Ablation of hemophilic FVIII inhibitors with FVIII priming, cyclophosphamide immune suppression, and rapid tapering of FVIII immune tolerance

American Journal of Hematology

Volumn 76, Issue 2, 2004, Pages 180-184

  Kobrinsky, Nathan L ^a,b   Sjolander, Diane E ^a   Moser, Deb K ^a   Stegman, Dottie A ^a  

^a MeritCare Roger Maris Cancer Center   (United States)

^b UNIVERSITY OF NORTH DAKOTA SCHOOL OF MEDICINE AND HEALTH SCIENCES   (United States)

Author keywords

Cyclophosphamide; Hemophilic inhibitor; Human FVIII; Immune tolerance

Indexed keywords

BLOOD CLOTTING FACTOR 8; BLOOD CLOTTING FACTOR 8 INHIBITOR; CYCLOPHOSPHAMIDE; IMMUNOGLOBULIN;

ADULT; ALOPECIA; ARTICLE; CLINICAL ARTICLE; DRUG DOSE REDUCTION; DRUG EFFECT; DRUG EFFICACY; DRUG TOLERANCE; DRUG TOXICITY; HEMOPHILIA; HUMAN; IMMUNOLOGICAL TOLERANCE; IMMUNOSUPPRESSIVE TREATMENT; MALE; PRESCHOOL CHILD; PRIORITY JOURNAL; RECURRENT INFECTION; RETREATMENT;

ADULT; CHILD, PRESCHOOL; CYCLOPHOSPHAMIDE; FACTOR VIII; HEMOPHILIA A; HUMANS; IMMUNE TOLERANCE; IMMUNOSUPPRESSIVE AGENTS; INFANT; TREATMENT OUTCOME;

EID: 2542562103     PISSN: 03618609     EISSN: None     Source Type: Journal    
DOI: 10.1002/ajh.20066     Document Type: Article

References (20)

1. McMillan CW, Shapiro SS, Whitehurst D, et al. The natural history of factor VIII inhibitors in patients with hemophilia A. A national cooperative study. II. Observations on the initial development of factor VIII.C inhibitors. Blood 1988;71:344-348.
2. Green D. Complications associated with the treatment of haemophiliacs with inhibitors. Haemophilia 1999;5(Suppl 3):11-17.
3. Hoyer LW. Inhibitors in hemophilia. In: Forbes CD, Aledort L, Madhok R, editors. Hemophilia. London: Chapman & Hall; 1997. p 213-227.
4. Hedner U III. Management of patients with factor inhibitors. In: Schechter GP, Berliner N, Telen MJ, editors. Hematology 2000 education program book. Washington, DC: American Society of Hematology; 2000. p 249-252.
5. Brackmann HH, Gormsen J. Massive factor-VIII infusion in haemophiliac with factor-VIII inhibitor, high responder. Lancet 1977;2:933.
6. Brackmann HH. Induced immunotolerance in factor VIII inhibitor patients. Progr Clin Biol Res 1986;150:180-185.
7. Gomperts ED, Jordan S, Church JA, Sakai R, Lemire J. Induction of tolerance to factor VIII in a child with a high-titer inhibitor: In vitro and in vivo observations. J Pediatr 1984;104:70-75.
8. White GC, Taylor RE, Blatt PM, Roberts HR. Treatment of a high titer anti-factor-VIII antibody by continuous factor VIII administration: Report of a case. Blood 1983;58:141.
9. Scheibel E, Ingerslev J, Dalsgaard-Nielsen J, Stensjerg S, Knudsen JB. Continuous high-dose factor VIII for the induction of immune tolerance in haemophilia A patients with high responder state: a description of eleven patients treated. Thromb Haemost 1987;58:1949-1952.
10. Van Leeuwen SF, Mauser-Bunschoten EP, van Dijken PJ, Kok AJ, Sjamsoedin EJ, Sixma JJ. Disappearance of factor VIII.C antibodies in patients with haemophilia A upon frequent administration of factor VIII in intermediate or low dose. Br J Haematol 1986;64:291-297.
11. Mauser-Bunschoten EP, Niewenhuis HK, Rosendaal G, van den Berg HM. Low-dose immune tolerance induction in hemophilia A patients with inhibitors. Blood 1995;86:983-988.
12. Ewing NP, Sanders NL, Dietrich SL, Kasper CK. Induction of immune tolerance of factor VIII in hemophiliacs with inhibitors. J Am Med Assoc 1988;259:65-68.
13. Anzar JA, Jorquera JI, Peiro A, Garcia I. The importance of corticoids added to continued treatment with factor VIII concentrates in the suppression of inhibitors in hemophilia A. Thromb Haemost 1984;51:217-221.
14. Green D. Factor VIII antibodies. Immunosuppressive therapy. Ann NY Acad Sci 1975;240:389-399.
15. Hedner U, Tengborn L. Management of hemophilia A with antibodies: the effect of combined treatment with factor VIII, hydrocortisone and cyclophosphamide. Thromb Haemost 1985;54:776-779.
16. Nilsson IM, Berntorp E, Zarvall D. Induction of immune tolerance in patients with haemophilia and antibodies to factor VIII by combined treatment with intravenous IgG, cyclophosphamide and factor VIII. N Engl J Med 1988;328:947-950.
17. Nilsson IM, Berntorp E, Zarvall D, Dahlback B. Noncoagulation inhibitory factor VIII antibodies after induction of tolerance to factor VIIII in hemophilia A patients. Blood 1990;75:378-383.
18. Nilsson IM, Jonsson S, Sundquist SB, Ahlberg A, Bergentz SE. A procedure for removing high titre antibodies by extracorporeal protein A Sepharose adsorption in hemophilia: substitution therapy and surgery in patients with hemophilia A and antibodies. Blood 1981;58:33-44.
19. Mariani G, Kroner B, Immune Tolerance Study Group (ITSG). Immune tolerance in hemophilia with factor FVIII inhibitors: predictors of success. Haemotologica 2001;86:1186-1193.
20. Kobrinsky NL, Stegman D, Sjolander D. Management of spontaneous inhibitors in children with porcine factor VIII. Haemophilia 2002;8(Suppl 1):1-4.