Open Access
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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