Cancer Res Treat.  2015 Oct;47(4):591-599. 10.4143/crt.2014.086.

Oncologist Perspectives on Rare Cancer Care: A Nationwide Survey

• Shin DW
• Cho J
• Yang HK
• Kim SY
• Lee SH
• Suh B
• Shin HY
• Lee HJ
• Kim DG
• Park JH

• KMID: 2403376
• DOI: http://doi.org/10.4143/crt.2014.086

Abstract

PURPOSE
In response to the challenges and difficulties imposed by rare cancers, multi-stakeholder initiatives dedicated to improving rare cancer care was launched, and several recommendations were made by professional societies. However, these primarily reflect the view of the advocates and supporters, and may not represent the views of the "average" clinician or researcher. In this study, we sought to investigate perceived difficulties with regard to rare cancer care and potential solutions endorsed by oncologists.
MATERIALS AND METHODS
A representative sample of 420 oncologists recruited in 13 cancer centers participated in a nationwide survey.
RESULTS
Oncologists faced various difficulties in treatment of patients with rare cancers, including the lack of clinical practice guidelines (65.7%) and personal experience (65.2%), lack of approved treatment options (39.8%), and reimbursement issues (44.5%). They were generally supportive of recent recommendations by multi-stakeholder initiatives as well as professional societies for development of clear clinical practice guidelines (66.0%), flexible reimbursement guidelines (52.9%), and a national rare cancer registry (47.4%). However, there was only moderate endorsement for referrals to high-volume centers (35.5%) and encouragement of off-label treatments (21.0%).
CONCLUSION
Insights into the general attitudes of oncologists gained through our nationwide survey of representative samples would be helpful in development of clinical practices and public health policies in rare cancer treatment and research.

Keyword

Rare diseases; Cancer; Oncologists; Attitude; Recommendation

MeSH Terms

Humans
Public Health
Rare Diseases
Referral and Consultation

Reference

References

1. Greenlee RT, Goodman MT, Lynch CF, Platz CE, Havener LA, Howe HL. The occurrence of rare cancers in U.S. adults, 1995-2004. Public Health Rep. 2010; 125:28–43.

2. National Cancer Institute Epidemiology and Genetics Research. Synergizing epidemiologic research on rare cancers [Internet]. Bethesda: National Cancer Institute Epidemiology and Genetics Research;2007. [cited 2014 Feb 1]. Available from: http://epi.grants.cancer.gov/workshops/synergizing/.

3. Gatta G, van der Zwan JM, Casali PG, Siesling S, Dei Tos AP, Kunkler I, et al. Rare cancers are not so rare: the rare cancer burden in Europe. Eur J Cancer. 2011; 47:2493–511.

4. Schaefer R. Rare Cancers Europe: joining forces to tackle a common problem. Rare Tumors. 2012; 4:e24.

5. de Vrueh R, Baekelandt ER, de Hann JM. Update on 2004 background paper: BP 6.19 rare diseases. Geneva: World Health Organization;2013.

6. Gatta G, Ciccolallo L, Kunkler I, Capocaccia R, Berrino F, Coleman MP, et al. Survival from rare cancer in adults: a population-based study. Lancet Oncol. 2006; 7:132–40.

7. Redmond K. Rare cancers in Europe: tackling a common problem [Internet]. ESO;2009. [cited 2014 Feb 1]. Available from: http://www.cancerworld.org/articles/issues_33/editorial/rare_cancers_in_europe_-_tackling_a_common_problem.html.

8. Shin DW, Park JH, Shim EJ, Hahm MI, Park JH, Park EC. Predictors and outcomes of feeling of insufficient consultation time in cancer care in Korea: results of a nationwide multicenter survey. Support Care Cancer. 2012; 20:1965–73.

9. European Society for Medical Oncology. Improving rare cancer care in Europe: recommendations on stakeholder actions and public policies [Internet]. Viganello-Lugano: European Society for Medical Oncology;c2010 [cited 2014 Feb 1]. Available from: http://www.esmo.org/content/download/16802/296577/file/improving-rare-cancer-care-europerecommendations-2010.pdf.

10. Lenaerts L, Remue E, Otter R, van Hoof E. Evaluation of the cancer plan 2008-2010: results 2011. Scientific Insititute fo Public Health, Cancer Centre [Internet]. Brussel: Kankercentrum Centre du Cancer;2012. [cited 2014 Feb 1]. Available from: https://workspaces.wiv-isp.be/e-cancer/PublicFiles/presentations/Belgian_Cancer_Center/Quantitative_evaluation_Cancer_Plan_Results%202011_EN.pdf.

11. Burls A, Austin D, Moore D. Commissioning for rare diseases: view from the frontline. BMJ. 2005; 331:1019–21.

12. Simoens S, Cassiman D, Dooms M, Picavet E. Orphan drugs for rare diseases: is it time to revisit their special market access status? Drugs. 2012; 72:1437–43.

13. Picavet E, Cassiman D, Simoens S. Evaluating and improving orphan drug regulations in Europe: a Delphi policy study. Health Policy. 2012; 108:1–9.

14. Abelson R, Pollack A. Medicare widens drugs it accepts for cancer [Internet]. New York: The New York Times;2009. [cited 2014 Feb 1]. Available from: http://www.nytimes.com/2009/01/27/health/27cancer.html?pagewanted=all&_r=0.

15. Dockser Marcus A. To make progress in rare cancers, patients must lead the way. J Clin Oncol. 2009; 27:2575–7.

16. Commission of the European Communities. Communication from the commission to the European parliament, the council, the European economic and social committee and the committee of the regions. On rare diseases: Europe's challenges [Internet]. Brussels: Commission of the European Communities;2008. [cited 2014 Feb 1]. Available from: http://ec.europa.eu/health/ph_threats/non_com/docs/rare_com_en.pdf.

17. Denis A, Mergaert L, Fostier C, Cleemput I, Simoens S. A comparative study of European rare disease and orphan drug markets. Health Policy. 2010; 97:173–9.

18. EURODIS. The voice of rare disease patients: experiences and expectations of over 3,000 patients on rare disease patient registries in Europe [Internet]. Brussels: EURODIS;2013. [cited 2014 Feb 1]. Available from: http://www.eurordis.org/.

19. HemOnc Today. Lack of guidelines, clinical data complicate treatment of rare cancers [Internet]. HemOnc Today;2012. [cited 2014 Feb 1]. Available from: http://www.healio.com/hematology-oncology/news/print/hematology-oncology/%7B1b579909-7aef-4f86-bd98-d8c3ea6d9d3e%7D/lackof-guidelines-clinical-data-complicate-treatment-of-rarecancers.

20. Jung KW, Won YJ, Kong HJ, Oh CM, Seo HG, Lee JS. Cancer statistics in Korea: incidence, mortality, survival and prevalence in 2010. Cancer Res Treat. 2013; 45:1–14.

21. The Council of the European Union. Council recommendation of 8 June 2009 on an action in the field of rare disease (2009/C 151/02) [Internet]. Viganello-Lugano: The Council of the European Union;2009. [cited 2014 Feb 1]. Available from: http://eur-lex.europa.eu/LexUriServ/LexUriServ.do?uri=OJ:C:2009:151:0007:0010:EN:PDF.

22. Blue Cross Blue Shiled Association. Blue Distinction Centers for Complex and Rare Cancers (Focusing on complex inpatient and surgical care) [Internet]. Blue Cross Blue Shiled Association;2014. [cited 2014 Feb 1]. Available from: http://www.arkansasbluecross.com/providers/BDCancers.aspx.

23. Derrow P. When you’re diagnosed with a rare cancer [Internet]. New York: NBC News;2010. [cited 2014 Feb 1]. Available from: http://www.nbcnews.com/id/36182363/ns/healthcancer/t/when-youre-diagnosed-rare-cancer/.

24. Peppercorn J, Burstein H, Miller FG, Winer E, Joffe S. Self-reported practices and attitudes of US oncologists regarding off-protocol therapy. J Clin Oncol. 2008; 26:5994–6000.

25. American Society of Clinical Oncology. Reimbursement for cancer treatment: coverage of off-label drug indications. J Clin Oncol. 2006; 24:3206–8.