Cancer Res Treat.  2021 Jan;53(1):279-282. 10.4143/crt.2020.384.

Chondroblastoma’s Lung Metastases Treated with Denosumab in Pediatric Patient

• Focaccia M
• Gambarotti M
• Hakim R
• Paioli A
• Cesari M
• Spazzoli B
• Spinnato P
• Donati D
• Rocca M
• Longhi A

• KMID: 2510670
• DOI: http://doi.org/10.4143/crt.2020.384

Abstract

Chondroblastoma is a rare benign chondrogenic tumor that occurs in skeletally immature patients between ages 10 and 20 years old. In literature are reported few cases of lung metastases, mainly occurred after surgery or local recurrences. There is no evidence on the pathogenesis of lung metastasis, as well as pulmonary disease course. Few treatments for metastases with aggressive behavior were based on chemotherapy regimen employed in other sarcoma with no results or not satisfying ones. Denosumab is approved for treatment of giant cell tumors and it is under investigation for other giant cell-rich bone tumors. Here, we report a case of a 16-year-old male chondroblastoma of the left humerus with bilateral lung metastases at presentation and progressing during follow-up, treated with denosumab for almost 2 years. We confirm that denosumab treatment can be effective in controlling chondroblastoma metastasis and it has been a safe procedure in an adolescent patient.

Keyword

Chondroblastoma; Lung; Metastasis; Chemotherapy; Denosumab; Pediatrics

Figure

• 

  Fig. 1. (A) Humeral lesion: histological features consistent with chondroblastoma (H&E staining, x10). (B) Lung metastasis: immunohistochemical positivity for H3F3B in neoplastic cells in the lung metastasis from chondroblastoma (x10).

• 

  Fig. 2. Lung computed tomography, at diagnosis in January 2018 (A), at the beginning of denosumab treatment in July 2018 (B) and after 20 months of treatment in March 2020 (C), shows metastasis at the right upper lobe slightly growing in dimensions before the treatment. The metastasis dimensions remain stable after 20 months of denosumab; intralesional calcifications are detectable in the last control (C).

• 

  Fig. 3. Lung computed tomography, at diagnosis in January 2018 (A), at the beginning of denosumab treatment in July 2018 (B) and after 20 months of treatment in March 2020 (C), shows metastasis at the right lower lobe growing in dimensions before the treatment. The metastasis dimensions remain stable after 20 months of denosumab; intralesional calcifications are detectable in the last control (C).

Reference

References

1. Dahlin DC, Ivins JC. Benign chondroblastoma: a study of 125 cases. Cancer. 1972; 30:401–13.

2. John I, Inwards CY, Wenger DE, Williams DD, Fritchie KJ. Chondroblastomas presenting in adulthood: a study of 39 patients with emphasis on histological features and skeletal distribution. Histopathology. 2020; 76:308–17.

3. Rehkamper J, Steinestel K, Jeiler B, Elges S, Hekeler E, Huss S, et al. Diagnostic tools in the differential diagnosis of giant cell-rich lesions of bone at biopsy. Oncotarget. 2018; 9:30106–14.

4. Righi A, Mancini I, Gambarotti M, Picci P, Gamberi G, Marraccini C, et al. Histone 3.3 mutations in giant cell tumor and giant cell-rich sarcomas of bone. Hum Pathol. 2017; 68:128–35.

5. Chen W, DiFrancesco LM. Chondroblastoma: an update. Arch Pathol Lab Med. 2017; 141:867–71.

6. Ostrowski ML, Johnson ME, Truong LD, Hicks MJ, Smith FE, Spjut HJ. Malignant chondroblastoma presenting as a recurrent pelvic tumor with DNA aneuploidy and p53 mutation as supportive evidence of malignancy. Skeletal Radiol. 1999; 28:644–50.

7. Kunze E, Graewe T, Peitsch E. Histology and biology of metastatic chondroblastoma: report of a case with a review of the literature. Pathol Res Pract. 1987; 182:113–23.

8. Ozkoc G, Gonlusen G, Ozalay M, Kayaselcuk F, Pourbagher A, Tandogan RN. Giant chondroblastoma of the scapula with pulmonary metastases. Skeletal Radiol. 2006; 35:42–8.

9. Kyriakos M, Land VJ, Penning HL, Parker SG. Metastatic chondroblastoma: report of a fatal case with a review of the literature on atypical, aggressive, and malignant chondroblastoma. Cancer. 1985; 55:1770–89.

10. Chawla S, Blay JY, Rutkowski P, Le Cesne A, Reichardt P, Gelderblom H, et al. Denosumab in patients with giant-cell tumour of bone: a multicentre, open-label, phase 2 study. Lancet Oncol. 2019; 20:1719–29.

11. Raux S, Bouhamama A, Gaspar N, Brugieres L, Entz-Werle N, Mallet C, et al. Denosumab for treating aneurysmal bone cysts in children. Orthop Traumatol Surg Res. 2019; 105:1181–5.

12. Upfill-Brown A, Bukata S, Bernthal NM, Felsenfeld AL, Nelson SD, Singh A, et al. Use of denosumab in children with osteoclast bone dysplasias: report of three cases. JBMR Plus. 2019; 3:e10210.

13. Elek EM, Grimer RJ, Mangham DC, Davies AM, Carter SR, Tillman RM. Malignant chondroblastoma of the os calcis. Sarcoma. 1998; 2:45–8.

14. Calvert N, Wood D. Use of denosumab in recurrent chondroblastoma of the squamous temporal bone: a case report. Clin Case Rep. 2017; 5:411–3.

15. Huang TH, Liu HC, Hou JY, Chang CY, Sun FJ, Yeh TC. Efficacy and safety of denosumab therapy for low bone mineral density in childhood cancer survivors: a report of preliminary experience. Pediatr Blood Cancer. 2019; 66:e27927.