Abstract
Purpose
Acromegaly is a rare disease that results in the enlargement of body extremities and in organomegaly. Treatments include surgery, drugs, and radiotherapy, which are all onerous. Therefore, well-conducted cost-analyses are crucial in the decision-making process.
Methods
A systematic review of cost-effectiveness studies on acromegaly therapies was performed following PRISMA and Cochrane recommendations. The search for records was conducted in PubMed, Scopus, and Web of Science (May 2018). The quality of the included studies was assessed using the Joana Briggs Institute Tool.
Results
From initial 547 records, 16 studies were included in the review. The studies could present more than one economic evaluation, and encompassed cost-effectiveness (n = 13), cost-utility (n = 5), and cost-consequence (n = 1) analyses. All studies were model-based and evaluated only direct medical costs. Eleven records did not mention discounting and only 10 performed sensitivity analyses. The characteristic of the studies, the cost-effectiveness results and the studies’ conclusions are described and commented upon. The main limitation of the studies was discussed and aspects to improve in future studies were pointed out.
Conclusions
Cost-effectiveness studies on acromegaly have been performed in several scenarios, evaluating different phases of treatment. However, the studies present limitations and, overall, were considered of moderate quality. Further economic models should be developed following health economics guidelines recommendations, and must improve transparency.
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References
Boguszewski CL: Ayuk J (2016) Management of endocrine disease: acromegaly and cancer: an old debate revisited. Eur J Endocrinol 175:R147–R156
Melmed S (2009) Acromegaly pathogenesis and treatment. J Clin Invest 119:3189–3202
Ayuk J, Sheppard MC (2008) Does acromegaly enhance mortality? Rev Endocr Metab Disord 9:33–39
Katznelson L, Laws ER Jr, Melmed S, Molitch ME, Murad MH, Utz A, Wass JA, Endocrine S (2014) Acromegaly: an endocrine society clinical practice guideline. J Clin Endocrinol Metab 99:3933–3951
Liberati A, Altman DG, Tetzlaff J, Mulrow C, Gotzsche PC, Ioannidis JP, Clarke M, Devereaux PJ, Kleijnen J, Moher D (2009) The PRISMA statement for reporting systematic reviews and meta-analyses of studies that evaluate healthcare interventions: explanation and elaboration. BMJ 339:b2700
Higgins JPT, Green S: Cochrane Handbook for systematic reviews of interventions—version 5.1.0. [updated March 2011]. (2011)
Husereau D, Drummond M, Petrou S, Carswell C, Moher D, Greenberg D, Augustovski F, Briggs AH, Mauskopf J, Loder E, Force IHEEPG-CGRPT (2013) Consolidated Health Economic Evaluation Reporting Standards (CHEERS)—explanation and elaboration: a report of the ISPOR Health Economic Evaluation Publication Guidelines Good Reporting Practices Task Force. Value Health 16:231–250
Gomersall JS, Jadotte YT, Xue Y, Lockwood S, Riddle D, Preda A (2015) Conducting systematic reviews of economic evaluations. Int J Evid Based Healthc 13:170–178
Drummond MF, Sculpher MJ, Torrance GW, O’Brien BJ, Stoddart GL (1997) Methods for the economic evaluation of health care programmes, 2 edn. Oxford Medical Publications, Oxford
Salinas EG, Idrovo J, Rivas R, Zapata L (2008) Economic evaluation of long term somatostatin analogs in the treatment of acromegaly in Mexico. Value Health 11:A227–A227
Valentim J, Passos V, Mataveli F, Calabro A (2008) Cost-effectiveness analysis of somatostatin analogues in the treatment of acromegaly in Brazil. Arquivos brasileiros de endocrinologia e metabologia 52:1452–1460
Biermasz NR, Roelfsema F, Pereira AM, Romijn JA (2009) Cost-effectiveness of lanreotide Autogel in treatment algorithms of acromegaly. Expert Rev Pharmacoecon Outcomes Res 9:223–234
Moore DJ, Adi Y, Connock MJ, Bayliss S (2009) Clinical effectiveness and cost-effectiveness of pegvisomant for the treatment of acromegaly: a systematic review and economic evaluation. BMC Endocr Disord 9:20
Alfonso-Cristancho R, Diazgranados SH, Martinez KM, Diaz-Sotelo OD (2012) Cost-effectiveness of somatostatin analogues for the treatment of acromegaly in Colombia. Open J Endocr Metab Dis 2:102–106
Fujii RK, Mould JF, Fernandes RA, Furlan F, Manfrin DF (2012) Economic evaluation of pegvisomant for active acromegaly patients who failed available therapies in Brazil—public health care system perspective. Value Health 15:A105–A105
Marko NF, LaSota E, Hamrahian AH, Weil RJ (2012) Comparative effectiveness review of treatment options for pituitary microadenomas in acromegaly. J Neurosurg 117:522–538
Marty R, Roze S, Kurth H (2012) Decision-tree model for health economic comparison of two long-acting somatostatin receptor ligand devices in France, Germany, and the UK. Med Dev (Auckland, N.Z.) 5:39–44
Duan L, Huang M, Yan H, Zhang Y, Gu F (2015) Cost-effectiveness analysis of two therapeutic schemes in the treatment of acromegaly: a retrospective study of 168 cases. J Endocrinol Investig 38:717–723
Souza CR, Ferreira CN, Ribeiro AP, Musolino NRC (2014) Economic evaluation of pegvisomanto for the treatment of patients with acromegaly with an inadequate response to a maximal dose of octreotide (alone or combined) in the context of the Unified Health System. J Bras Econ Saúde 6:81–88
Hahl J, Kuiki S, Miettinen T, Snicker R (2015) Cost-effectiveness of pasireotide long-acting in a treatment of acromegaly in Finland. Economic Evaluation Based on Finnish Auria Biobank Data on Health Care Resource Utilization. Value Health 18:A609–A609
Margusino-Framinan L, Pertega-Diaz S, Pena-Bello L, Sangiao-Alvarellos S, Outeirino-Blanco E, Pita-Gutierrez F, Pita-Fernandez S, Cordido F (2015) Cost-effectiveness analysis of preoperative treatment of acromegaly with somatostatin analogue on surgical outcome. Eur J Int Med 26:736–741
Kimmell KT, Weil RJ, Marko NF (2015) Multi-modal management of acromegaly: a value perspective. Pituitary 18:658–665
Carlqvist P, Wilen-Koort A (2016) A cost-effectiveness analysis of pasireotide long-acting compared to continued use of a first-line somatostatin antagonist for the treatment of acromegaly in Sweden. Value Health 19:A591–A591
Xuan J, Zhang Z, Wang Y, Mao Z, Lu YJ, Wang R (2016) Cost-effectiveness analysis of octreotide lar versus lanreotide Sr for the treatment of patients with active acromegaly in China. Value Health 19:A248–A248
Peral C, Cordido F, Gimeno V, Sanchez-Cenizo L, Mir N, Parrondo J (2017) Cost-effectiveness analysis of second-line pharmacological treatments of acromegaly in Spain. Value Health 20:A557–A558
Pita-Gutierrez F, Pertega-Diaz S, Pita-Fernandez S, Pena L, Lugo G, Sangiao-Alvarellos S, Cordido F (2013) Place of preoperative treatment of acromegaly with somatostatin analog on surgical outcome: a systematic review and meta-analysis. PLoS ONE 8:e61523
Brasil, Ministério da Saúde (2002) Protocolo Clínico e Diretrizes Terapêuticas para Acromegalia Portaria no 471, de 24 de junho de 2002
Freda PU, Katznelson L, van der Lely AJ, Reyes CM, Zhao S, Rabinowitz D (2005) Long-acting somatostatin analog therapy of acromegaly: a meta-analysis. J Clin Endocrinol Metab 90:4465–4473
Murray RD, Melmed S (2008) A critical analysis of clinically available somatostatin analog formulations for therapy of acromegaly. J Clin Endocrinol Metab 93:2957–2968
Adelman DT, Burgess A, Davies PR: Evaluation of long-acting somatostatin analog injection devices by nurses: a quantitative study. Med Dev (Auckland, N.Z.) 5:103–109 (2012)
Giustina A, Chanson P, Kleinberg D, Bronstein MD, Clemmons DR, Klibanski A, van der Lely AJ, Strasburger CJ, Lamberts SW, Ho KK, Casanueva FF, Melmed S, G Consensus Acromegaly (2014) Expert consensus document: a consensus on the medical treatment of acromegaly. Nat Rev Endocrinol 10:243–248
Alfonso R, Izquierdo C, Diaz-Sotelo OD, Maestre K, Zarate JP (2010) Cost-effectiveness of somastatin analogues for the treatment of acromegaly in Colombia. Value Health 13:A59–A59
Sandret L, Maison P, Chanson P (2011) Place of cabergoline in acromegaly: a meta-analysis. J Clin Endocrinol Metab 96:1327–1335
Asa SL, Ezzat S (2009) The pathogenesis of pituitary tumors. Annu Rev Pathol 4:97–126
Couldwell WT, Cannon-Albright L (2010) A heritable predisposition to pituitary tumors. Pituitary 13:130–137
Acknowledgements
The authors would like to thank the Brazilian National Counsel of Technological and Scientific Development (CNPq) and the Coordination for the Improvement of Higher Education Personnel (CAPES) for providing scholarships.
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Leonart, L.P., Borba, H.H.L., Ferreira, V.L. et al. Cost-effectiveness of acromegaly treatments: a systematic review. Pituitary 21, 642–652 (2018). https://doi.org/10.1007/s11102-018-0908-0
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DOI: https://doi.org/10.1007/s11102-018-0908-0