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The International Immune Tolerance Induction Study and its follow-up study on Japanese hemophilia A patients with inhibitors

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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.

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Acknowledgments

The authors thank the Blood Products Research Organization, Japan, for its financial support of the domestic follow-up study and Mrs. Michiyo Goto (NICP) and Miss Nari Hashimoto (Nara Medical University) for their assistance in preparing the manuscript.

Conflict of interest

The authors declare that they have no conflict of interest.

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Authors and Affiliations

  1. Nara Medical University, Emeritus Professor, 840 Shijo-cho, Kashihara, Nara, 634-8521, Japan

    Akira Yoshioka

  2. Division of Hematology/Oncology, Nagano Prefectural Suzaka Hospital, Suzaka, Nagano, Japan

    Eizaburo Ishii

  3. Division of Pediatrics, Ayabe City Hospital, Ayabe, Kyoto, Japan

    Tamaki Ueno

  4. Division of Pediatrics, Hyogo Prefectural Tsukaguchi Hospital, Amagasaki, Hyogo, Japan

    Ikuya Usami

  5. Department of Pediatrics, Hiroshima University School of Medicine, Hiroshima, Japan

    Masao Kobayashi

  6. Division of Pediatrics, Sapporo Hokuyu Hospital, Sapporo, Hokkaido, Japan

    Ryoji Kobayashi

  7. Division of Hematology/Oncology, Gunma Children’s Medical Center, Shibukawa, Gunma, Japan

    Manabu Sotomatsu

  8. Kitakyushu Yahata Higashi Hospital, Kitakyushu, Fukuoka, Japan

    Akira Shirahata

  9. Department of Clinical Laboratory Medicine, Tokyo Medical University, Tokyo, Japan

    Takashi Suzuki & Katsuyuki Fukutake

  10. Department of Pediatrics, St. Marianna University School of Medicine, Yokohama City Seibu Hospital, Yokohama, Kanagawa, Japan

    Masashi Taki

  11. Division of Pediatrics, Ehime Prefectural Central Hospital, Matsuyama, Ehime, Japan

    Yasushi Ishida

  12. Division of Blood Transfusion, Nagoya University Hospital, Nagoya, Aichi, Japan

    Tadashi Matsushita

  13. Department of Pediatrics, Nara Medical University, Kashihara, Nara, Japan

    Midori Shima & Keiji Nogami

  14. Department of Pediatrics, University of Occupational and Environmental Health, Japan, Kitakyushu, Fukuoka, Japan

    Michio Sakai

  15. Division of Hematology/Oncology, Kanagawa Children’s Medical Center, Yokohama, Kanagawa, Japan

    Hisato Kigasawa

Authors
  1. Akira Yoshioka
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  2. Eizaburo Ishii
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  3. Tamaki Ueno
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  4. Ikuya Usami
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  5. Masao Kobayashi
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  6. Ryoji Kobayashi
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  7. Manabu Sotomatsu
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  8. Akira Shirahata
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  9. Takashi Suzuki
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  10. Masashi Taki
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  11. Yasushi Ishida
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  12. Tadashi Matsushita
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  13. Midori Shima
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  14. Keiji Nogami
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  15. Michio Sakai
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  16. Hisato Kigasawa
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  17. Katsuyuki Fukutake
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Corresponding author

Correspondence to Akira Yoshioka.

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Yoshioka, A., Ishii, E., Ueno, T. et al. The International Immune Tolerance Induction Study and its follow-up study on Japanese hemophilia A patients with inhibitors. Int J Hematol 101, 362–368 (2015). https://doi.org/10.1007/s12185-015-1734-z

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  • DOI: https://doi.org/10.1007/s12185-015-1734-z

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