Denosumab in patients with aneurysmal bone cysts: A case series with preliminary results

Emanuela Palmerini, Pietro Ruggieri, Andrea Angelini, Stefano Boriani, Domenico Campanacci, Giuseppe M Milano, Marilena Cesari, Anna Paioli, Alessandra Longhi, Massimo E Abate, Guido Scoccianti, Silvia Terzi, Giulia Trovarelli, Alessandro Franchi, Piero Picci, Stefano Ferrari

Research output: Contribution to journalArticle

Abstract

PURPOSE: Aneurysmal bone cyst (ABC) is a rare skeletal tumor usually treated with surgery/embolization. We hypothesized that owing to similarities with giant cell tumor of bone (GCTB), denosumab was active also in ABC.

METHODS: In this observational study, a retrospective analysis of ABC patients treated with denosumab was performed. Patients underwent radiologic disease assessment every 3 months. Symptoms and adverse events were noted.

RESULTS: Nine patients were identified (6 male, 3 female), with a median age of 17 years (range 14-42 years). Primary sites were 6 spine-pelvis, 1 ulna, 1 tibia, and 1 humerus. Patients were followed for a median time of 23 months (range 3-55 months). Patients received a median of 8 denosumab administrations (range 3-61). All symptomatic patients had pain relief and 1 had paresthesia improvement. Signs of denosumab activity were observed after 3 to 6 months of administration: bone formation by computed tomography scan was demonstrated in all patients and magnetic resonance imaging gadolinium contrast media decrease was observed in 7/9 patients. Adverse events were negligible. At last follow-up, all patients were progression-free: 5 still on denosumab treatment, 2 off denosumab were disease-free 11 and 17 months after surgery, and the last 2 patients reported no progression 12 and 24 months after denosumab interruption and no surgery.

CONCLUSIONS: Denosumab has substantial activity in ABCs, with favorable toxicity profile. We strongly support the use of surgery and/or embolization for the treatment of ABC, but denosumab could have a role as a therapeutic option in patients with uncontrollable, locally destructive, or recurrent disease.

Original languageEnglish
Pages (from-to)344-351
Number of pages8
JournalTumori
Volume104
Issue number5
DOIs
Publication statusE-pub ahead of print - Aug 8 2018

Keywords

  • Aneurysmal bone cyst
  • bone tumor
  • denosumab
  • receptor activator of nuclear factor-kappa B ligand

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    Palmerini, E., Ruggieri, P., Angelini, A., Boriani, S., Campanacci, D., Milano, G. M., Cesari, M., Paioli, A., Longhi, A., Abate, M. E., Scoccianti, G., Terzi, S., Trovarelli, G., Franchi, A., Picci, P., & Ferrari, S. (2018). Denosumab in patients with aneurysmal bone cysts: A case series with preliminary results. Tumori, 104(5), 344-351. https://doi.org/10.1177/0300891618784808