Second primary osteosarcomas in patients with retinoblastoma

Tomohiro Fujiwara, Miyuki Fujiwara, Kunihiko Numoto, Koichi Ogura, Akihiko Yoshida, Tsukasa Yonemoto, Shigenobu Suzuki, Akira Kawai

Research output: Contribution to journalArticle

5 Citations (Scopus)

Abstract

Objective: Second primary malignancies have become the leading cause of death in retinoblastoma survivors. Although osteosarcoma is the most common second malignancy, little is known about its clinical and therapeutic features. Methods: We retrospectively reviewed a database of patients with retinoblastoma and osteosarcoma occurring as a second malignancy between 1964 and 2010 at the National Cancer Center Hospital of Japan. Results: Among 857 patients with retinoblastoma registered in the database, 10 (1.1%) developed osteosarcoma as a second malignancy. The median age at the onset of retinoblastoma was 3 months, being bilateral in nine patients and unilateral in one. Systemic chemoreduction was performed in three patients and intra-arterial chemotherapy in six; all patients received external beam radiotherapy. The median age at the onset of second primary osteosarcoma was 11.2 years; four were radiation-related and six were located in an extremity. Among five patients treated at our institute, four patients with tumors on an extremity were treated by wide resection with neoadjuvant and adjuvant chemotherapy. Three of these four patients (75%) were good responders to high-dose methotrexate-based multi-agent chemotherapy and survived with no evidence of disease (median follow-up period, 17.3 years). One patient whose temporal bone was affected underwent radiotherapy with chemotherapy but died after local recurrence. Conclusions: The clinical outcomes of second primary osteosarcoma in an extremity occurring in retinoblastoma survivors may be more favorable than those of conventional osteosarcoma. Early diagnosis of radiation-related osteosarcoma arising in the craniofacial region should be made at a stage where complete resection is possible.

Original languageEnglish
Pages (from-to)1139-1145
Number of pages7
JournalJapanese Journal of Clinical Oncology
Volume45
Issue number12
DOIs
Publication statusPublished - 2015
Externally publishedYes

Fingerprint

Retinoblastoma
Osteosarcoma
Second Primary Neoplasms
Extremities
Age of Onset
Drug Therapy
Survivors
Radiotherapy
Databases
Radiation
Cancer Care Facilities
Temporal Bone
Adjuvant Chemotherapy
Methotrexate
Early Diagnosis
Cause of Death
Japan
Recurrence

Keywords

  • Chemotherapy
  • Osteosarcoma
  • Radiotherapy
  • Retinoblastoma
  • Surgery

ASJC Scopus subject areas

  • Oncology
  • Cancer Research
  • Radiology Nuclear Medicine and imaging
  • Medicine(all)

Cite this

Fujiwara, T., Fujiwara, M., Numoto, K., Ogura, K., Yoshida, A., Yonemoto, T., ... Kawai, A. (2015). Second primary osteosarcomas in patients with retinoblastoma. Japanese Journal of Clinical Oncology, 45(12), 1139-1145. https://doi.org/10.1093/jjco/hyv140

Second primary osteosarcomas in patients with retinoblastoma. / Fujiwara, Tomohiro; Fujiwara, Miyuki; Numoto, Kunihiko; Ogura, Koichi; Yoshida, Akihiko; Yonemoto, Tsukasa; Suzuki, Shigenobu; Kawai, Akira.

In: Japanese Journal of Clinical Oncology, Vol. 45, No. 12, 2015, p. 1139-1145.

Research output: Contribution to journalArticle

Fujiwara, T, Fujiwara, M, Numoto, K, Ogura, K, Yoshida, A, Yonemoto, T, Suzuki, S & Kawai, A 2015, 'Second primary osteosarcomas in patients with retinoblastoma', Japanese Journal of Clinical Oncology, vol. 45, no. 12, pp. 1139-1145. https://doi.org/10.1093/jjco/hyv140
Fujiwara T, Fujiwara M, Numoto K, Ogura K, Yoshida A, Yonemoto T et al. Second primary osteosarcomas in patients with retinoblastoma. Japanese Journal of Clinical Oncology. 2015;45(12):1139-1145. https://doi.org/10.1093/jjco/hyv140
Fujiwara, Tomohiro ; Fujiwara, Miyuki ; Numoto, Kunihiko ; Ogura, Koichi ; Yoshida, Akihiko ; Yonemoto, Tsukasa ; Suzuki, Shigenobu ; Kawai, Akira. / Second primary osteosarcomas in patients with retinoblastoma. In: Japanese Journal of Clinical Oncology. 2015 ; Vol. 45, No. 12. pp. 1139-1145.
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