Nationwide survey of bisphosphonate therapy for children with reactivated Langerhans cell histiocytosis in Japan

Akira Morimoto, Yoko Shioda, Toshihiko Imamura, Hirokazu Kanegane, Takashi Sato, Kazuko Kudo, Shinichiro Nakagawa, Hisaya Nakadate, Hisamichi Tauchi, Asahito Hama, Masahiro Yasui, Yoshihisa Nagatoshi, Akitoshi Kinoshita, Ryosuke Miyaji, Tadashi Anan, Miharu Yabe, Junji Kamizono

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Abstract

Background: Several studies have suggested that Langerhans cell histiocytosis (LCH) is responsive to treatment with bisphosphonates (BPs). However the efficacy and safety of BPs therapy for childhood LCH is unknown. Procedure: Data on children with LCH who had received BPs therapy were collected retrospectively from hospitals participating in the Japanese Pediatric Leukemia/Lymphoma Study Group. Results: Twenty-one children with histologically proven LCH were identified. Of these, the case histories of 16 children who had been treated with pamidronate (PAM) for disease reactivation were analyzed in detail. The median post-PAM therapy follow-up period was 2.8 years (range: 0.9-9.3 years). The median age at commencement of PAM therapy was 9.4 years (range: 2.3-15.0 years). All children had one or more bone lesions but none had risk organ (RO) involvement. In the majority of the children, six courses of PAM were administered at a dose of 1.0 mg/kg/course at 4-week intervals. In 12 of the 16 children, all active lesions including lesions of the skin (n = 3) and soft tissues (n = 3) resolved. Of these children, eight children had no active disease for a median of 3.3 years post-PAM therapy (range: 1.8-9.3 years). Progression-free survival (PFS) was 56.3 ± 12.4% at 3 years. PFS was significantly higher in children with a first reactivation compared with children experiencing a second or subsequent reactivation. Conclusions: PAM may be an effective treatment for reactivated LCH with bone lesions. A prospective trial of the efficacy of PAM in recurrent pediatric LCH is warranted.

Original languageEnglish
Pages (from-to)110-115
Number of pages6
JournalPediatric Blood and Cancer
Volume56
Issue number1
DOIs
Publication statusPublished - 01-2011

All Science Journal Classification (ASJC) codes

  • Pediatrics, Perinatology, and Child Health
  • Hematology
  • Oncology

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    Morimoto, A., Shioda, Y., Imamura, T., Kanegane, H., Sato, T., Kudo, K., Nakagawa, S., Nakadate, H., Tauchi, H., Hama, A., Yasui, M., Nagatoshi, Y., Kinoshita, A., Miyaji, R., Anan, T., Yabe, M., & Kamizono, J. (2011). Nationwide survey of bisphosphonate therapy for children with reactivated Langerhans cell histiocytosis in Japan. Pediatric Blood and Cancer, 56(1), 110-115. https://doi.org/10.1002/pbc.22703