Massive aneurysmal bone cyst of the skull base treated with denosumab

Asi, Karim W.; Abdelmeguid, Ahmed S.; Bell, Diana; Hanna, Ehab Y.

doi:10.1002/hed.25438

Cited by 15 publications

(6 citation statements)

References 25 publications

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“…In 2018, Asi et al [26] (a case report from the United States of America) reported a 32-year-old male patient with a massive primary ABC involving the maxillary sinus and anterior skull base. The patient had significant uncontrolled tumor-related pain and multiple progressive cranial neuropathies.…”

Section: Reviewmentioning

confidence: 99%

Denosumab Therapy in the Management of Aneurysmal Bone Cysts: A Comprehensive Literature Review

Alhumaid¹,

Abu‐Zaid²

2019

Cureus

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Aneurysmal bone cysts (ABCs) are uncommon lesions that involve the axial and appendicular bones. Although biologically benign, ABCs have the tendency to assume an aggressive behavior causing local destruction of the underlying bone and neighboring soft tissues. Morphologically, ABCs are composed of cyst-like cavities filled with blood and bounded by an array of diverse cells including fibroblasts, inflammatory infiltrates and osteoclast-like multinucleated giant cells. From a molecular perspective, the osteoclast-like multinucleated giant cells harbor high expression of receptor activator of nuclear factor kappa B (RANK) receptors, whereas the neoplastic stromal cells harbor high expression of RANK ligand (RANKL). The RANK-RANKL interaction has been implicated in the carcinogenesis of ABCs and giant cell tumor of bones (GCTBs). Currently, the optimal management of ABCs remains a hotly debated topic. There are a multitude of treatment modalities (that is, surgery, sclerotherapy, radiotherapy and selective arterial embolization), and each modality has its own benefits, morbidity and risk of complications. The local aggressiveness of ABC and its high rates of relapse following treatment has demanded the march towards discovering more innovative therapies. One of such therapies is denosumab, a monoclonal antibody targeted against the RANKL. Denosumab is already approved by the United States Food and Drug Administration (FDA) for the treatment of adults and skeletally mature adolescents with GCTB that is unamenable to surgery, or initial surgery is anticipated to result in significant morbidity. However, denosumab is not approved by the FDA for the management of ABCs. However, taking into consideration the morphological similarity between GCTBs and ABCs, some treating physicians have logically opted to use denosumab in an off-label manner to treat select ABCs. To the best of knowledge, no study has attempted to summarize the related literature on the use of denosumab in ABCs. Therefore, the primary aim of this study is to narratively review all the available literature about the efficacy and safety of the use of off-label denosumab in the management of patients with ABCs.

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Section: Reviewmentioning

confidence: 99%

Denosumab Therapy in the Management of Aneurysmal Bone Cysts: A Comprehensive Literature Review

Alhumaid¹,

Abu‐Zaid²

2019

Cureus

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“…To our best knowledge, 2 case series (with 9 patients each) have previously been published [18][19][20] with an additional 11 cases having been published as individual case reports [20][21][22][23][24][25][26][27][28][29].…”

Section: Introductionmentioning

confidence: 99%

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

Dürr¹,

Grahneis²,

Baur‐Melnyk

et al. 2019

BMC Musculoskelet Disord

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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.

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“…Since the initial report of denosumab treatment in two aneurysmal bone cysts of the spine [17], the clinico‐radiologic features of denosumab‐treated aneurysmal bone cysts were described in subsequent case reports and small series [18–34]. Clinically, most patients with denosumab‐treated aneurysmal bone cysts were alive with stable disease; and no denosumab‐related mortality has been reported to date [35].…”

Section: Discussionmentioning

confidence: 99%

“…Both aneurysmal bone cyst and osteoblastoma can be treated by intralesional curettage with bone grafting or cement packing, en bloc resection, or other techniques. Off‐label use of denosumab has been reported in aneurysmal bone cyst [17–35] and osteoblastoma [36,37] in the setting of multiple local recurrences or to minimize the extent of surgery to limit the morbidity of resection.…”

Section: Introductionmentioning

confidence: 99%

Aneurysmal bone cyst and osteoblastoma after neoadjuvant denosumab: histologic spectrum and potential diagnostic pitfalls

Hung

Bredella

Lobmaier

et al. 2022

APMIS

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The use of denosumab to treat giant cell tumors of bone (GCT) and other giant cell‐containing bone tumors has become more common. While the clinicopathologic features of denosumab‐treated giant cell tumors of bone have been well‐illustrated, descriptions of other denosumab‐treated bone tumors are very limited. Surgical pathology files of two institutions and consultation files from two authors were searched for denosumab‐treated aneurysmal bone cysts and denosumab‐treated osteoblastomas. Clinicopathologic features were reviewed and analyzed. We identified four patients with denosumab‐treated bone tumors other than GCT from our surgical pathology and consultation files, including two aneurysmal bone cysts and two osteoblastomas. All were treated with denosumab for 0.5‐7.0 (median 4.5) months. Radiologically, denosumab‐treated tumors showed decreased size with increased ossification and mineralization on CT and heterogeneous intermediate to hypointense signal on MRI. Histologically, denosumab‐treated aneurysmal bone cyst contained thin, elongated, curvilinear, and anastomosing strands of bone with empty lacunae, while denosumab‐treated osteoblastoma showed circumscribed nodules of woven bone lined by small osteoblasts. Denosumab‐treated aneurysmal bone cyst and osteoblastoma showed treatment‐related morphologic changes that can mimic other bone neoplasms. Their recognition requires correlation with the clinical history of denosumab use and radiologic findings.

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Massive aneurysmal bone cyst of the skull base treated with denosumab

Cited by 15 publications

References 25 publications

Denosumab Therapy in the Management of Aneurysmal Bone Cysts: A Comprehensive Literature Review

Denosumab Therapy in the Management of Aneurysmal Bone Cysts: A Comprehensive Literature Review

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

Aneurysmal bone cyst and osteoblastoma after neoadjuvant denosumab: histologic spectrum and potential diagnostic pitfalls

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