Aneurysmal bone cyst: results of an off label treatment with Denosumab.

BACKGROUND: The treatment of aneurysmal bone cysts (ABCs) has evolved and less invasive methods have been tried. Denosumab is a monoclonal antibody which inhibits osteoclasts. It has been shown to be effective in giant cell tumour of bone (GCT) of bone and hence promises some effect also in ABC. We report on 6 patients treated with Denosumab and compare our results to the cases already published. METHODS: Data of 6 patients with ABCs and patients whose treatment included Denosumab were retrospectively analyzed. Denosumab was used at a dose of 120 mg on days 1, 8, 15 and 29, and every 4 weeks thereafter. In some of these patients the dose was reduced at the end of the treatment. Clinical and radiological responses were evaluated. RESULTS: In 4 female and 2 male patients with a mean age of 17 years (range: 6-30 years) the lesions were located in the sacrum (2), in distal radius, distal femur, talus and pelvis. One of the sacral lesions healed after 12 months and has stayed stable for 3 years since. The second patient received 2 years of therapy with recalcification, but recurred 1 year later and is under renewed therapy. The pelvic lesion improved but recurred. This patient has a 13-years history of intermittent therapy including surgery, two pregnancies and remains in a stable situation. The lesion of the talus did not improve with Denosumab after surgery and was complicated by destruction of the ankle joint with osteoarthritis. Recurrent lesions of the distal femur and the distal radius, previously treated by curettage and bone grafting healed under Denosumab and have remained stable for 2 and 3 years, respectively. One case of severe hypercalcemia was observed in a 7-year old child 6 months after discontinuation of Denosumab. CONCLUSION: Denosumab provides a treatment option for ABCs in anatomically critical locations. Adjuvant application might reduce the rate of local recurrence. In young patients, severe rebound hypercalcemia months after discontinuation of Denosumab may occur.

Aneurysmal bone cyst: A review of 65 patients.

BACKGROUND: Aneurysmal bone cysts (ABC) are benign but locally aggressive lesions. The treatment of ABC has evolved over the years, but curettage with or without local adjuvants still represents the standard. Less invasive methods such as embolization, sclerotherapy or RANKL inhibitors (Denosumab) are also established. The aim of this study was to report and compare the results of a series of patients mainly treated with curettage with and without subsequent phenolization. METHODS: 65 patients with the unequivocal diagnosis of primary ABC were treated. 61 of them were located within the bone whereas 4 patients had an ABC of the soft tissues. All patient were treated surgically by means of curettage with or without adjuvants, resection, or with minimally invasive methods such as Polidocanol injections, embolizations or Denosumab treatment. In total 80 procedures had been performed. RESULTS: Our patients had a mean age of 25.3 ±â€¯16.0 years, ranging from 4 to 74 years. The most common skeletal locations were the pelvis in 23%, the femur in 18%, the tibia in 16% and the spine in 10%. Six lesions were resected and showed no recurrence. 5 patients were treated with polidocanol injections (n = 3) or embolization plus systemic treatment with Denosumab (n = 2). With embolization and Denosumab both patients showed stable disease and required no further treatment. Polidocanol injections resulted in stable disease with no further treatment required in one patient and in subsequent curettage with adjuvant phenolization in two other patients.In 54 initial curettages 21 were performed with adjuvant phenolization. In this group, 16 lesions healed (76%), 3 showed persistent disease and 2 patients had a local recurrence (9%). Out of 33 patients without phenolization 21 (64%) healed, 3 showed stable persistent disease and 9 (27%) experienced a recurrence. In total we performed 66 curettages, 27 with and 39 without adjuvant phenol treatment. Resolution was achieved in 19 (70%) and 25 (64%) of cases. respectively. Persistent disease was evident in 5 cases each and recurrence in 3 and 9 cases, respectively (n.s.). CONCLUSION: Curettage is still the standard of treatment for ABC. Local recurrence does not depend on the use of adjuvant phenol as shown in this and other studies. Minimally invasive methods such as selective embolization and injections of sclerosing agents may result in healing or at least in tolerable persistence of residual lesions but needs repetitive treatments and does not show homogenous results throughout the institutions. Denosumab appears to be an additional option, especially in surgically critical locations such as the spine or the sacrum.