Protocol and registration
A systematic review on the economic evaluations of healthcare interventions on OD in patients who suffered a stroke will be performed. This systematic review will follow the recommendations stated by the Preferred Reporting Items for Systematic Reviews and Meta-analysis.13 This protocol for a systematic review has been designed following the recommendations stated by PRISMA protocols annex (PRISMA-P).14 The protocol for this systematic review was registered on the international prospective register of systematic reviews of the Center for Reviews and Dissemination (PROSPERO) (registration number: CRD42020136245).15 The main outcome of interest will be the costs and the associated health benefits of available sanitary/healthcare interventions on the appropriate management of post-stroke OD.
Literature search
We will search MEDLINE using Pubmed, Embase using Ovid, the National Health Service Economic Evaluation Database using the Center for Reviews and Dissemination Database of the University of York and the Cost-Effectiveness Analysis Registry database of the Center for the Evaluation of Value and Risk in Health. These Databases will be searched up to 31th December 2020. A publication date restriction will not be imposed. English and Spanish literature will be included. This systematic review will not include posters, abstracts, book chapters or unpublished literature. Search strategy (combined MeSH and search terms used) applied at Pubmed is described in Table 1. A similar strategy will be used in the other databases.
Table 1
Search terms and MeSH terms used in the bibliographic search
Terms related to Oropharyngeal Dysphagia and connected among themselves by “OR”
|
Terms related to Stroke and connected among themselves by “OR”
|
Terms related to Economic Evaluations and connected among themselves by “OR”
|
1. ”Deglutition”[Mesh]
2. “Deglutition Disorders"[Mesh]
3. "Oropharynx/abnormalities"[Mesh]
4."Oropharynx/diagnosis"[Mesh] 5."Oropharynx/diagnostic imaging"[Mesh] 6."Oropharynx/pathology"[Mesh] 7."Oropharynx/pharmacology"[Mesh] 8."Oropharynx/physiopathology"[Mesh]
9."Oropharynx/therapy"[Mesh]
10.Enteral tube feed*/
11.Swallow[ti/abs]
12.Dysphag*[ti/abs]
13.Deglut*[ti/abs]
14.Dysphagia[tw]
15.Dysphag*/
16.Dysphagia therapy/
|
17.”Stroke”[Mesh]
18."Stroke Rehabilitation" [Mesh]
19. "Brain Ischemia/ complications"[Mesh]
20. “Cerebral Infarction"[Mesh]
21.” Cerebral Hemorrhage"[Mesh]
22.”Intracranial Embolism and Thrombosis”[Mesh]
23. “Intracranial Hemorrhages”[Mesh]
24.”Intracranial Arteriosclerosis”[Mesh]
25.”Cerebrovascular Disorders”[Mesh]
26.Stroke[ti/abs]
27.Post stroke[ti/abs]
28.Poststroke[ti/abs]
29.Post-stroke[ti/abs]
30.Cerebral Ischaemia[ti/abs]
31.Brain Ischaemia[ti/abs]
32.Brain infarct[ti/abs]
33.Intracranial hemorrhage[ti/abs]
34.Intracranial haemorrhage[ti/abs]
35.Cerebral Hemorrhage[ti/abs]
36.Cerebral Haemorrhage[ti/abs]
37.Brain Hemorrhage[ti/abs]
38.Brain Haemorrhage[ti/abs]
39.Stroke discharge/
40.Post-stroke/
|
41.”Economics”[Mesh]
42."Economics" [Subheading]
43."Models, Economic"[Mesh]
44. Health Resources"[Mesh]
45. “Tertiary Care Centers/economics"[Mesh]
46. "Rehabilitation Centers/economics"[Mesh]
47."Length of Stay/ economics"[Mesh]
48. "Medicare/economics" [Mesh]
49. “Physical Therapy Modalities/economics"[Mesh]
50. “Emergency Medical Services/economics" [Mesh]
51. "Food, Formulated/ economics"[Mesh]
52. “Cerebrovascular Disorders/ economics"[Mesh]
53.Cost effectiveness analysis
54.Cost utility analysis
55.Cost minimization analysis
56.Cost benefit analysis
57.Cost[tw]
58.Costs[tw]
59.Quality-adjusted life years/
60.Cost utility[ti/abs]
61.Cost-utility[ti/abs]
62.Cost benefit[ti/abs]
63. Cost-benefit[ti/abs]
64.Cost minimization[ti/abs]
65-Cost-minimization[ti/abs]
66.Cost effectiveness[ti/abs]
67.Cost-effectiveness[ti/abs]
|
Terms, detailed in the three columns above, related to Oropharyngeal Dysphagia, Stroke and Health Economics will be connected using “AND”. |
Selection Process Including Eligibility Criteria
Studies will be identified through literature search and will be selected using a double-phase process. In the first phase one reviewer will assess the title and abstract of the identified articles. These articles will be excluded if they do not contain at least minimal relevant information about: “stroke” or “cerebral infarction” or “cerebral hemorrhage” or “brain ischemia”, “dysphagia” or “deglutition” or “swallowing assessment” or “swallowing disorders” and, “economic evaluation” or “economics” or “economic models” or “costs” on their abstracts or titles. A second reviewer will check the excluded articles using the same criteria. In the second phase, articles will be selected according to eligibility criteria. We will include articles if they had economic evaluations in which the intervention effect was quantified by effectiveness results or other measures of effect on healthcare (cost minimization studies, cost-utility studies, cost-effectiveness studies, cost-benefit analysis) or studies in which costs saving applying interventions in OD management were assessed (for instance, studies in which potential savings in post-stroke patients to whom interventions were different to the usual management were applied) and provided information on post-stroke adult patients (≥ 18 years) with OD. Studies will be excluded if they had only partial economic evaluation studies (cost of illness studies, cost-description studies, costs analysis or cost-consequence analysis among others), if they referred to esophageal dysphagia, OD caused by causes different from stroke or if there were duplicate publications from the same study (in that case only one will be considered). Two independent reviewers will perform this process independently. Subsequently, the results will be compared and a third reviewer will make a decision for disagreements.
Data Collection
One reviewer will extract all data from included studies in a specific data sheet. Total articles will be divided in three groups and each of the other three reviewers will extract data from one group using the same datasheet. The extracted data will be compared and, in case of disagreement, a third reviewer will participate in order to reach a final agreed decision. We will gather data related to the design, the participants, the quality and the results of each study. All study data will be transferred from the data collection form to a specific datasheet for each study. We will gather this data from each study: a) Study identification: first author, title, journal and year of publication; b) Main study characteristics: aim, type of economic evaluation (cost minimization, cost-utility, cost-effectiveness, cost-benefit, cost savings), intervention/s assessed, eligibility criteria, retrospective or prospective data gathering, data source, time horizon, economic perspective (patient, hospital, payer, healthcare system or societal), country, year, currency, use of a temporary discount rate (yes/no), presence of a sensitivity analysis (yes/no), data source, location/setting; c) Study sample characteristics (if applicable, studies could use an economic model of the disease): sample size, sociodemographic data (age, average and range; gender), stroke type, mean value of National Institutes of Health Stroke Scale (NIHSS) or Canadian Neurologic Scale (if available on admission, hospitalization or discharge); d) Description of the Healthcare intervention assessed; e) Elements of cost considered, all of them (yes/no): direct healthcare costs (hospitalization: emergency room, hospitalization ward, intensive care unit; long-term care: nursing home, social and health center, hospitalization at home; primary care, medication, ambulance, special diets, tube-feeding, percutaneous endoscopic gastrostomy (PEG) insertion, outpatient visits: nutritionist, physical therapist, speech therapist, nurse; complication related costs: pneumonia, malnutrition), direct non-healthcare costs (social services, time, transportation) and, indirect costs (loss of productivity or time off from work, morbidity, mortality and/or impairment); f) Specific data depending on the type of economic evaluation: for cost minimization studies the data related to the equivalence of interventions, for cost-utility studies the data on the quality-adjusted life years (QUALYs) and/or the disability-adjusted life years (DALYs) applied, for cost-effectiveness studies the effect units applied and for cost-benefit studies the measured benefits; and g) Results of studies depending on the type of economic evaluation: for cost minimization studies the economic savings by applying the most efficient intervention, for cost-utility studies the incremental cost-utility ratio (ICUR), for cost-effectiveness studies the incremental cost-effectiveness ratio (ICER) and for cost-benefit studies the cost-benefit ratio. These data will be reported on their original format. If necessary, we will contact study authors of eligible articles to answer questions about unreported information or to clarify possible misunderstandings. Data obtained from study authors that is not available in the original articles will be clearly identified. We will not plan any calculation based on study data nor any assumption resulting from lost or unavailable information. Moreover, any assumption resulting from lost or unavailable information will be reported.
Quality Assessment
A specific tool to assess the internal validity and the reporting key factors of economic evaluation studies will be used. We will apply the Consolidated Health Economic Evaluation Reporting Standards (CHEERS) Statement.16 A set of items that apply to a critical appraisal of economic evaluation studies is provided in this checklist. Each item represents a study aspect that we will rate as “Yes, partly, no or not applicable”. For each study, the total amount of items will be rated as “yes (1 point)” and partly (0.5 points) and then it will be divided between the total applicable items. This total score will be expressed as a percentage; a higher score will represent a lower risk of bias. We will consider a score of 100% as a very low risk of bias study. As we want to assess the current state of the literature on this topic and not to create a final sum of the evidence, we did not exclude from this review any study based on its quality assessment score.
Data Presentation And Data Synthesis
We will use two different strategies to synthesize the information of this systematic review. A narrative method will be used to describe main study characteristics, characteristics of the study sample, cost elements considered and specific data depending on the type of economic evaluation. Identification of studies, results and global score on quality assessment will be presented on a Table. Finally, a meta-narrative synthesis of the extracted information will be performed. In this meta-narrative synthesis we will describe both the assessed evidence on the efficiency/cost effectiveness of different clinical interventions for post-stroke OD together with some of the key aspects of quality assessment evaluation. We will report if studies identified, measured and assessed the complete form of all the important costs for each assessed alternative, if the study structure (study approach, data source) was performed in the most appropriate way to answer the study question and if the most important factors to understand these economic evaluations were properly reported. Finally, the possibility of creating an economic model of the disease will be studied and we will evaluate which pharmacoeconomic studies are still pending to be carried out in order to understand the cost-effectiveness of the adequate management of post-stroke OD.