title: The International Immune Tolerance Induction Study and its follow-up study on Japanese hemophilia A patients with inhibitors.
abstract: The International Immune Tolerance Induction (I-ITI) Study in hemophilia A patients with inhibitors included 16 Japanese patients among a total of 115 test subjects. The results within this group of Japanese patients were 11 cases of I-ITI off-study, three cases of I-ITI on-study, and two cases of tolerance on prophylaxis. There was no significant difference in success rate between the low-dose and high-dose groups (Study I). Successively, independent follow-up survey in Japan was conducted in 14 cases, with consent (Study II). Ten cases were off-study at the end of the I-ITI Study. Of these 10 cases, seven of seven successful cases remained clinical successes at the end of the follow-up study, one partial success became a full success while a second relapsed, and one failure was subsequently evaluated as a partial success. Four cases that were on-study at the end of I-ITI Study were classified as three successes and one failure at the end of the follow-up study. As a result, the status at the end of follow-up study was: 11 ITI successes (78.6 %); one partial success; one failure; and one relapse. Thus, the ITI follow-up study was helpful in providing a long-term prognostic determination of inhibitors.
authors.affiliation: Nara Medical University, Emeritus Professor, 840 Shijo-cho, Kashihara, Nara, 634-8521, Japan, ks840@naramed-u.ac.jp.
authors.forename: Akira
authors.initials: A
authors.lastname: Yoshioka
authors.forename: Eizaburo
authors.initials: E
authors.lastname: Ishii
authors.forename: Tamaki
authors.initials: T
authors.lastname: Ueno
authors.forename: Ikuya
authors.initials: I
authors.lastname: Usami
authors.forename: Masao
authors.initials: M
authors.lastname: Kobayashi
authors.forename: Ryoji
authors.initials: R
authors.lastname: Kobayashi
authors.forename: Manabu
authors.initials: M
authors.lastname: Sotomatsu
authors.forename: Akira
authors.initials: A
authors.lastname: Shirahata
authors.forename: Takashi
authors.initials: T
authors.lastname: Suzuki
authors.forename: Masashi
authors.initials: M
authors.lastname: Taki
authors.forename: Yasushi
authors.initials: Y
authors.lastname: Ishida
authors.forename: Tadashi
authors.initials: T
authors.lastname: Matsushita
authors.forename: Midori
authors.initials: M
authors.lastname: Shima
authors.forename: Keiji
authors.initials: K
authors.lastname: Nogami
authors.forename: Michio
authors.initials: M
authors.lastname: Sakai
authors.forename: Hisato
authors.initials: H
authors.lastname: Kigasawa
authors.forename: Katsuyuki
authors.initials: K
authors.lastname: Fukutake
mesh_terms: D044466:Asian People; D002675:Child, Preschool; D005169:Factor VIII / Q000008:administration & dosage / Q000276:immunology* / Q000627:therapeutic use*; D005260:Female; D005500:Follow-Up Studies; D006467:Hemophilia A / Q000453:epidemiology / Q000276:immunology* / Q000628:therapy*; D006801:Humans; D007108:Immune Tolerance; D007223:Infant; D007564:Japan / Q000453:epidemiology; D008297:Male; D011994:Recombinant Proteins / Q000008:administration & dosage / Q000276:immunology / Q000627:therapeutic use
chemical_list: D011994:Recombinant Proteins; D005169:Factor VIII