title: Small cell osteosarcoma successfully treated by high-dose ifosfamide and methotrexate, combined with carboplatin and pirarubicin.
abstract: Small cell osteosarcoma (SCO) is the most rare subtype of osteosarcoma and has a poor prognosis. An 11-year-old boy presented with 2-month history of painful tumefaction in the lower leg. Imaging analysis demonstrated a mixture of osteolytic and osteosclerotic lesions in the proximal tibia and extraskeletal area. Histology of the open biopsy showed small round cells producing mucous matrix. Based on these findings, SCO was suspected. The patient received three cycles of neoadjuvant chemotherapy using high-dose ifosfamide, high-dose methotrexate, pirarubicin and carboplatin. Wide-margin resection was performed followed by tibial lengthening using the Ilizarov method and two cycles of adjuvant chemotherapy with the same drugs as for neoadjuvant chemotherapy. Histology of the resected specimen showed that almost all tumor cells were necrotized. Neither recurrence nor metastasis was found after 4 years. Our experience suggests that neoadjuvant chemotherapy, such as the one used here, would be exceedingly effective for SCO without serious non-hematological toxicities.
authors.affiliation: Department of Orthopaedic Surgery, Hyogo College of Medicine, 1-1 Mukogawa Nishinomiya, Hyogo 663-8501, Japan. h-futani@hyo-med.ac.jp
authors.forename: Hiroyuki
authors.initials: H
authors.lastname: Futani
authors.forename: Satoru
authors.initials: S
authors.lastname: Fukunaga
authors.forename: Yoshitane
authors.initials: Y
authors.lastname: Tsukamoto
authors.forename: Nobuyuki
authors.initials: N
authors.lastname: Terada
authors.forename: Junichiro
authors.initials: J
authors.lastname: Ono
authors.forename: Noriaki
authors.initials: N
authors.lastname: Okamoto
authors.forename: Yoshitoshi
authors.initials: Y
authors.lastname: Otsuka
authors.forename: Takakuni
authors.initials: T
authors.lastname: Tanizawa
authors.forename: Masaki
authors.initials: M
authors.lastname: Tomatsuri
authors.forename: Shinichi
authors.initials: S
authors.lastname: Yoshiya
mesh_terms: D000971:Antineoplastic Combined Chemotherapy Protocols / Q000627:therapeutic use*; D001859:Bone Neoplasms / Q000188:drug therapy* / Q000601:surgery; D016190:Carboplatin / Q000008:administration & dosage; D017024:Chemotherapy, Adjuvant; D002648:Child; D003131:Combined Modality Therapy; D004317:Doxorubicin / Q000008:administration & dosage / Q000031:analogs & derivatives; D006801:Humans; D007069:Ifosfamide / Q000008:administration & dosage; D008297:Male; D008727:Methotrexate / Q000008:administration & dosage; D012516:Osteosarcoma / Q000188:drug therapy* / Q000601:surgery
chemical_list: D004317:Doxorubicin; D016190:Carboplatin; C027260:pirarubicin; D007069:Ifosfamide; D008727:Methotrexate