Abstract
Background
The economics of dengue is complex and multifaceted.
Objectives
We performed a systematic review of the literature to provide a critical overview of the issues related to dengue economics research and to form a background with which to address the question of cost.
Methods
Three literature databases were searched [PubMed, Embase and Latin American and Caribbean Health Sciences Literature (LILACS)], covering a period from 1980 to 2013, to identify papers meeting preset inclusion criteria. Studies were reviewed for methodological quality on the basis of a quality checklist developed for this purpose. An expert survey was designed to identify priority areas in dengue economics research and to identify gaps between the methodology and actual practice. Survey responses were combined with the literature review findings to determine stakeholder priorities in dengue economics research.
Results
The review identified over 700 papers. Forty-two of these papers met the selection criteria. The studies that were reviewed presented results from 32 dengue-endemic countries, underscoring the importance of dengue as a global public health problem. Cost analyses were the most common, with 21 papers, followed by nine cost-effectiveness analyses and seven cost-of-illness studies, indicating a relatively strong mix of methodologies. Dengue annual overall costs (in 2010 values) ranged from US$13.5 million (in Nicaragua) to $56 million (in Malaysia), showing cost variations across countries. Little consistency exists in the way costs were estimated and dengue interventions evaluated, making generalizations around costs difficult.
Conclusions
The current evidence suggests that dengue costs are substantial because of the cost of hospital care and lost earnings. Further research in this area will broaden our understanding of the true economic impact of dengue.
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Authors’ contributions
DC is the overall guarantor for the study. DC guided the design of this review and the survey. CG and DC developed the search methodology and created the data abstraction sheets and the survey. CG and NL conducted the searches and abstracted the data, removed the duplicates, and screened and selected the studies, with the support of DC. DC drafted the article and created the tables and figures with support from CG and NL.
Acknowledgments
JHU’s IVAC received a sponsored grant from the Pan-American Health and Education Foundation (PAHEF), which was primarily funded by Sanofi Pasteur. JHU’s IVAC is a consortium member of the Dengue Vaccine Initiative (DVI). The views expressed do not necessarily represent those of the other DVI consortium members. We thank the following experts, who formed part of the Costing Dengue Working Group, for their contributions in completing the survey, attending the workshop and assisting in the development of the costing guidelines. These include B. Armien from Instituto Conmemorativo Gorgas de Estudios de la Salud, Panama; J. Arredondo from the Secretary of Health, Vector Borne Diseases Control Division, Mexico; M. Carabali from the DVI, Colombia Office; G. Carrasquilla from the Centro de Estudios e Investigación en Salud (CEIS), Colombia; R. Castro from the Universidad de los Andes, Colombia; L. Durand from Sanofi Pasteur, France; L. Durán from the Universidad Nacional Autónoma de México (UNAM), Mexico; M.E. García from the Health Services Management, Mexico; V. Gallegos from the National Center for Health Technology Excellence, Ministry of Health, Mexico; M. Gontes from the Instituto Nacional de Salud Pública (INSP), Mexico; J. G. López from Sanofi Pasteur, Latin America; C. McFarlane from the Ministry of Health, Jamaica; R. Montoya from the Pan-American Health Organization (PAHO), El Salvador; A. M. C. Sartori from the Hospital das Clínicas da Faculdade de Medicina da Universidade de São Paulo, Brazil; J. B. Siqueira from the Universidade Federal de Goiás, Brazil; and C. Martelli from the Universidade Federal de Goiás, Brazil and Universidade Federal de Pernambuco, Brazil. We also thank PAHEF for providing communications and administrative oversight of the workshop. We are grateful to various members of the DVI, including Drs. Orin Levine, Brian Maskery, Richard Mahoney and Luiz da Silva, for either attending the workshop as observers or for providing feedback on earlier versions of this manuscript. We are indebted to the Bill and Melinda Gates Foundation for their financial contribution to the DVI. The current paper would not have been possible if the lead author of this paper had not been a member of the DVI receiving funding from Gates.
Conflicts of interest
DC, GC and NL declare no conflicts of interest in conducting this review.
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Appendices
Appendix 1: Example of MeSH Search Terms for PubMed
Search Group 1: Organism, Morbidity and Mortality
(“Dengue” [MeSH] OR “Dengue” [all fields] OR “Classical Dengue Fever” [all fields] OR “Classical Dengue Fevers” [all fields] OR “Dengue Fever” [all fields] OR “Dengue Fevers” [all fields] OR “dengue virus” [all fields] OR “dengue vaccine” [all fields] OR “dengue vaccines” [all fields] OR “dengue virus vaccine” [all fields] OR “dengue virus vaccines” [all fields] OR “dengue fever virus” [all fields] OR “dengue flavivirus” [all fields] OR “Aden fever” [all fields] OR “Aden fevers” [all fields] OR “Dengue Hemorrhagic Fever” [MeSH] OR “Dengue Hemorrhagic Fevers” [all fields] OR “Bangkok hemorrhagic fever” [all fields] OR “Bangkok hemorrhagic fevers” [all fields] OR “bouquet fever” [all fields] OR “bouquet fevers” [all fields] OR “dengue shock syndrome” [all fields] OR “dengue shock syndromes” [all fields] OR “breakbone” [all fields] OR “breakbone fever” [all fields] OR dandy [all fields] OR “dandy fever” [all fields] OR “dandy fevers” [all fields] OR “dengue hemorrhagic fever” [all fields] OR “hemorrhagic fevers” [all fields] OR “dengue shock syndrome” [all fields] OR “Philippine hemorrhagic fever” [all fields] OR “Singapore hemorrhagic fever” [all fields] OR “Singapore hemorrhagic fevers” [all fields] OR “Thai hemorrhagic fever” [all fields] OR “Thai hemorrhagic fevers” [all fields] OR “hemorrhagic dengue” [all fields] OR “hemorrhagic dengues” [all fields ]OR “red fever” [all fields] OR “red fevers” [all fields] OR “solar fever” [all fields] OR “sun fever” [all fields] OR “solar fevers” [all fields] OR “sun fevers” [all fields] OR DENV [all fields] OR DENV-1 [all fields] OR DENV1 [all fields] OR “dengue 1 virus” [all fields] OR “dengue virus type 1” [all fields] OR “DENV-2” [all fields] OR DENV2 [all fields] OR “dengue 2 virus” [all fields] OR “dengue virus type 2” [all fields] OR DENV-3 [all fields] OR DENV3 [all fields] OR “dengue 3 virus” [all fields] OR “dengue virus type 3” [all fields] OR DENV-4 [all fields] OR DENV4 [all fields] OR “dengue 4 virus” [all fields] OR “dengue virus type 4” [all fields]
Search Group 2: Region
(“Latin America” [MeSH] OR “Latin America” [all fields] OR “Central America” [all fields] OR “South America” [all fields] OR “West Indies” [all fields] OR “Argentina” [MeSH] OR Argentina [all fields] OR Aruba [all fields] OR “Belize” [MeSH] OR “Belize” [all fields] OR “British Honduras” [all fields] OR “Bolivia” [MeSH] OR Bolivia [all fields] OR “Brazil” [MeSH] OR Brazil [all fields] OR “Chile” [MeSH] OR Chile [all fields] OR “Colombia” [MeSH] OR Columbia [all fields] OR “Cuba” [MeSH] OR Cuba [all fields] OR “Dominica” [MeSH] OR Dominica [all fields] OR “Dominican Republic” [MeSH] OR “Dominican Republic” [all fields] OR “Ecuador” [MeSH] OR “Galapagos Islands” [all fields] OR Ecuador [all fields] OR “French Guiana” [all fields] OR “Grenada” [MeSH] OR Grenada [all fields] OR “Guyana” [MeSH] OR Guyana [all fields] OR “British Guyana” [all fields] OR “Haiti” [MeSH] OR Haiti [all fields] OR “Jamaica” [MeSH] OR Jamaica [all fields] OR “Netherlands Antilles” [all fields] OR Paraguay [MeSH] OR Paraguay [all fields] OR Peru [MeSH] OR Peru [all fields] OR “Saint Kitts and Nevis” [MeSH] OR “St Kitts and Nevis” [all fields] OR “St. Kitts and Nevis” [all fields] OR “Saint Kitts and Nevis” [all fields] OR Nevis [all fields] OR “Saint Lucia” [MeSH] OR “Saint Lucia” [all fields] OR “St Lucia” [all fields] OR “St. Lucia” [all fields] OR “Saint Vincent and the Grenadines” [MeSH] OR “Saint Vincent and the Grenadines” [all fields] OR “St Vincent and the Grenadines” [all fields] OR “St. Vincent and the Grenadines” [all fields] OR Grenadines [all fields] OR Suriname [MeSH] OR Suriname [all fields] OR “Dutch Guiana” [all fields] OR “Netherlands Guiana” [all fields] OR “Uruguay” [MeSH] OR Uruguay [all fields] OR Venezuela [all fields] OR Venezuela [MeSH] OR Mexico [all fields] OR Caribbean [all fields] OR “Caribbean Region” [all fields] OR “south and Central America” [all fields] OR Belize [all fields] OR “Costa Rica” [MeSH] OR “Costa Rica” [all fields] OR “El Salvador” [MeSH] OR “El Salvador” [all fields] OR “Guatemala” [MeSH] OR Guatemala [all fields] OR “Honduras” [MeSH] OR Honduras [all fields] OR Nicaragua [all fields] OR Nicaragua [MeSH] OR Panama [MeSH] OR Panama [all fields] OR “Sao Tome and Principe” [all fields] OR “Antigua and Barbuda” [MeSH] OR “Antigua and Barbuda” [all fields] OR Barbuda [all fields] OR Antigua [all fields])
Search Group 3: Economics
(costs and cost analysis [MeSH] OR “Cost Measure” [all fields] OR “Cost Measures” [all fields] OR “pricing” [all fields] OR cost of illness [MeSH] OR “cost of illness” [all fields] OR “Illness Cost” [all fields] OR “Illness Costs” [all fields] OR “Cost of Disease” [all fields] OR “Costs of Disease” [all fields] OR “Sickness Cost” [all fields] OR “Costs” [all fields] OR “Disease Costs” [all fields] OR “Disease Cost” [all fields] OR “Burden of Illness” [all fields] OR “Illness Burden” [all fields] OR “Illness Burdens” [all fields] OR “Cost of Sickness” [all fields] OR cost-benefit analysis [MeSH] OR “Cost-Benefit Analysis” [all fields] OR “Cost-Benefit Analyses” [all fields] OR “Cost-Benefit Data” [all fields] OR “Cost Benefit” [all fields] OR “Benefits and Costs” [all fields] OR “Costs and Benefits” [all fields] OR hospital costs [MeSH] OR “hospital costs” [all fields] OR “hospital cost” [all fields] OR cost control [MeSH] OR “cost control” [all fields] OR “cost controls” [all fields] OR “cost containment” [all fields] OR “cost containments” [all fields] OR “cost-effectiveness” [all fields] OR “cost effectiveness” [all fields] OR “cost savings” [all fields] OR cost savings [MeSH] OR “cost savings” [all fields] OR “cost saving” [all fields] OR drug costs [MeSH] OR “drug costs” [all fields] OR “drug cost” [all fields] OR “economic value of life” [all fields])
Other Potential Terms
1. Health Expenditures
2. Health Care Costs [MeSH]
(health care costs [MeSH] OR “health care costs” [all fields] OR “health care cost” [all fields] OR “Healthcare Cost” [all fields] OR “Healthcare Costs” [all fields] OR “Medical Care Costs” [all fields] OR “Medical Care Cost” [all fields] OR “Treatment Cost” OR “Treatment Costs” OR “Medical Care Cost” OR “Medical Care Costs” OR “health cost” [all fields] OR “health costs” [all fields])
3. Managed Care Programmes [MeSH]
(managed care programs [MeSH] OR “managed care programs” [all fields] OR “Managed Health Care Insurance Plans” [all fields] OR “Managed Care” [all fields] OR “Insurance Case Management” [all fields])
Appendix 2. Quality Checklist
Appendix 3. Survey: Developing Guidelines for Estimation of the Cost of Dengue in the Latin America and Caribbean (LAC) region
Limited research has been done to estimate the costs of dengue in the Latin American and Caribbean (LAC) region. Consequently, there is little development of the necessary underlying theory of costs of dengue, and limited data systematically collected for this purpose is available. The research difficulties in this area are compounded by the fact that there is little consensus on which guidelines to adopt to estimate costs associated with dengue.
To make future costs of dengue studies more comparable, it is important to follow standards for estimating costs of dengue. In this survey we would like you to identify areas in a cost of dengue analysis that are considered high priority to cover in the workshop. In other words, areas where there is little development or agreement about its use or applicability to cost analysis. For example, while the study perspective is key in the design of a cost analysis, you may not deem this a priority for the workshop as this is an area that has been well explored in the literature and few disagree of its importance.
Please base your response on your expert opinion and past experience in conducting dengue research and/or dengue economics research in the region. We will compile a list of priority areas based on your survey response, which will serve as the basis for group discussions during the workshop.
1. Name:
2. Country of origin:
3. Area of expertise:
4. E-mail:
5. Have you ever been involved or are you planning to be involved in the design of a cost evaluation of dengue?
a. Yes b. No
If no, please describe areas that you hope to contribute to during the workshop:
6. There are a number of key economic concepts and/or issues that are important in a cost analysis. What concepts and/or issues are high priorities to cover in the workshop? (Please indicate with a check your response on each row)
Key concepts and/or issues in cost analysis | Very high priority | Somewhat of a priority | A low priority | Not a priority at all |
---|---|---|---|---|
Scope of analysis | ||||
Study duration considerations | ||||
Study population | ||||
Study perspective | ||||
Study outcome measures | ||||
Sample size calculation | ||||
Case definition of dengue | ||||
Health care utilization calculation | ||||
Full vs incremental cost analysis | ||||
Financial vs economic costing | ||||
Description of programme costs | ||||
Data collection methods | ||||
Source of information costs | ||||
Retrospective vs prospective studies | ||||
Sensitivity analysis | ||||
Representativeness and/or generalizability of data | ||||
Other (please specify): | ||||
Other (please specify): | ||||
Other (please specify): | ||||
Other (please specify): | ||||
Other (please specify): | ||||
Other (please specify): | ||||
Other (please specify): |
7. When defining the scope of a cost analysis of dengue, what criteria are high priorities to cover in the workshop? (Please indicate with a check your response on each row)
Criteria for defining scope of study | Very high priority | Somewhat of a priority | A low priority | Not a priority at all |
---|---|---|---|---|
Study question | ||||
Study duration | ||||
Study population (or geographical boundary) | ||||
Study perspective | ||||
Other (please specify): | ||||
Other (please specify): | ||||
Other (please specify): |
8. For dengue costing, what measures/indicators are high priorities to cover in the workshop? (Please indicate with a check your response on each row)
Measures/indicators | Very high priority | Somewhat of a priority | A low priority | Not a priority at all |
---|---|---|---|---|
Hospital dengue (severe) case | ||||
Ambulatory dengue (non- severe) case | ||||
Dengue fever | ||||
Dengue infection (including asymptomatic) | ||||
Dengue-related death | ||||
Dengue serology | ||||
Haemorrhagic dengue | ||||
Dengue fever vector-control | ||||
Other (please specify): | ||||
Other (please specify): | ||||
Other (please specify): |
9. Given the clinical and epidemiological differences of dengue between children and adults, what age groups are high priorities when costing dengue? (Please indicate with a check your response on each row)
Target age groups (years) | Very high priority | Somewhat of a priority | A low priority | Not a priority at all |
---|---|---|---|---|
<2 years | ||||
2–4 years | ||||
10–14 years | ||||
15+ years | ||||
Other (please specify): | ||||
Other (please specify): | ||||
Other (please specify): |
10. When costing dengue, what sources of information are high priorities? (Please indicate with a check your response on each row)
Possible sources of information | Very high priority | Somewhat of a priority | A low priority | Not a priority at all |
---|---|---|---|---|
Alongside trial | ||||
Along surveillance study | ||||
Published/grey literature | ||||
National administrative data | ||||
Expert opinion | ||||
Patient interviews | ||||
Hospital data | ||||
Other (please specify): | ||||
Other (please specify): | ||||
Other (please specify): |
11. When costing dengue, what cost components are high priorities? (Please indicate with a check your response on each row)
Cost components | Very high priority | Somewhat of a priority | A low priority | Not a priority at all |
---|---|---|---|---|
Direct medical costs | ||||
Hospital/outpatient care | ||||
Medication | ||||
Diagnostics/procedures | ||||
Other (please specify): | ||||
Other (please specify): | ||||
Direct non-medical costs | ||||
Transportation | ||||
Food | ||||
Lodging | ||||
Other (please specify): | ||||
Other (please specify): | ||||
Indirect costs (patients) | ||||
Lost wages/non-wage income | ||||
Lost time at school | ||||
Other (please specify): | ||||
Other (please specify): | ||||
Indirect costs (caretakers) | ||||
Lost wages/non-wage income | ||||
Lost time at school | ||||
Other (please specify): | ||||
Other (please specify): |
12. For costing a dengue outbreak, what cost components are high priorities? (Please indicate with a check your response on each row)
Cost component | Very high priority | Somewhat of a priority | A low priority | Not a priority at all |
---|---|---|---|---|
Treatment | ||||
Vector control | ||||
Surveillance | ||||
Community mobilization | ||||
Media and promotional activities | ||||
Promotional material | ||||
Laboratory equipment | ||||
Laboratory procedures | ||||
Other (please specify): | ||||
Other (please specify): | ||||
Other (please specify): |
13. For costing dengue, what approaches are high priorities? (Please indicate with a check your response on each row)
Type of approach | Very high priority | Somewhat of a priority | A low priority | Not a priority at all |
---|---|---|---|---|
Micro-costing | ||||
Macro-costing | ||||
Resource valuation | ||||
Govt. price/reimbursement rates | ||||
Other (please specify): | ||||
Other (please specify): | ||||
Other (please specify): |
14. When assessing the economic burden of dengue, what output units are high priorities? (Please indicate with a check your response on each row)
Type of approach | Very high priority | Somewhat of a priority | A low priority | Not a priority at all |
---|---|---|---|---|
Money | ||||
QALYs | ||||
DALYs | ||||
Other (please specify): | ||||
Other (please specify): | ||||
Other (please specify): |
15. Does the government in your country have a standardized method for conducting cost analysis? If so, does the government specify what to include in the analysis? What components of a cost analysis does it specify?
16. What other issues do you think are important when costing dengue?
17. What do you want to get out of participating in this workshop?
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Constenla, D., Garcia, C. & Lefcourt, N. Assessing the Economics of Dengue: Results from a Systematic Review of the Literature and Expert Survey. PharmacoEconomics 33, 1107–1135 (2015). https://doi.org/10.1007/s40273-015-0294-7
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DOI: https://doi.org/10.1007/s40273-015-0294-7