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Table 1.

Published and current cases of Denosumab treatment of ABC.

Case 1
Case 2
Case 3
Case 4
Case 5
Case 6
Case 7
Current case
Age/sex 8-year male 11-year male 5-year male 27-year male 26-year female 42-year male 16-year male 14-year female
Location C5 C5 Sacrum Sacrum C7–T1 L5–S1 L5 D5
Diagnosis Typical ABC Typical ABC Typical ABC Typical ABC Typical ABC Aggressive ABC Typical ABC Typical ABC
Initial treatment Surgery Surgery None None C4–T4 fixation, cervical laminectomy Seven consecutive SAE Five consecutive monthly SAEs Surgery twice
Outcome of initial treatment Recurrence at 1 month Recurrence at 8 months NA NA NA (adjuvant therapy) Recurrence at 6 months, worsening radiculopathy Pathologic vascularity after embolisation, right sciaticalgia Recurrence at 2 months
Denosumab treatment 70 mg/m2 sc monthly 70 mg/m2 sc D1, 8, 15, 21, 28 and then monthly 1.2 mg/kg/dose sc D1, 8, 15, 21, 28 then monthly Denosumab 120 mg sc D1, 8, 15, 28 and then monthly for 1 yr., followed by observation 120 mg sc monthly for 16 months 120 mg once a week for 4 weeks consecutively, then once every 40 days, 13 Denosumab administrations 120 mg once a week for 4 weeks consecutively, then once every 40 days, 11 Denosumab administrations Denosumab 120 mg sc D1, 8, 15, 28 and then monthly for 6 months, followed by observation
Response to Denosumab Recovery from neurological symptoms, and no progression of ABC, with evidence of bone formation on MRI at 2 months Recovery from neurological symptoms, and no progression of ABC, with evidence of bone formation on MRI at 4 months Resolution of pain and new bone formation with healing of pathologic fracture on MRI Resolution of pain and new bone formation on CT scan, decrease in giant cells, and decrease in cellularity of fibrous stroma Persistence of C8–T1 deficit, peripheral bone reconstruction, decrease in tumour size and bone fusion at 19.5 months follow-up Complete ossification at 35 months CT scan. Pain-free gradual mass reduction and progressive ossification since the 3rd month of administration. 33 months follow-up Resolution of pain and new bone formation on MRI scan, complete recovery of neurologic worsening. No recurrence at 1.5 year follow-up
Follow-up 2 months 4 months 12 months 12 months 19.5 months 35 months 33 months 24 months
References Lange et al. [6] Lange et al. [6] Pelle et al. [17] Skubitz et al. [13] Arnaud Dubory et al. [15] Ghermandi et al. [14] Ghermandi et al. [14]

Note: ABC indicates aneurysmal bone cyst; SAE, Selective Arterial Embolisation; NA, not applicable; CT, computed tomographic; MRI, magnetic resonance image; sc, sub-cutaneous.

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