Recurrent Aneurysmal Bone Cyst of the Distal Fibula Treated with Denosumab and Curettage

Fontenot, Philip B; Jesurajan, Jose; Reed, Damon R.; Binitie, Odion

doi:10.1155/2018/1574343

Cited by 10 publications

(23 citation statements)

References 23 publications

(36 reference statements)

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“…In 2018, Fontenot et al [25] (a case report from the United States) reported a 13-year-old female patient with a history of recurrent ABC involving the distal fibula (long bone). The patient had severe pain and limited activity of his forearm.…”

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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“…However, prolonged treatment with denosumab is associated with osteonecrosis of the jaw, peripheral neuropathy, skin rash, hypophosphatemia, and atypical femoral fracture [ 15 ]. Furthermore, the use of denosumab is contraindicated in pregnancy [ 16 ]. Yet, secukinumab has much milder adverse effects, including nasopharyngitis, headache, nausea, diarrhea, and pyrexia, and is not contraindicated for pregnant patients [ 17 , 18 ].…”

Section: Discussionmentioning

confidence: 99%

Anti-IL17 antibody Secukinumab therapy is associated with ossification in giant cell tumor of bone: a case report of pathologic similarities and therapeutic potential similar to Denosumab

Chandler

Bartelstein

Fujiwara

et al. 2021

BMC Musculoskelet Disord

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Background Giant cell tumor of bone is a benign, locally aggressive neoplasm. Surgical resection is the preferred treatment method. However, for cases in which resection poses an increased risk to the patient, denosumab (anti-RANKL monoclonal antibody) is considered. Secukinumab is an anti-IL-17 antibody that is used in psoriatic arthritis to reduce bone resorption and articular damage. Case presentation One case of giant cell tumor of bone (GCTB) in a patient treated with secukinumab for psoriatic arthritis demonstrated findings significant for intra-lesional calcifications. Histologic examination showed ossification, new bone formation, and remodeling. A paucity of osteoclast type giant cells was noted. Real-time quantitative polymerase-chain-reaction (qRT-PCR) analysis revealed decreased osteoclast function compared to treatment-naive GCTB. Conclusions Secukinumab may play a role in bone remodeling for GCTB. Radiologists, surgeons, and pathologists should be aware of this interaction, which can cause lesional ossification. Further research is required to define the therapeutic potential of this drug for GCTB and osteolytic disease.

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Recurrent Aneurysmal Bone Cyst of the Distal Fibula Treated with Denosumab and Curettage

Cited by 10 publications

References 23 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

Anti-IL17 antibody Secukinumab therapy is associated with ossification in giant cell tumor of bone: a case report of pathologic similarities and therapeutic potential similar to Denosumab

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