Treatment of hemophilia: a review of current advances and ongoing issues - PubMed
Treatment of hemophilia: a review of current advances and ongoing issues
Antonio Coppola et al. J Blood Med. 2010.
Abstract
Replacement of the congenitally deficient factor VIII or IX through plasma-derived or recombinant concentrates is the mainstay of treatment for hemophilia. Concentrate infusions when hemorrhages occur typically in joint and muscles (on-demand treatment) is able to resolve bleeding, but does not prevent the progressive joint deterioration leading to crippling hemophilic arthropathy. Therefore, primary prophylaxis, ie, regular infusion of concentrates started after the first joint bleed and/or before the age of two years, is now recognized as first-line treatment in children with severe hemophilia. Secondary prophylaxis, whenever started, aims to avoid (or delay) the progression of arthropathy and improve patient quality of life. Interestingly, recent data suggest a role for early prophylaxis also in preventing development of inhibitors, the most serious complication of treatment in hemophilia, in which multiple genetic and environmental factors may be involved. Treatment of bleeds in patients with inhibitors requires bypassing agents (activated prothrombin complex concentrates, recombinant factor VIIa). However, eradication of inhibitors by induction of immune tolerance should be the first choice for patients with recent onset inhibitors. The wide availability of safe factor concentrates and programs for comprehensive care has now resulted in highly satisfactory treatment of hemophilia patients in developed countries. Unfortunately, this is not true for more than two-thirds of persons with hemophilia, who live in developing countries.
Keywords: bleeding; clotting factor concentrates; comprehensive care; hemophilia; inhibitors; prophylaxis; treatment.
Figures

Factors contributing to inhibitor development in hemophilic patients. Multiple genetic and environmental factors interact during the first exposures to factor concentrate replacement treatment. Most data have been obtained from studies in hemophilia A patients, who develop inhibitors with higher frequency than those with hemophilia B.

Current treatment strategies for patients with hemophilia who develop inhibitors.
Similar articles
-
Prophylaxis in children with hemophilia: evidence-based achievements, old and new challenges.
Coppola A, Tagliaferri A, Di Capua M, Franchini M. Coppola A, et al. Semin Thromb Hemost. 2012 Feb;38(1):79-94. doi: 10.1055/s-0031-1300954. Epub 2012 Feb 7. Semin Thromb Hemost. 2012. PMID: 22314606 Review.
-
[No authors listed] [No authors listed] CMAJ. 1995 Jul 15;153(2):147-57. CMAJ. 1995. PMID: 7600466 Free PMC article.
-
Stobart K, Iorio A, Wu JK. Stobart K, et al. Cochrane Database Syst Rev. 2006 Apr 19;(2):CD003429. doi: 10.1002/14651858.CD003429.pub3. Cochrane Database Syst Rev. 2006. PMID: 16625581 Updated. Review.
-
Stobart K, Iorio A, Wu JK. Stobart K, et al. Cochrane Database Syst Rev. 2005 Apr 18;(2):CD003429. doi: 10.1002/14651858.CD003429.pub2. Cochrane Database Syst Rev. 2005. PMID: 15846666 Updated. Review.
-
Bypassing agent prophylaxis in people with hemophilia A or B with inhibitors.
Chai-Adisaksopha C, Nevitt SJ, Simpson ML, Janbain M, Konkle BA. Chai-Adisaksopha C, et al. Cochrane Database Syst Rev. 2017 Sep 25;9(9):CD011441. doi: 10.1002/14651858.CD011441.pub2. Cochrane Database Syst Rev. 2017. PMID: 28944952 Free PMC article. Review.
Cited by
-
An Orthogonal Array Optimization of Lipid-like Nanoparticles for mRNA Delivery in Vivo.
Li B, Luo X, Deng B, Wang J, McComb DW, Shi Y, Gaensler KM, Tan X, Dunn AL, Kerlin BA, Dong Y. Li B, et al. Nano Lett. 2015 Dec 9;15(12):8099-107. doi: 10.1021/acs.nanolett.5b03528. Epub 2015 Nov 6. Nano Lett. 2015. PMID: 26529392 Free PMC article.
-
Understanding patient preferences and willingness to pay for hemophilia therapies.
Chaugule SS, Hay JW, Young G. Chaugule SS, et al. Patient Prefer Adherence. 2015 Nov 11;9:1623-30. doi: 10.2147/PPA.S92985. eCollection 2015. Patient Prefer Adherence. 2015. PMID: 26635471 Free PMC article.
-
Ghosh S, Mukherjee S, Dutta C, Chakraborty K, Gayen P, Jan S, Bhattacharyya D, Roy RS. Ghosh S, et al. Sci Rep. 2017 Jul 26;7(1):6509. doi: 10.1038/s41598-017-06360-3. Sci Rep. 2017. PMID: 28747673 Free PMC article.
-
Turoctocog alfa: an evidence-based review of its potential in the treatment of hemophilia A.
Takedani H, Hirose J. Takedani H, et al. Drug Des Devel Ther. 2015 Mar 24;9:1767-72. doi: 10.2147/DDDT.S57967. eCollection 2015. Drug Des Devel Ther. 2015. PMID: 25848213 Free PMC article. Review.
-
Low Dose Prophylaxis in Hemophilia Care.
Sidharthan N, Sudevan R. Sidharthan N, et al. Indian J Hematol Blood Transfus. 2020 Jan;36(1):16-25. doi: 10.1007/s12288-019-01147-0. Epub 2019 Jun 15. Indian J Hematol Blood Transfus. 2020. PMID: 32158085 Free PMC article. Review.
References
-
- Mannucci PM, Tuddenham EG. The hemophilias – from royal genes to gene therapy. N Engl J Med. 2001;344(5):1773–1779. - PubMed
-
- van Dijk K, Fischer K, van der Bom JG, Grobbee DE, van den Berg HM. Variability in clinical phenotype of severe haemophilia: The role of the first joint bleed. Haemophilia. 2005;11(5):438–443. - PubMed
-
- Santagostino E, Mannucci PM, for the Italian Association of Haemophilia Centres (AICE) Guidelines on replacement therapy for haemophilia and inherited coagulation disorders in Italy. Haemophilia. 2000;6(1):1–10. - PubMed
-
- Rosendaal G, Lafeber FP. Pathogenesis of hemophilic arthropathy. Haemophilia. 2006;12(Suppl 3):117–121. - PubMed
-
- Mannucci PM. Back to the future: A recent history of hemophilia treatment. Haemophilia. 2008;14(Suppl 3):10–18. - PubMed
LinkOut - more resources
Full Text Sources
Other Literature Sources