Bibliometric and visual analysis of haemophilia rehabilitation research_West China Medical Journal

Authors：

FENG Qiyang , LIU Shufen , GAN Yingshan , ZHANG Houqiang , SHI Mingnan , CUI Xingyu ,  CHEN Lixia

Department of Rehabilitation Medicine, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100730, P. R. China;

Corresponding?author：

CHEN Lixia, Email: clx727@126.com

Keywords：

Bibliometrics; hemophilia; rehabilitation

DOI：

10.7507/1002-0179.202509316

Video：

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Objective To analyze the total literature published in the field of hemophilia rehabilitation, explore the main research forces and current status in this field, and provide direction for future research. Methods Articles on hemophilia rehabilitation in the Web of Science Core Collection (as of July 27, 2024) were collected. VOSviewer and CiteSpace were used for analysis and visualization of countries, institutions, authors, and keywords. Results Finally, 499 articles related to hemophilia rehabilitation were included. Prior to 1996, the number of publications was extremely low. From 1996 to 2007, there was a slow growth, and after 2007, it entered a phase of rapid growth. There were a total of 88 core authors. Cuesta-Barriuso R had the highest publication volume. Except for a small number of comprehensive journals, most of the papers published belonged to the fields of hemophilia and hematology, with Haemophilia being the highest publication volume. The United Kingdom had a strong level of cooperation with other countries, with Spain, the United States, and the United Kingdom being the main contributing countries. The most common keywords were “haemophilia” and “children”. In the early days, the keyword “muscle strength” received the most sustained attention. In recent years, research had focused on keywords such as “pain” “adult” and “guideline”. Conclusions Since 2007, hemophilia rehabilitation has developed rapidly and has now formed a relatively stable group of author collaborations. Early research focused more on the complications of hemophilia, while current research focuses more on pain management and patient quality of life. In the future, there may be a greater emphasis on guideline development and standardized rehabilitation treatment.

Citation： FENG Qiyang, LIU Shufen, GAN Yingshan, ZHANG Houqiang, SHI Mingnan, CUI Xingyu, CHEN Lixia. Bibliometric and visual analysis of haemophilia rehabilitation research. West China Medical Journal, 2026, 41(6): 921-926. doi: 10.7507/1002-0179.202509316 Copy

1.	Gualtierotti R, Tafuri F, Arcudi S, et al. Current and emerging approaches for pain management in hemophilic arthropathy. Pain Ther, 2022, 11(1): 1-15.
2.	Song X, Liu W, Xue F, et al. Real-world analysis of haemophilia patients in China: a single centre’s experience. Haemophilia, 2020, 26(4): 584-590.
3.	Stonebraker JS, Bolton-Maggs PH, Soucie JM, et al. A study of variations in the reported haemophilia A prevalence around the world. Haemophilia, 2010, 16(1): 20-32.
4.	Berntorp E, Fischer K, Hart DP, et al. Haemophilia. Nat Rev Dis Primers, 2021, 7(1): 45.
5.	Sch?fer GS, Valderramas S, Gomes AR, et al. Physical exercise, pain and musculoskeletal function in patients with haemophilia: a systematic review. Haemophilia, 2016, 22(3): e119-e129.
6.	von Mackensen S, Czepa D, Herbsleb M, et al. Development and validation of a new questionnaire for the assessment of subjective physical performance in adult patients with haemophilia--the HEP-Test-Q. Haemophilia, 2010, 16(1): 170-178.
7.	Pelletier JR, Findley TW, Gemma SA. Isometric exercise for an individual with hemophilic arthropathy. Phys Ther, 1987, 67(9): 1359-1364.
8.	Santavirta N, Solovieva S, Helkama O, et al. Musculoskeletal pain and functional ability in haemophilia A and B. Physiotherapy and rehabilitation in haemophilia patients. Rheumatol Int, 2001, 21(1): 15-19.
9.	Stephensen D, Rodriguez-Merchan EC. Orthopaedic co-morbidities in the elderly haemophilia population: a review. Haemophilia, 2013, 19(2): 166-173.
10.	Souza JC, Simoes HG, Campbell CS, et al. Haemophilia and exercise. Int J Sports Med, 2012, 33(2): 83-88.
11.	Negrier C, Seuser A, Forsyth A, et al. The benefits of exercise for patients with haemophilia and recommendations for safe and effective physical activity. Haemophilia, 2013, 19(4): 487-498.
12.	Elshennawy S, Zahreldin AA, Mortada H, et al. The efficacy of physical therapy modalities in patients with hemophilia: a systematic review of randomized controlled trials with meta-analysis. Arch Phys Med Rehabil, 2023, 104(3): 475-489.
13.	曾雪鵑. 文獻信息增長模型與網絡信息增長模型. 中國科技資源導刊, 2008, 40(5): 15-18.
14.	Price DJDS. Little science, big science. New York: Columbia University Press, 1963.
15.	van Eck NJ, Waltman L. Software survey: VOSviewer, a computer program for bibliometric mapping. Scientometrics, 2010, 84(2): 523-538.
16.	Chen C. CiteSpace II: detecting and visualizing emerging trends and transient patterns in scientific literature. J Am Soc Inf Sci Technol, 2006, 57(3): 359-377.
17.	Cuesta-Barriuso R, Gómez-Conesa A, López-Pina JA. The effectiveness of manual therapy in addition to passive stretching exercises in the treatment of patients with haemophilic knee arthropathy: a randomized, single-blind clinical trial. Haemophilia, 2021, 27(1): e110-e118.
18.	Cuesta-Barriuso R, Mero?o-Gallut J, Donoso-úbeda E, et al. Effect of a fascial therapy treatment on quality of life in patients with hemophilic elbow arthropathy: a randomized controlled trial. Arch Phys Med Rehabil, 2022, 103(5): 867-874.
19.	Cuesta-Barriuso R, Pérez-Llanes R, Donoso-úbeda E, et al. Effects of myofascial release on frequency of joint bleedings, joint status, and joint pain in patients with hemophilic elbow arthropathy: a randomized, single-blind clinical trial. Medicine (Baltimore), 2021, 100(20): e26025.
20.	Cuesta-Barriuso R, Pérez-Llanes R, López-Pina JA, et al. Manual therapy reduces the frequency of clinical hemarthrosis and improves range of motion and perceived disability in patients with hemophilic elbow arthropathy. A randomized, single-blind, clinical trial. Disabil Rehabil, 2022, 44(15): 3938-3945.
21.	Cuesta-Barriuso R, Trelles-Martínez RO. Manual therapy in the treatment of patients with hemophilia B and inhibitor. BMC Musculoskelet Disord, 2018, 19(1): 26.
22.	Calatayud J, Pérez-Alenda S, Carrasco JJ, et al. Safety and effectiveness of progressive moderate-to-vigorous intensity elastic resistance training on physical function and pain in people with hemophilia. Phys Ther, 2020, 100(9): 1632-1644.
23.	Carrasco JJ, Pérez-Alenda S, Casa?a J, et al. Physical activity monitoring and acceptance of a commercial activity tracker in adult patients with haemophilia. Int J Environ Res Public Health, 2019, 16(20): 3851.
24.	Cruz-Montecinos C, Pérez-Alenda S, Casa?a J, et al. Effectiveness of progressive moderate-vigorous intensity elastic resistance training on quality of life and perceived functional abilities in people with hemophilia: Secondary analysis of a randomized controlled trial. Eur J Haematol, 2023, 110(3): 253-261.
25.	Moreno-Segura N, Pérez-Alenda S, García-Dasí M, et al. Effectiveness of therapeutic exercise and cognitive-behavioural therapy combined protocol on functionality, pain and joint health in people with haemophilia: Secondary analysis of a controlled trial. Haemophilia, 2023, 29(2): 629-639.
26.	Pérez-Alenda S, Carrasco JJ, Aguilar-Rodríguez M, et al. Balance evaluation in haemophilic preadolescent patients using Nintendo Wii Balance Board^?. Haemophilia, 2017, 23(1): e18-e24.
27.	Pérez-Alenda S, Carrasco JJ, Querol-Fuentes F, et al. Benefits of physical activity self-monitoring in patients with haemophilia: a prospective study with one-year follow-up. Haemophilia, 2024, 30(3): 791-799.
28.	García-Dasí M, Pérez-Alenda S, Carrasco JJ, et al. Effects of a non-pharmacological approach for chronic pain management in patients with haemophilia: efficacy of cognitive-behavioural therapy associated with physiotherapy. Haemophilia, 2021, 27(3): e357-e367.
29.	Calatayud J, Ogrezeanu DC, Carrasco JJ, et al. Safety, feasibility, and neuromuscular activity of acute low-load resistance exercise with or without blood flow restriction in patients with severe hemophilia. Eur J Haematol, 2023, 111(1): 47-56.
30.	Franchini M. The modern treatment of haemophilia: a narrative review. Blood Transfus, 2013, 11(2): 178-182.
31.	Marchesini E, Morfini M, Valentino L. Recent advances in the treatment of hemophilia: a review. Biologics, 2021, 15: 221-235.
32.	Rodriguez-Merchan EC. Surgery in haemophilic patients with inhibitors. Haemophilia, 2004, 10(Suppl 2): 1-2.
33.	Lopes TJ, Rios R, Nogueira T. Computational analyses reveal fundamental properties of the hemophilia literature in the last 6 decades. Bioinform Biol Insights, 2022, 16: 11779322221125604.
34.	Matlary RED, Grydeland M, Glosli H, et al. Factors associated with physical activity in young people with haemophilia A on prophylaxis. Haemophilia, 2023, 29(3): 900-909.
35.	Wagner B, Krüger S, Hilberg T, et al. The effect of resistance exercise on strength and safety outcome for people with haemophilia: a systematic review. Haemophilia, 2020, 26(2): 200-215.
36.	Kumar R, Dunn AL, Schneiderman JE, et al. Moderate-intensity aerobic exercise vs desmopressin in adolescent males with mild hemophilia A: a randomized trial. Blood, 2022, 140(10): 1156-1166.
37.	Fares HM, Ahmed SH, Farhat ES, et al. The efficacy of aerobic training on the pulmonary functions of hemophilic A patients: a randomized controlled trial. Eur Rev Med Pharmacol Sci, 2022, 26(11): 3950-3957.
38.	Gualtierotti R, Solimeno LP, Peyvandi F. Hemophilic arthropathy: current knowledge and future perspectives. J Thromb Haemost, 2021, 19(9): 2112-2121.
39.	McLaughlin P, Chowdary P, Harrison C, et al. Gap analysis investigating healthcare professionals’ skills and knowledge for pain management in haemophilia-a qualitative study. Haemophilia, 2025, 31(5): 988-996.
40.	Kempton CL, Buckner TW, Fridman M, et al. Factors associated with pain severity, pain interference, and perception of functional abilities independent of joint status in US adults with hemophilia: multivariable analysis of the Pain, Functional Impairment, and Quality of Life (P-FiQ) study. Eur J Haematol, 2018, 100(Suppl 1): 25-33.
41.	中華醫學會血液學分會血栓與止血學組, 中國血友病協作組. 血友病治療中國指南(2025 年版). 中華血液學雜志, 2025, 46(8): 681-690.
42.	中國血友病協作組, 中國罕見病聯盟血友病學組. 中國血友病骨骼肌肉并發癥康復評估與治療專家共識. 罕見病研究, 2022, 1(4): 420-427.
43.	Blokzijl J, Pisters MF, Aspdahl M, et al. A clinical practice guideline for primary care physiotherapy in patients with haemophilia. Haemophilia, 2024, 30(5): 1115-1129.
44.	中華醫學會骨科學分會關節外科學組, 中國血友病協作組, 童培建, 等. 中國血友病性骨關節病髖膝關節置換圍手術期管理指南. 中華骨與關節外科雜志, 2022, 15(7): 481-490.
45.	中國血友病協作組, 陳麗霞, 楊仁池. 藥物代謝動力學指導下的血友病康復個體化管理——基于改良 Delphi 法制訂的中國專家共識. 罕見病研究, 2025, 4(1): 96-105.

1. Gualtierotti R, Tafuri F, Arcudi S, et al. Current and emerging approaches for pain management in hemophilic arthropathy. Pain Ther, 2022, 11(1): 1-15.
2. Song X, Liu W, Xue F, et al. Real-world analysis of haemophilia patients in China: a single centre’s experience. Haemophilia, 2020, 26(4): 584-590.
3. Stonebraker JS, Bolton-Maggs PH, Soucie JM, et al. A study of variations in the reported haemophilia A prevalence around the world. Haemophilia, 2010, 16(1): 20-32.
4. Berntorp E, Fischer K, Hart DP, et al. Haemophilia. Nat Rev Dis Primers, 2021, 7(1): 45.
5. Sch?fer GS, Valderramas S, Gomes AR, et al. Physical exercise, pain and musculoskeletal function in patients with haemophilia: a systematic review. Haemophilia, 2016, 22(3): e119-e129.
6. von Mackensen S, Czepa D, Herbsleb M, et al. Development and validation of a new questionnaire for the assessment of subjective physical performance in adult patients with haemophilia--the HEP-Test-Q. Haemophilia, 2010, 16(1): 170-178.
7. Pelletier JR, Findley TW, Gemma SA. Isometric exercise for an individual with hemophilic arthropathy. Phys Ther, 1987, 67(9): 1359-1364.
8. Santavirta N, Solovieva S, Helkama O, et al. Musculoskeletal pain and functional ability in haemophilia A and B. Physiotherapy and rehabilitation in haemophilia patients. Rheumatol Int, 2001, 21(1): 15-19.
9. Stephensen D, Rodriguez-Merchan EC. Orthopaedic co-morbidities in the elderly haemophilia population: a review. Haemophilia, 2013, 19(2): 166-173.
10. Souza JC, Simoes HG, Campbell CS, et al. Haemophilia and exercise. Int J Sports Med, 2012, 33(2): 83-88.
11. Negrier C, Seuser A, Forsyth A, et al. The benefits of exercise for patients with haemophilia and recommendations for safe and effective physical activity. Haemophilia, 2013, 19(4): 487-498.
12. Elshennawy S, Zahreldin AA, Mortada H, et al. The efficacy of physical therapy modalities in patients with hemophilia: a systematic review of randomized controlled trials with meta-analysis. Arch Phys Med Rehabil, 2023, 104(3): 475-489.
13. 曾雪鵑. 文獻信息增長模型與網絡信息增長模型. 中國科技資源導刊, 2008, 40(5): 15-18.
14. Price DJDS. Little science, big science. New York: Columbia University Press, 1963.
15. van Eck NJ, Waltman L. Software survey: VOSviewer, a computer program for bibliometric mapping. Scientometrics, 2010, 84(2): 523-538.
16. Chen C. CiteSpace II: detecting and visualizing emerging trends and transient patterns in scientific literature. J Am Soc Inf Sci Technol, 2006, 57(3): 359-377.
17. Cuesta-Barriuso R, Gómez-Conesa A, López-Pina JA. The effectiveness of manual therapy in addition to passive stretching exercises in the treatment of patients with haemophilic knee arthropathy: a randomized, single-blind clinical trial. Haemophilia, 2021, 27(1): e110-e118.
18. Cuesta-Barriuso R, Mero?o-Gallut J, Donoso-úbeda E, et al. Effect of a fascial therapy treatment on quality of life in patients with hemophilic elbow arthropathy: a randomized controlled trial. Arch Phys Med Rehabil, 2022, 103(5): 867-874.
19. Cuesta-Barriuso R, Pérez-Llanes R, Donoso-úbeda E, et al. Effects of myofascial release on frequency of joint bleedings, joint status, and joint pain in patients with hemophilic elbow arthropathy: a randomized, single-blind clinical trial. Medicine (Baltimore), 2021, 100(20): e26025.
20. Cuesta-Barriuso R, Pérez-Llanes R, López-Pina JA, et al. Manual therapy reduces the frequency of clinical hemarthrosis and improves range of motion and perceived disability in patients with hemophilic elbow arthropathy. A randomized, single-blind, clinical trial. Disabil Rehabil, 2022, 44(15): 3938-3945.
21. Cuesta-Barriuso R, Trelles-Martínez RO. Manual therapy in the treatment of patients with hemophilia B and inhibitor. BMC Musculoskelet Disord, 2018, 19(1): 26.
22. Calatayud J, Pérez-Alenda S, Carrasco JJ, et al. Safety and effectiveness of progressive moderate-to-vigorous intensity elastic resistance training on physical function and pain in people with hemophilia. Phys Ther, 2020, 100(9): 1632-1644.
23. Carrasco JJ, Pérez-Alenda S, Casa?a J, et al. Physical activity monitoring and acceptance of a commercial activity tracker in adult patients with haemophilia. Int J Environ Res Public Health, 2019, 16(20): 3851.
24. Cruz-Montecinos C, Pérez-Alenda S, Casa?a J, et al. Effectiveness of progressive moderate-vigorous intensity elastic resistance training on quality of life and perceived functional abilities in people with hemophilia: Secondary analysis of a randomized controlled trial. Eur J Haematol, 2023, 110(3): 253-261.
25. Moreno-Segura N, Pérez-Alenda S, García-Dasí M, et al. Effectiveness of therapeutic exercise and cognitive-behavioural therapy combined protocol on functionality, pain and joint health in people with haemophilia: Secondary analysis of a controlled trial. Haemophilia, 2023, 29(2): 629-639.
26. Pérez-Alenda S, Carrasco JJ, Aguilar-Rodríguez M, et al. Balance evaluation in haemophilic preadolescent patients using Nintendo Wii Balance Board^?. Haemophilia, 2017, 23(1): e18-e24.
27. Pérez-Alenda S, Carrasco JJ, Querol-Fuentes F, et al. Benefits of physical activity self-monitoring in patients with haemophilia: a prospective study with one-year follow-up. Haemophilia, 2024, 30(3): 791-799.
28. García-Dasí M, Pérez-Alenda S, Carrasco JJ, et al. Effects of a non-pharmacological approach for chronic pain management in patients with haemophilia: efficacy of cognitive-behavioural therapy associated with physiotherapy. Haemophilia, 2021, 27(3): e357-e367.
29. Calatayud J, Ogrezeanu DC, Carrasco JJ, et al. Safety, feasibility, and neuromuscular activity of acute low-load resistance exercise with or without blood flow restriction in patients with severe hemophilia. Eur J Haematol, 2023, 111(1): 47-56.
30. Franchini M. The modern treatment of haemophilia: a narrative review. Blood Transfus, 2013, 11(2): 178-182.
31. Marchesini E, Morfini M, Valentino L. Recent advances in the treatment of hemophilia: a review. Biologics, 2021, 15: 221-235.
32. Rodriguez-Merchan EC. Surgery in haemophilic patients with inhibitors. Haemophilia, 2004, 10(Suppl 2): 1-2.
33. Lopes TJ, Rios R, Nogueira T. Computational analyses reveal fundamental properties of the hemophilia literature in the last 6 decades. Bioinform Biol Insights, 2022, 16: 11779322221125604.
34. Matlary RED, Grydeland M, Glosli H, et al. Factors associated with physical activity in young people with haemophilia A on prophylaxis. Haemophilia, 2023, 29(3): 900-909.
35. Wagner B, Krüger S, Hilberg T, et al. The effect of resistance exercise on strength and safety outcome for people with haemophilia: a systematic review. Haemophilia, 2020, 26(2): 200-215.
36. Kumar R, Dunn AL, Schneiderman JE, et al. Moderate-intensity aerobic exercise vs desmopressin in adolescent males with mild hemophilia A: a randomized trial. Blood, 2022, 140(10): 1156-1166.
37. Fares HM, Ahmed SH, Farhat ES, et al. The efficacy of aerobic training on the pulmonary functions of hemophilic A patients: a randomized controlled trial. Eur Rev Med Pharmacol Sci, 2022, 26(11): 3950-3957.
38. Gualtierotti R, Solimeno LP, Peyvandi F. Hemophilic arthropathy: current knowledge and future perspectives. J Thromb Haemost, 2021, 19(9): 2112-2121.
39. McLaughlin P, Chowdary P, Harrison C, et al. Gap analysis investigating healthcare professionals’ skills and knowledge for pain management in haemophilia-a qualitative study. Haemophilia, 2025, 31(5): 988-996.
40. Kempton CL, Buckner TW, Fridman M, et al. Factors associated with pain severity, pain interference, and perception of functional abilities independent of joint status in US adults with hemophilia: multivariable analysis of the Pain, Functional Impairment, and Quality of Life (P-FiQ) study. Eur J Haematol, 2018, 100(Suppl 1): 25-33.
41. 中華醫學會血液學分會血栓與止血學組, 中國血友病協作組. 血友病治療中國指南(2025 年版). 中華血液學雜志, 2025, 46(8): 681-690.
42. 中國血友病協作組, 中國罕見病聯盟血友病學組. 中國血友病骨骼肌肉并發癥康復評估與治療專家共識. 罕見病研究, 2022, 1(4): 420-427.
43. Blokzijl J, Pisters MF, Aspdahl M, et al. A clinical practice guideline for primary care physiotherapy in patients with haemophilia. Haemophilia, 2024, 30(5): 1115-1129.
44. 中華醫學會骨科學分會關節外科學組, 中國血友病協作組, 童培建, 等. 中國血友病性骨關節病髖膝關節置換圍手術期管理指南. 中華骨與關節外科雜志, 2022, 15(7): 481-490.
45. 中國血友病協作組, 陳麗霞, 楊仁池. 藥物代謝動力學指導下的血友病康復個體化管理——基于改良 Delphi 法制訂的中國專家共識. 罕見病研究, 2025, 4(1): 96-105.

West China Medical Journal

Bibliometric and visual analysis of haemophilia rehabilitation research

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