Osteochondroma in long-term survivors of high-risk neuroblastoma Journal Article


Authors: Kushner, B. H.; Roberts, S. S.; Friedman, D. N.; Kuk, D.; Ostrovnaya, I.; Modak, S.; Kramer, K.; Basu, E. M.; Cheung, N. K. V.
Article Title: Osteochondroma in long-term survivors of high-risk neuroblastoma
Abstract: BACKGROUND Osteochondromas are benign bony protrusions that can be spontaneous or associated with radiotherapy (RT). Current treatment of high-risk neuroblastoma includes dose-intensive chemotherapy, local RT, an anti-G<inf>D2</inf> monoclonal antibody (MoAb), and isotretinoin. Late effects are emerging. METHODS The authors examined osteochondromas in 362 patients who were aged <10 years when diagnosed with neuroblastoma, had received a MoAb plus isotretinoin since 2000, and had survived >24 months from the time of the first dose of the MoAb. The incidence rate of osteochondroma was determined using the competing risks approach, in which the primary event was osteochondroma calculated from the date of neuroblastoma diagnosis and the competing event was death without osteochondroma. RESULTS A total of 21 osteochondroma cases were found among 14 patients who were aged 5.7 to 15.3 years (median, 10.4 years) and 3.1 to 11.2 years (median, 8.2 years) from the time of neuroblastoma diagnosis. The cumulative incidence rate was 0.6% at 5 years and 4.9% at 10 years from the neuroblastoma diagnosis. Nine osteochondromas were revealed incidentally during assessments of neuroblastoma disease status or bone age. Thirteen osteochondromas were detected outside RT portals and had characteristics of spontaneous forms. Complications were limited to pain necessitating surgical resection in 3 patients, but follow-up was short at 0.3 to 7.7 years (median, 3.5 years). CONCLUSIONS Osteochondromas in long-term survivors of neuroblastoma should be expected because these benign growths can be related to RT and these patients undergo radiologic studies over years, are monitored for late toxicities through and beyond adolescence, and receive special attention (because of concerns about disease recurrence) if they develop a bony protuberance. A pathogenic role for chemotherapy, anti-G<inf>D2</inf> MoAbs, or isotretinoin remains speculative. Cancer 2015. © 2015 American Cancer Society.
Keywords: adult; cancer survival; clinical article; cancer surgery; unclassified drug; postoperative period; drug efficacy; cancer radiotherapy; radiation dose; cancer incidence; cancer immunotherapy; multiple cycle treatment; granulocyte macrophage colony stimulating factor; high risk patient; radiation response; cancer survivor; monoclonal antibody; neuroblastoma; drug response; age distribution; isotretinoin; radiotoxicity; osteochondroma; bone tumors; monoclonal antibody gd2; human; male; female; priority journal; article; long-term toxicity
Journal Title: Cancer
Volume: 121
Issue: 12
ISSN: 0008-543X
Publisher: Wiley Blackwell  
Date Published: 2015-06-15
Start Page: 2090
End Page: 2096
Language: English
DOI: 10.1002/cncr.29316
PROVIDER: scopus
PUBMED: 25728463
PMCID: PMC4970322
DOI/URL:
Notes: Export Date: 2 July 2015 -- Source: Scopus
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MSK Authors
  1. Brian Kushner
    311 Kushner
  2. Nai-Kong Cheung
    648 Cheung
  3. Kim Kramer
    236 Kramer
  4. Shakeel Modak
    249 Modak
  5. Danielle Novetsky Friedman
    110 Friedman
  6. Deborah Kuk
    87 Kuk
  7. Ellen Marlese Basu
    101 Basu
  8. Stephen Stacy Roberts
    107 Roberts