1727–36. 10.1007/s11136-011-9903-x
    1. Reenen Mv, Janssen B. EQ-5D-5L user guide. EuroQol, 2015 (version 2.1, 2015: 1–28.
    1. M Versteegh M, M Vermeulen K, M A A Evers S, et al. . Dutch tariff for the five-level version of EQ-5D. Value Health 2016;19:343–52. 10.1016/j.jval.2016.01.003
    1. Devlin N, Shah K, Feng Y, et al. . Valuing health-related quality of life: an EQ-5D-5L value set for England, 2016. Available:
    1. Zigmond AS, Snaith RP. The hospital anxiety and depression scale. Acta Psychiatr Scand 1983;67:361–70. 10.1111/j.1600-0447.1983.tb09716.x
    1. Marshall G, Hays R. The patient satisfaction questionnaire short form (PSQ-18. Santa Monica, CA: RAND Corporation, 1994: 7865.
    1. Keeble S, Abel GA, Saunders CL, et al. . Variation in promptness of presentation among 10,297 patients subsequently diagnosed with one of 18 cancers: evidence from a national audit of cancer diagnosis in primary care. Int J Cancer 2014;135:1220–8. 10.1002/ijc.28763
    1. Forbes LJL, Warburton F, Richards MA, et al. . Risk factors for delay in symptomatic presentation: a survey of cancer patients. Br J Cancer 2014;111:581–8. 10.1038/bjc.2014.304
    1. van Eenbergen MCHJ, Vromans RD, Boll D, et al. . Changes in Internet use and wishes of cancer survivors: a comparison between 2005 and 2017. Cancer 2020;126:408–15. 10.1002/cncr.32524
    1. Cocks K, King MT, Velikova G, et al. . Evidence-Based guidelines for interpreting change scores for the European organisation for the research and treatment of cancer quality of life questionnaire core 30. Eur J Cancer 2012;48:1713–21. 10.1016/j.ejca.2012.02.059
    1. Cocks K, King MT, Velikova G, et al. . Evidence-based guidelines for determination of sample size and interpretation of the European organisation for the research and treatment of cancer quality of life questionnaire core 30. J Clin Oncol 2011;29:89–96. 10.1200/JCO.2010.28.0107
    1. Fayers P, et al. The EORTC QLQ-C30 scoring manual. 3rd ed Brussels: European Organisation for Research and Treatment of Cancer, 2001.
    1. Dinmohamed AG, Visser O, Verhoeven RHA, et al. . Fewer cancer diagnoses during the COVID-19 epidemic in the Netherlands. Lancet Oncol 2020;21:750–1. 10.1016/S1470-2045(20)30265-5
    1. Maringe C, Spicer J, Morris M, et al. . The impact of the COVID-19 pandemic on cancer deaths due to delays in diagnosis in England, UK: a national, population-based, modelling study. Lancet Oncol 2020;21:1023–34. 10.1016/S1470-2045(20)30388-0
    1. Sud A, Torr B, Jones ME, et al. . Effect of delays in the 2-week-wait cancer referral pathway during the COVID-19 pandemic on cancer survival in the UK: a modelling study. Lancet Oncol 2020;21:1035–44. 10.1016/S1470-2045(20)30392-2

Source: PubMed

Sponsors and Collaborators

Medical Conditions

Drug Interventions

3
Subscribe