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Published in: BMC Cancer 1/2019

Open Access 01-12-2019 | Research article

Exploring the current status of neuroendocrine tumours: a population-based analysis of epidemiology, management and use of resources

Authors: Josep Darbà, Alicia Marsà

Published in: BMC Cancer | Issue 1/2019

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Abstract

Background

Neuroendocrine tumours (NETs) are rare malignancies characterised by its capacity to synthesise and secrete monoamines, due to its neuroendocrine origin. Its varied locations and symptoms have traditionally been responsible for extended delays in their diagnosis. The interest of this study was to characterise the patient population diagnosed with NETs in Spain and to revise how the disease is managed, together with the hospitalisation costs of these patients.

Methods

The database included records of all patients diagnosed with a NET between 2010 and 2015. Admission records were used to evaluate hospitalisation, disease management data and costs, and single-patient files were used to characterise the population.

Results

Nine Thousand One Hundred Twenty patients were diagnosed with a neuroendocrine tumour between 2010 and 2015, with a 2 fold increase in the diagnosis rate over the study period. 42.25% of the patients were females, while 57.75% were males, and mean diagnosis age was 62.58 years (SD = 14.65). Considering all the registered neuroendocrine neoplasms, 46.86% of the patients had malignant well-differentiated NETs, 32.02% had a malignant poorly differentiated neuroendocrine carcinoma and 42.93% of patients developed metastatic NETs. In addition, 18.59% of patients were diagnosed with benign well-differentiated NETs. The most common tumour sites were the bronchus, lung and other sites, including pancreatic tumours; metastasis was found in the liver and distant lymph nodes. Pancreatic resection was the most common surgical procedure utilised in these patients, summing 19% of total expenses, the injection of an unspecified therapeutic substance (including targeted therapies) was registered in 11.40% of admissions, while chemotherapy was registered in only 6.85% of admissions. The annual healthcare cost of NETs was €15,373,961, corresponding to €9092 per patient.

Conclusions

The implementation of standard diagnosis procedures should be prioritised, with a focus on the pancreas and lung, and taking into account that 42.93% of the patients develop a metastatic tumour. The presence of comorbidities and multimorbidities should be considered in order to develop more efficient disease management protocols.
Literature
1.
go back to reference Oronsky B, Ma PC, Morgensztern D, Carter CA. Nothing but NET: a review of neuroendocrine tumors and carcinomas. Neoplasia. 2017;19(12):991–1002.CrossRef Oronsky B, Ma PC, Morgensztern D, Carter CA. Nothing but NET: a review of neuroendocrine tumors and carcinomas. Neoplasia. 2017;19(12):991–1002.CrossRef
2.
go back to reference Raphael MJ, Chan DL, Law C, Singh S. Principles of diagnosis and management of neuroendocrine tumours. CMAJ. 2017;189(10):E398–404.CrossRef Raphael MJ, Chan DL, Law C, Singh S. Principles of diagnosis and management of neuroendocrine tumours. CMAJ. 2017;189(10):E398–404.CrossRef
3.
go back to reference van der Zwan JM, Trama A, Otter R, Larrañaga N, Tavilla A, Marcos-Gragera R, et al. Rare neuroendocrine tumours: results of the surveillance of rare cancers in Europe project. Eur J Cancer. 2013;49(11):2565–78.CrossRef van der Zwan JM, Trama A, Otter R, Larrañaga N, Tavilla A, Marcos-Gragera R, et al. Rare neuroendocrine tumours: results of the surveillance of rare cancers in Europe project. Eur J Cancer. 2013;49(11):2565–78.CrossRef
4.
go back to reference Taal BG, Visser O. Epidemiology of neuroendocrine tumours. Neuroendocrinology. 2004;80(Suppl 1):3–7.CrossRef Taal BG, Visser O. Epidemiology of neuroendocrine tumours. Neuroendocrinology. 2004;80(Suppl 1):3–7.CrossRef
5.
go back to reference Hallet J, Law CH, Cukier M, Saskin R, Liu N, Singh S. Exploring the rising incidence of neuroendocrine tumors: a population-based analysis of epidemiology, metastatic presentation, and outcomes. Cancer. 2015;121(4):589–97.CrossRef Hallet J, Law CH, Cukier M, Saskin R, Liu N, Singh S. Exploring the rising incidence of neuroendocrine tumors: a population-based analysis of epidemiology, metastatic presentation, and outcomes. Cancer. 2015;121(4):589–97.CrossRef
6.
go back to reference Singh S, Granberg D, Wolin E, Warner R, Sissons M, Kolarova T, et al. Patient-reported burden of a neuroendocrine tumor (NET) diagnosis: results from the first global survey of patients with NETs. J Glob Oncol. 2016;3(1):43–53.CrossRef Singh S, Granberg D, Wolin E, Warner R, Sissons M, Kolarova T, et al. Patient-reported burden of a neuroendocrine tumor (NET) diagnosis: results from the first global survey of patients with NETs. J Glob Oncol. 2016;3(1):43–53.CrossRef
7.
go back to reference Delle Fave G, O'Toole D, Sundin A, Taal B, Ferolla P, Ramage JK, et al. ENETS consensus guidelines update for Gastroduodenal neuroendocrine neoplasms. Neuroendocrinology. 2016;103(2):119–24.CrossRef Delle Fave G, O'Toole D, Sundin A, Taal B, Ferolla P, Ramage JK, et al. ENETS consensus guidelines update for Gastroduodenal neuroendocrine neoplasms. Neuroendocrinology. 2016;103(2):119–24.CrossRef
8.
go back to reference O'Toole D, Kianmanesh R, Caplin M. ENETS 2016 consensus guidelines for the Management of Patients with digestive neuroendocrine tumors: an update. Neuroendocrinology. 2016;103(2):117–8.CrossRef O'Toole D, Kianmanesh R, Caplin M. ENETS 2016 consensus guidelines for the Management of Patients with digestive neuroendocrine tumors: an update. Neuroendocrinology. 2016;103(2):117–8.CrossRef
9.
go back to reference Niederle B, Pape UF, Costa F, Gross D, Kelestimur F, Knigge U, et al. ENETS consensus guidelines update for neuroendocrine neoplasms of the jejunum and ileum. Neuroendocrinology. 2016;103(2):125–38.CrossRef Niederle B, Pape UF, Costa F, Gross D, Kelestimur F, Knigge U, et al. ENETS consensus guidelines update for neuroendocrine neoplasms of the jejunum and ileum. Neuroendocrinology. 2016;103(2):125–38.CrossRef
10.
go back to reference Falconi M, Eriksson B, Kaltsas G, Bartsch DK, Capdevila J, Caplin M, et al. ENETS consensus guidelines update for the Management of Patients with functional pancreatic neuroendocrine tumors and non-functional pancreatic neuroendocrine tumors. Neuroendocrinology. 2016;103(2):153–71.CrossRef Falconi M, Eriksson B, Kaltsas G, Bartsch DK, Capdevila J, Caplin M, et al. ENETS consensus guidelines update for the Management of Patients with functional pancreatic neuroendocrine tumors and non-functional pancreatic neuroendocrine tumors. Neuroendocrinology. 2016;103(2):153–71.CrossRef
11.
go back to reference Pavel M, O'Toole D, Costa F, Capdevila J, Gross D, Kianmanesh R, et al. ENETS consensus guidelines update for the Management of Distant Metastatic Disease of intestinal, pancreatic, bronchial neuroendocrine neoplasms (NEN) and NEN of unknown primary site. Neuroendocrinology. 2016;103(2):172–85.CrossRef Pavel M, O'Toole D, Costa F, Capdevila J, Gross D, Kianmanesh R, et al. ENETS consensus guidelines update for the Management of Distant Metastatic Disease of intestinal, pancreatic, bronchial neuroendocrine neoplasms (NEN) and NEN of unknown primary site. Neuroendocrinology. 2016;103(2):172–85.CrossRef
12.
go back to reference Ramage JK, De Herder WW, Delle Fave G, Ferolla P, Ferone D, Ito T, et al. ENETS consensus guidelines update for colorectal neuroendocrine neoplasms. Neuroendocrinology. 2016;103(2):139–43.CrossRef Ramage JK, De Herder WW, Delle Fave G, Ferolla P, Ferone D, Ito T, et al. ENETS consensus guidelines update for colorectal neuroendocrine neoplasms. Neuroendocrinology. 2016;103(2):139–43.CrossRef
13.
go back to reference Pape UF, Niederle B, Costa F, Gross D, Kelestimur F, Kianmanesh R, et al. ENETS consensus guidelines for neuroendocrine neoplasms of the appendix (excluding goblet cell carcinomas). Neuroendocrinology. 2016;103(2):144–52.CrossRef Pape UF, Niederle B, Costa F, Gross D, Kelestimur F, Kianmanesh R, et al. ENETS consensus guidelines for neuroendocrine neoplasms of the appendix (excluding goblet cell carcinomas). Neuroendocrinology. 2016;103(2):144–52.CrossRef
14.
go back to reference Chau I, Casciano R, Willet J, Wang X, Yao JC. Quality of life, resource utilisation and health economics assessment in advanced neuroendocrine tumours: a systematic review. Eur J Cancer Care (Engl). 2013;22(6):714–25.CrossRef Chau I, Casciano R, Willet J, Wang X, Yao JC. Quality of life, resource utilisation and health economics assessment in advanced neuroendocrine tumours: a systematic review. Eur J Cancer Care (Engl). 2013;22(6):714–25.CrossRef
15.
go back to reference Vernieri C, Pusceddu S, Fucà G, Indelicato P, Centonze G, Castagnoli L, et al. Impact of systemic and tumor lipid metabolism on everolimus efficacy in advanced pancreatic neuroendocrine tumors (pNETs). Int J Cancer. 2019;144(7):1704–12.CrossRef Vernieri C, Pusceddu S, Fucà G, Indelicato P, Centonze G, Castagnoli L, et al. Impact of systemic and tumor lipid metabolism on everolimus efficacy in advanced pancreatic neuroendocrine tumors (pNETs). Int J Cancer. 2019;144(7):1704–12.CrossRef
16.
go back to reference Santos AP, Santos AC, Castro C, Raposo L, Pereira SS, Torres I, et al. Visceral obesity and metabolic syndrome are associated with well-differentiated gastroenteropancreatic neuroendocrine tumors. Cancers (Basel). 2018;10(9):E293.CrossRef Santos AP, Santos AC, Castro C, Raposo L, Pereira SS, Torres I, et al. Visceral obesity and metabolic syndrome are associated with well-differentiated gastroenteropancreatic neuroendocrine tumors. Cancers (Basel). 2018;10(9):E293.CrossRef
17.
go back to reference Jilesen AP, van Eijck CH, Busch OR, van Gulik TM, Gouma DJ, van Dijkum EJ. Postoperative outcomes of Enucleation and standard resections in patients with a pancreatic neuroendocrine tumor. World J Surg. 2016;40(3):715–28.CrossRef Jilesen AP, van Eijck CH, Busch OR, van Gulik TM, Gouma DJ, van Dijkum EJ. Postoperative outcomes of Enucleation and standard resections in patients with a pancreatic neuroendocrine tumor. World J Surg. 2016;40(3):715–28.CrossRef
18.
go back to reference Boyar Cetinkaya R, Vatn M, Aabakken L, Bergestuen DS, Thiis-Evensen E. Survival and prognostic factors in well-differentiated pancreatic neuroendocrine tumors. Scand J Gastroenterol. 2014;49(6):734–41.CrossRef Boyar Cetinkaya R, Vatn M, Aabakken L, Bergestuen DS, Thiis-Evensen E. Survival and prognostic factors in well-differentiated pancreatic neuroendocrine tumors. Scand J Gastroenterol. 2014;49(6):734–41.CrossRef
19.
go back to reference Norton JA, Kivlen M, Li M, Schneider D, Chuter T, Jensen RT. Morbidity and mortality of aggressive resection in patients with advanced neuroendocrine tumors. Arch Surg. 2003;138(8):859–66.CrossRef Norton JA, Kivlen M, Li M, Schneider D, Chuter T, Jensen RT. Morbidity and mortality of aggressive resection in patients with advanced neuroendocrine tumors. Arch Surg. 2003;138(8):859–66.CrossRef
20.
go back to reference Shen C, Dasari A, Gu D, Chu Y. S Zhou, Xu Y, et al. costs of cancer care for elderly patients with neuroendocrine tumors. Pharmacoeconomics. 2018;36(8):1005–13.CrossRef Shen C, Dasari A, Gu D, Chu Y. S Zhou, Xu Y, et al. costs of cancer care for elderly patients with neuroendocrine tumors. Pharmacoeconomics. 2018;36(8):1005–13.CrossRef
21.
go back to reference Hallet J, Law CHL, Cheung M, Mittmann N, Liu N, Fischer HD. Patterns and drivers of costs for neuroendocrine tumor care: a comparative population-based analysis. Ann Surg Oncol. 2017;24(11):3312–23.CrossRef Hallet J, Law CHL, Cheung M, Mittmann N, Liu N, Fischer HD. Patterns and drivers of costs for neuroendocrine tumor care: a comparative population-based analysis. Ann Surg Oncol. 2017;24(11):3312–23.CrossRef
22.
go back to reference Payne RA, Abel GA, Guthrie B, Mercer SW. The effect of physical multimorbidity, mental health conditions and socioeconomic deprivation on unplanned admissions to hospital: a retrospective cohort study. CMAJ. 2013;185(5):E221–8.CrossRef Payne RA, Abel GA, Guthrie B, Mercer SW. The effect of physical multimorbidity, mental health conditions and socioeconomic deprivation on unplanned admissions to hospital: a retrospective cohort study. CMAJ. 2013;185(5):E221–8.CrossRef
23.
go back to reference Tinetti ME, Fried TR, Boyd CM. Designing health care for the most common chronic condition—multimorbidity. JAMA. 2012;307(23):2493–4.CrossRef Tinetti ME, Fried TR, Boyd CM. Designing health care for the most common chronic condition—multimorbidity. JAMA. 2012;307(23):2493–4.CrossRef
Metadata
Title
Exploring the current status of neuroendocrine tumours: a population-based analysis of epidemiology, management and use of resources
Authors
Josep Darbà
Alicia Marsà
Publication date
01-12-2019
Publisher
BioMed Central
Published in
BMC Cancer / Issue 1/2019
Electronic ISSN: 1471-2407
DOI
https://doi.org/10.1186/s12885-019-6412-8

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