Osteochondroma in long‐term survivors of high‐risk neuroblastoma

Abstract

Background Osteochondromas are benign bony protrusions that can be spontaneous or associated with radiation therapy (RT). Current treatment of high-risk neuroblastoma includes dose-intensive chemotherapy, local RT, anti-GD2 monoclonal antibody (MAb), and isotretinoin. Late effects are emerging. Methods We studied osteochondromas in 362 patients who were <10 years old when diagnosed with neuroblastoma; received MAb+isotretinoin since 2000; and survived ≥24 months from the first dose of MAb. The incidence rate of osteochondromas was determined using the competing risks approach where the primary event was osteochondroma calculated from the date of neuroblastoma diagnosis and the competing event was death without osteochondroma. Results Twenty-one osteochondromas were found in 14 patients who were 5.7-15.3 (median 10.4) years of age and 3.1-11.2 (median 8.2) years from neuroblastoma diagnosis. The cumulative incidence rate was 0.6% at five years and 4.9% at 10 years from neuroblastoma diagnosis. Nine osteochondromas were revealed incidentally during assessments of neuroblastoma disease status or bone age. Thirteen osteochondromas were outside RT portals and had characteristics of spontaneous forms. Complications were limited to pain necessitating resection in three patients, but follow-up is short at 0.3-7.7 (median 3.5) years. Conclusions Osteochondromas in long-term survivors of neuroblastoma should be expected because these benign growths can be RT-related and these patients undergo radiologic studies over years, are monitored for late toxicities through and beyond adolescence, and receive special attention (because of concern about relapse) if they develop a bony protuberance. A pathogenic role for chemotherapy, anti-GD2 MAb, or isotretinoin remains speculative.

Osteochondroma: Review of 431 patients from one medical institution in South China

“…Osteochondroma (OC), also known as osteocartilaginous exostosis or cartilage-capped exostosis, is a broad (sessile) or narrow (pedunculated) skeletal protrusion comprised of marrow and cortical bone [1] , [2] , [3] , [4] . The cartilaginous cap is the site of growth, which normally diminishes after skeletal maturity.…”

“…The tumor is presented as a locally benign neoplasm, which favors the meta-epiphyseal region of long bones like the distal femur, as well as the proximal tibia or humerus in the extremity [5] , [6] , [8] . Of all benign bone tumors, OC accounts for more than one-third, the most common benign bone tumor around the world [4] , [7] , [8] , [9] , [10] , [11] , [15] . Although it is benign, its biological behavior still has a malignant potential.…”

Late effects in survivors of high‐risk neuroblastoma following stem cell transplant with and without total body irradiation

“…Benign bone tumors are commonly noted in survivors, particularly post radiation. [31][32][33] Several studies have noted osteochondromas and other orthopedic findings in neuroblastoma survivors. 3,4 In this cohort, we found SCFEs in 18% of survivors.…”

Characterization of Solitary Lesions in the Extremities on Whole-Body Bone Scan in Patients With Known Cancer: Contribution of Single-Photon Emission Computed Tomography/Computed Tomography

“…In addition, the majority (13/20.65%) of benign bone tumors were enchondromas, which were frequently found in the proximal femur, accounting for 61.53% (8/13). Although enchondroma occurs mostly in the metaphysis (16, 17), only 2.6% of all benign bone tumors are enchondromas (18–25). On the one hand, the majority of enchondromas are asymptomatic and most often identified incidentally for other reasons (26–28).…”