A methodological exploration of systematic reviews on disease burden_Chinese Journal of Evidence-Based Medicine

Authors：

CUI Lu ^1,2,3 , XU Meng ⁴ , ZHANG Xiaolin ⁵ , LIANG Cui ^1,2,3 , HU Xiaoye ^1,2,3 , WANG Hanbin ^1,2,3 , HUANG Jiayi ^1,2,3 ,  YANG Kehu ^2,3,6 ,  LI Xiuxia ^1,2,3,6

1. Evidence Based Social Science Research Center/Health Technology Assessment Center, School of Public Health, Lanzhou University, Lanzhou 730000, P. R. China;
2. Evidence Based Medicine Center, School of Basic Medical Sciences, Lanzhou University, Lanzhou 730000, P. R. China;
3. Key Laboratory of Evidence Based Medicine and Knowledge Translation of Gansu Province, Lanzhou University, Lanzhou 730000, P. R. China;
4. Department of Neurosurgery, Tangdu Hospital, Fourth Military Medical University, Xi'an 710038, P. R. China;
5. Imperial College London, London SW72AZ, UK;
6. Research Center for Medical Device Regulatory Science, Lanzhou 730000, P. R. China;

Corresponding?author：

YANG Kehu, Email: yangkh-ebm@lzu.edu.cn; LI Xiuxia, Email: lixiuxia@lzu.edu

Keywords：

Disease burden; Cost of illness; Systematic reviews; Meta-analysis; Methodology

DOI：

10.7507/1672-2531.202409064

Video：

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Objective To understand the current status of research methods in disease burden systematic reviews, identify limitations and shortcomings of existing research methods, and provide suggestions to address relevant issues. Methods A computer search of the PubMed database was conducted to collect systematic reviews on disease burden, with search limits set from database inception to December 21, 2023. Two independent researchers utilized Endnote 20 for literature screening and Excel 2019 for data extraction and descriptive analysis. Results A total of 216 articles were included in the review, revealing a year-on-year increase in the number of systematic reviews on disease burden since 2004. The journal PharmacoEconomics published the most articles (n=22), while research on certain infectious diseases and parasitic infections was the most prevalent (n=51). Only 31 articles provided a complete account of the entire systematic review process. The reporting rates for inclusion/exclusion criteria, information retrieval, literature screening, and statistical analysis steps were all 100%. However, the rate of protocol registration was relatively low at 19%. Eighty-eight percent of the articles utilized software such as Excel and Epidata for data extraction, yet only 32% adhered to the reproducibility principles outlined in AMSTAR-2. In terms of quality assessment, 105 articles underwent evaluation, with the Joanna Briggs Institute checklist and Newcastle-Ottawa scale being the most commonly used quality assessment tools for epidemiological studies, while economic studies preferred the Drummond checklist (n=9). Regarding the details of inclusion/exclusion criteria, only 53% of studies reported their study design in detail, and less than one-sixth provided a comprehensive description of the interventions and control measures. Statistical analyses predominantly employed qualitative methods (80%), with quantitative analyses comprising a minority (20%), all of which were conducted using meta-analysis techniques, primarily utilizing R software (n=15). Conclusion The number of systematic reviews on disease burden has shown a yearly increasing trend; however, most studies have failed to comprehensively adhere to the fundamental processes of systematic reviews, significantly limiting their quality. Currently, the primary issues include a lack of protocol registration, incomplete supplementary searches, mismatched quality assessment tools, and insufficiently comprehensive outcome measures. To address these challenges, it is essential to develop a methodological guideline for systematic reviews on disease burden that incorporates these concerns. Such a guideline would standardize researchers' practices and ensure strict adherence to systematic review methodologies, thereby enhancing the scientific rigor of the research and its support for clinical decision-making.

Citation： CUI Lu, XU Meng, ZHANG Xiaolin, LIANG Cui, HU Xiaoye, WANG Hanbin, HUANG Jiayi, YANG Kehu, LI Xiuxia. A methodological exploration of systematic reviews on disease burden. Chinese Journal of Evidence-Based Medicine, 2025, 25(1): 88-94. doi: 10.7507/1672-2531.202409064 Copy

1.	李秀霞, 郭康樂, 商雪, 等. 針刺臨床有效性優勢病癥的系統評價. 中國循證醫學雜志, 2021, 21(6): 703-709.
2.	陳耀龍, 李幼平, 杜亮, 等. 醫學研究中證據分級和推薦強度的演進. 中國循證醫學雜志, 2008, (2): 127-133.
3.	劉成成, 黃慧瑤, 石菊芳, 等. 我國1996~2015年結直腸癌經濟負擔的系統評價. 中國腫瘤, 2017, 26(11): 859-867.
4.	楊駿, 王樂, 石春雷, 等. 我國1996-2015年間肝癌經濟負擔研究系統評價. 中國疾病控制與預防雜志, 2017, 21(8): 835-840, 851.
5.	艾力亞力·艾力, 孫立, 李波霖, 等. 中國包蟲病患者經濟負擔研究的系統評價. 中國循證醫學雜志, 2019, 19(12): 1416-1422.
6.	曾瑜, 楊曉妍, 周海龍, 等. 中國人群結核病疾病負擔的系統評價. 中國循證醫學雜志, 2018, 18(6): 570-579.
7.	張曌慧, 黃磊, 石璐, 等. 中國人群塵肺病疾病負擔的系統評價. 中國循證醫學雜志, 2021, 21(3): 276-283.
8.	World Health Organization. International classification of diseases 11th revision.
9.	高亞, 劉明, 楊珂璐, 等. 系統評價報告規范: PRISMA 2020與PRISMA 2009的對比分析與實例解讀. 中國循證醫學雜志, 2021, 21(5): 606-616.
10.	葛龍, 潘蓓, 潘佳雪, 等. 解讀AMSTAR-2—基于隨機和(或)非隨機對照試驗系統評價的質量評價工具. 中國藥物評價, 2017, 34(5): 334-338.
11.	The Joanna Briggs Institute. The Joanna Briggs Institute’s critical appraisal tools.
12.	Stang A. Critical evaluation of the Newcastle-Ottawa scale for the assessment of the quality of nonrandomized studies in meta-analyses. Eur J Epidemiol, 2010, 25(9): 603-605.
13.	Hoy D, Brooks P, Woolf A, et al. Assessing risk of bias in prevalence studies: modification of an existing tool and evidence of interrater agreement. J Clin Epidemiol, 2012, 65(9): 934-939.
14.	Von Elm E, Altman D G, Egger M, et al. The strengthening the reporting of observational studies in epidemiology (STROBE) statement: guidelines for reporting observational studies. Lancet, 2007, 370(9596): 1453-1457.
15.	劉津池, 劉暢, 華成舸. 隨機對照試驗偏倚風險評價工具RoB2(2019修訂版)解讀. 中國循證醫學雜志, 2021, 21(6): 737-744.
16.	Liberati A, Altman DG, Tetzlaff J, et al. The PRISMA statement for reporting systematic reviews and meta-analyses of studies that evaluate healthcare interventions: explanation and elaboration. BMJ, 2009, 339: b2700.
17.	Shea BJ, Reeves BC, Wells G, et al. AMSTAR 2: a critical appraisal tool for systematic reviews that include randomized or non-randomized studies of healthcare interventions, or both. BMJ, 2017, 358: j4008.
18.	Drummond MF, Jefferson TO. Guidelines for authors and peer reviewers of economic submissions to the BMJ. The BMJ Economic Evaluation Working Party. BMJ, 1996, 313(7052): 275-283.
19.	Husereau D, Drummond M, Petrou S, et al. 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, 2013, 16(2): 231-250.
20.	Evers S, Goossens M, De Vet H, et al. Criteria list for assessment of methodological quality of economic evaluations: consensus on health economic criteria. Int J Technol Assess Health Care, 2005, 21(2): 240-245.
21.	Wolff RF, Moons KGM, Riley RD, et al. PROBAST: a tool to assess the risk of bias and applicability of prediction model studies. Ann Intern Med, 2019, 170(1): 51-58.
22.	Jaime Caro J, Eddy DM, Kan H, et al. Questionnaire to assess relevance and credibility of modeling studies for informing health care decision making: an ISPOR-AMCP-NPC Good Practice Task Force Report. Value Health, 2014, 17(2): 174-182.
23.	Bennett C, Manuel DG. Reporting guidelines for modeling studies. BMC Med Res Methodol, 2012, 12: 168.
24.	Critical Appraisal Skills Programme (2023). CASP checklist: CASP qualitative studies checklist.
25.	Shemilt I, James T, Marcello M. A web-based tool for adjusting costs to a specific target currency and price year. Evid Policy, 2010, 6(1): 51-59.
26.	GBD 2019 Diseases and Injuries Collaborators. Global burden of 369 diseases and injuries in 204 countries and territories, 1990-2019: a systematic analysis for the Global Burden of Disease Study 2019. Lancet, 2020, 396(10258): 1204-1222.
27.	Puljak L, Lund H. Definition, harms, and prevention of redundant systematic reviews. Syst Rev, 2023, 12(1): 63.
28.	Stewart L, Moher D, Shekelle P. Why prospective registration of systematic reviews makes sense. Syst Rev, 2012, 1: 7.
29.	Wijnen B, Van Mastrigt G, Redekop WK, et al. How to prepare a systematic review of economic evaluations for informing evidence-based healthcare decisions: data extraction, risk of bias, and transferability (part 3/3). Expert Rev Pharmacoecon Outcomes Res, 2016, 16(6): 723-732.
30.	Szucs TD, Mantovani LG. The perspective of a pharmacoeconomic study: targeting for audiences. Pharmacol Res, 1997, 35(5): 471-475.
31.	Yin D, Van Oorschot D, Jiang N, et al. A systematic literature review to assess the burden of herpes zoster disease in China. Expert Rev Anti Infect Ther, 2021, 19(2): 165-179.
32.	Moskalewicz A, Oremus M. No clear choice between Newcastle-Ottawa scale and appraisal tool for cross-sectional studies to assess methodological quality in cross-sectional studies of health-related quality of life and breast cancer. J Clin Epidemiol, 2020, 120: 94-103.
33.	Herzog R, álvarez-Pasquin MJ, Díaz C, et al. Are healthcare workers' intentions to vaccinate related to their knowledge, beliefs and attitudes. A systematic review. BMC Public Health, 2013, 13: 154.
34.	Shaheen N, Shaheen A, Ramadan A, et al. Appraising systematic reviews: a comprehensive guide to ensuring validity and reliability. Front Res Metr Anal, 2023, 8: 1268045.
35.	徐婷婷, 方海. 衛生經濟學評價指南介紹. 中國衛生經濟, 2016, 35(5): 4.
36.	Critical Appraisal Skills Programme (2023). CASP checklist: CASP economic evaluation checklist.
37.	Palacios A, Rojas-Roque C, González L, et al. Direct medical costs, productivity loss costs and out-of-pocket expenditures in women with breast cancer in Latin America and the Caribbean: a systematic review. Pharmacoeconomics, 2021, 39(5): 485-502.
38.	Husereau D, Drummond M, Augustovski F, et al. Consolidated health economic evaluation reporting standards 2022 (CHEERS 2022) statement: updated reporting guidance for health economic evaluations. Value Health, 2022, 25(1): 3-9.
39.	米雪, 李佳蓮, 陳敏, 等. 藥物經濟學系統評價的再評價. 中國循證醫學雜志, 2019, 19(2): 212-217.
40.	周文靜, 楊瀟, 李艷飛, 等. 疾病負擔研究證據的現狀分析. 中國循證醫學雜志, 2019, 19(11): 1317-1324.
41.	Kaplan RM, Hays RD. Health-related quality of life measurement in public health. Annu Rev Public Health, 2022, 43: 355-373.
42.	邱雨歡, 符秋燕, 史舒婷, 等. 慢性傷口患者特異性健康相關生活質量量表的系統評價. 護理學進展, 2023, 38(16): 1484-1489.
43.	吳景, 姜玉, 畢京浩, 等. 2002?2019年上海市長寧區肝癌傷殘調整壽命年的趨勢分析. 中國腫瘤, 2023, 32(9): 683-690.
44.	Polinder S, Haagsma J A, Stein C, et al. Systematic review of general burden of disease studies using disability-adjusted life years. Popul Health Metr, 2012, 10(1): 21.

1. 李秀霞, 郭康樂, 商雪, 等. 針刺臨床有效性優勢病癥的系統評價. 中國循證醫學雜志, 2021, 21(6): 703-709.
2. 陳耀龍, 李幼平, 杜亮, 等. 醫學研究中證據分級和推薦強度的演進. 中國循證醫學雜志, 2008, (2): 127-133.
3. 劉成成, 黃慧瑤, 石菊芳, 等. 我國1996~2015年結直腸癌經濟負擔的系統評價. 中國腫瘤, 2017, 26(11): 859-867.
4. 楊駿, 王樂, 石春雷, 等. 我國1996-2015年間肝癌經濟負擔研究系統評價. 中國疾病控制與預防雜志, 2017, 21(8): 835-840, 851.
5. 艾力亞力·艾力, 孫立, 李波霖, 等. 中國包蟲病患者經濟負擔研究的系統評價. 中國循證醫學雜志, 2019, 19(12): 1416-1422.
6. 曾瑜, 楊曉妍, 周海龍, 等. 中國人群結核病疾病負擔的系統評價. 中國循證醫學雜志, 2018, 18(6): 570-579.
7. 張曌慧, 黃磊, 石璐, 等. 中國人群塵肺病疾病負擔的系統評價. 中國循證醫學雜志, 2021, 21(3): 276-283.
8. World Health Organization. International classification of diseases 11th revision.
9. 高亞, 劉明, 楊珂璐, 等. 系統評價報告規范: PRISMA 2020與PRISMA 2009的對比分析與實例解讀. 中國循證醫學雜志, 2021, 21(5): 606-616.
10. 葛龍, 潘蓓, 潘佳雪, 等. 解讀AMSTAR-2—基于隨機和(或)非隨機對照試驗系統評價的質量評價工具. 中國藥物評價, 2017, 34(5): 334-338.
11. The Joanna Briggs Institute. The Joanna Briggs Institute’s critical appraisal tools.
12. Stang A. Critical evaluation of the Newcastle-Ottawa scale for the assessment of the quality of nonrandomized studies in meta-analyses. Eur J Epidemiol, 2010, 25(9): 603-605.
13. Hoy D, Brooks P, Woolf A, et al. Assessing risk of bias in prevalence studies: modification of an existing tool and evidence of interrater agreement. J Clin Epidemiol, 2012, 65(9): 934-939.
14. Von Elm E, Altman D G, Egger M, et al. The strengthening the reporting of observational studies in epidemiology (STROBE) statement: guidelines for reporting observational studies. Lancet, 2007, 370(9596): 1453-1457.
15. 劉津池, 劉暢, 華成舸. 隨機對照試驗偏倚風險評價工具RoB2(2019修訂版)解讀. 中國循證醫學雜志, 2021, 21(6): 737-744.
16. Liberati A, Altman DG, Tetzlaff J, et al. The PRISMA statement for reporting systematic reviews and meta-analyses of studies that evaluate healthcare interventions: explanation and elaboration. BMJ, 2009, 339: b2700.
17. Shea BJ, Reeves BC, Wells G, et al. AMSTAR 2: a critical appraisal tool for systematic reviews that include randomized or non-randomized studies of healthcare interventions, or both. BMJ, 2017, 358: j4008.
18. Drummond MF, Jefferson TO. Guidelines for authors and peer reviewers of economic submissions to the BMJ. The BMJ Economic Evaluation Working Party. BMJ, 1996, 313(7052): 275-283.
19. Husereau D, Drummond M, Petrou S, et al. 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, 2013, 16(2): 231-250.
20. Evers S, Goossens M, De Vet H, et al. Criteria list for assessment of methodological quality of economic evaluations: consensus on health economic criteria. Int J Technol Assess Health Care, 2005, 21(2): 240-245.
21. Wolff RF, Moons KGM, Riley RD, et al. PROBAST: a tool to assess the risk of bias and applicability of prediction model studies. Ann Intern Med, 2019, 170(1): 51-58.
22. Jaime Caro J, Eddy DM, Kan H, et al. Questionnaire to assess relevance and credibility of modeling studies for informing health care decision making: an ISPOR-AMCP-NPC Good Practice Task Force Report. Value Health, 2014, 17(2): 174-182.
23. Bennett C, Manuel DG. Reporting guidelines for modeling studies. BMC Med Res Methodol, 2012, 12: 168.
24. Critical Appraisal Skills Programme (2023). CASP checklist: CASP qualitative studies checklist.
25. Shemilt I, James T, Marcello M. A web-based tool for adjusting costs to a specific target currency and price year. Evid Policy, 2010, 6(1): 51-59.
26. GBD 2019 Diseases and Injuries Collaborators. Global burden of 369 diseases and injuries in 204 countries and territories, 1990-2019: a systematic analysis for the Global Burden of Disease Study 2019. Lancet, 2020, 396(10258): 1204-1222.
27. Puljak L, Lund H. Definition, harms, and prevention of redundant systematic reviews. Syst Rev, 2023, 12(1): 63.
28. Stewart L, Moher D, Shekelle P. Why prospective registration of systematic reviews makes sense. Syst Rev, 2012, 1: 7.
29. Wijnen B, Van Mastrigt G, Redekop WK, et al. How to prepare a systematic review of economic evaluations for informing evidence-based healthcare decisions: data extraction, risk of bias, and transferability (part 3/3). Expert Rev Pharmacoecon Outcomes Res, 2016, 16(6): 723-732.
30. Szucs TD, Mantovani LG. The perspective of a pharmacoeconomic study: targeting for audiences. Pharmacol Res, 1997, 35(5): 471-475.
31. Yin D, Van Oorschot D, Jiang N, et al. A systematic literature review to assess the burden of herpes zoster disease in China. Expert Rev Anti Infect Ther, 2021, 19(2): 165-179.
32. Moskalewicz A, Oremus M. No clear choice between Newcastle-Ottawa scale and appraisal tool for cross-sectional studies to assess methodological quality in cross-sectional studies of health-related quality of life and breast cancer. J Clin Epidemiol, 2020, 120: 94-103.
33. Herzog R, álvarez-Pasquin MJ, Díaz C, et al. Are healthcare workers' intentions to vaccinate related to their knowledge, beliefs and attitudes. A systematic review. BMC Public Health, 2013, 13: 154.
34. Shaheen N, Shaheen A, Ramadan A, et al. Appraising systematic reviews: a comprehensive guide to ensuring validity and reliability. Front Res Metr Anal, 2023, 8: 1268045.
35. 徐婷婷, 方海. 衛生經濟學評價指南介紹. 中國衛生經濟, 2016, 35(5): 4.
36. Critical Appraisal Skills Programme (2023). CASP checklist: CASP economic evaluation checklist.
37. Palacios A, Rojas-Roque C, González L, et al. Direct medical costs, productivity loss costs and out-of-pocket expenditures in women with breast cancer in Latin America and the Caribbean: a systematic review. Pharmacoeconomics, 2021, 39(5): 485-502.
38. Husereau D, Drummond M, Augustovski F, et al. Consolidated health economic evaluation reporting standards 2022 (CHEERS 2022) statement: updated reporting guidance for health economic evaluations. Value Health, 2022, 25(1): 3-9.
39. 米雪, 李佳蓮, 陳敏, 等. 藥物經濟學系統評價的再評價. 中國循證醫學雜志, 2019, 19(2): 212-217.
40. 周文靜, 楊瀟, 李艷飛, 等. 疾病負擔研究證據的現狀分析. 中國循證醫學雜志, 2019, 19(11): 1317-1324.
41. Kaplan RM, Hays RD. Health-related quality of life measurement in public health. Annu Rev Public Health, 2022, 43: 355-373.
42. 邱雨歡, 符秋燕, 史舒婷, 等. 慢性傷口患者特異性健康相關生活質量量表的系統評價. 護理學進展, 2023, 38(16): 1484-1489.
43. 吳景, 姜玉, 畢京浩, 等. 2002?2019年上海市長寧區肝癌傷殘調整壽命年的趨勢分析. 中國腫瘤, 2023, 32(9): 683-690.
44. Polinder S, Haagsma J A, Stein C, et al. Systematic review of general burden of disease studies using disability-adjusted life years. Popul Health Metr, 2012, 10(1): 21.

Chinese Journal of Evidence-Based Medicine

A methodological exploration of systematic reviews on disease burden

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