title: Successful immune tolerance induction using turoctocog alfa in an adult haemophilia A patient.
abstract: We present here the first known case of successful immune tolerance induction (ITI) using recombinant factor VIII (rFVIII), turoctocog alfa, in a patient with severe haemophilia A. The 38-year-old patient with a long-standing inhibitor required urgent surgery for severe arthropathy. rFVIII was administered throughout the surgical period. Surgery was considered successful, but on day 7 after surgery, an increased level of FVIII inhibitors were detected. ITI was attempted immediately thereafter according to the Bonn protocol. Inhibitors were no longer detected on day 17; 13 months later, successful ITI was achieved. This case suggests that a long-time interval between inhibitor appearance and the start of ITI therapy may not necessarily indicate poor prognosis.
authors.affiliation: aDepartment of Haematology and Oncology - Internal Medicine V, Medical University of Innsbruck, Innsbruck, Austria bCentral Institute for Medical and Chemical Laboratory Diagnostics, Innsbruck, Austria cDepartment of Internal Medicine VI - Infectious Diseases, Immunology, Rheumatology, Pneumology, Medical University of Innsbruck, Innsbruck, Austria dHaematology, Hemostasis, Oncology, and Stem Cell Transplantation, Hannover Medical School, Hannover, Germany.
authors.forename: Clemens
authors.initials: C
authors.lastname: Feistritzer
authors.forename: Sophie Maria
authors.initials: SM
authors.lastname: Wildner
authors.forename: Philipp
authors.initials: P
authors.lastname: Würtinger
authors.forename: David
authors.initials: D
authors.lastname: Nachbaur
authors.forename: Andreas
authors.initials: A
authors.lastname: Tiede
authors.forename: Michael
authors.initials: M
authors.lastname: Steurer
mesh_terms: D000328:Adult; D005169:Factor VIII / Q000008:administration & dosage / Q000627:therapeutic use*; D006467:Hemophilia A / Q000276:immunology*; D006490:Hemostatics; D006801:Humans; D007108:Immune Tolerance; D008297:Male; D011994:Recombinant Proteins / Q000008:administration & dosage / Q000627:therapeutic use*
chemical_list: D006490:Hemostatics; D011994:Recombinant Proteins; C577506:recombinant factor VIII N8; D005169:Factor VIII