Targeting receptor-activator of nuclear kappaB ligand in aneurysmal bone cysts: verification of target and therapeutic response

Pelle, Dominic W.; Ringler, Jonathan W.; Peacock, Jacqueline D.; Kampfschulte, Kevin; Scholten, Donald J.; Davis, Mary M.; Mitchell, Deanna; Steensma, Matthew R.

doi:10.1016/j.trsl.2014.03.005

Cited by 70 publications

(93 citation statements)

References 68 publications

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“…Furthermore, as described, the agents have a multifactorial action, which is related to the inhibition of MMP production, angiogenesis, and osteoclast function, as well as to the induction of osteoclast apoptosis and promotion of osteoblast regeneration. These findings are consistent with the description of ABC as a clonal tumor expressing high levels of MMPs and vascular endothelial growth factor, which has a high osteoclast activity and presents a blockage in osteoblast maturation [28][29][30][31][32]38].…”

Section: Nosupporting

confidence: 91%

Primary Aneurysmal Bone Cyst of Long Bones Treated with a Single Dose of Calcitonin and Methylprednisolone Percutaneous Intralesional Injection: A Case Series and Literature Review

Oliveira¹,

Meohas²,

Carvalho³

et al. 2017

J Orthop Oncol

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Aneurysmal bone cyst is a rare benign bone tumor usually treated by surgery, which may in turn result in poor functional outcome and recurrence. This study aims to report a series of cases of primary ABC treated with a single dose of calcitonin and methylprednisolone injection. Six patients treated with a single-dose combination of calcitonin (200 UI) and methylprednisolone (120 mg) at a single institution from January 2011 to December 2013 were clinically and radiologically evaluated. After the follow-up period, all patients presented cyst healing. The mean time to healing was 2.8 months. The final mean overall functional score rating was 89.0%. We conclude that intralesional calcitonin and methylprednisolone injection results in aneurysmal bone cyst healing with good functional outcomes and thus can be an effective treatment method.

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Section: Nosupporting

confidence: 91%

Primary Aneurysmal Bone Cyst of Long Bones Treated with a Single Dose of Calcitonin and Methylprednisolone Percutaneous Intralesional Injection: A Case Series and Literature Review

Oliveira¹,

Meohas²,

Carvalho³

et al. 2017

J Orthop Oncol

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“…By reducing tumor size, denosumab has been found to reduce potential morbidity of surgical interventions. Similarly, Skubitz et al and Pelle et al observed pain relief, good drug tolerance, and radiographic evidence healing in patients with sacral ABCs [62,63]. Tumor regression, pain reduction, and resolution of neurologic symptoms were also reported in a separate case series of two spinal ABCs following denosumab treatment [64•].…”

Section: Rankl Inhibition and The Role Of Denosumabmentioning

confidence: 86%

Current management of aneurysmal bone cysts

Park

Yang

Sheppard

et al. 2016

Curr Rev Musculoskelet Med

188

259

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Aneurysmal bone cysts (ABCs) are benign bone lesions arising predominantly in the pediatric population that can cause local pain, swelling, and pathologic fracture. Primary lesions, which constitute roughly two thirds of all ABCs, are thought to be neoplastic in nature, with one third of ABCs arising secondary to other tumors. Diagnosis is made with various imaging modalities, which exhibit characteristic features such as Bfluid-fluid levels,^although biopsy is critical, as telangiectatic osteosarcoma cannot be excluded based on imaging alone. Currently, the standard of care and most widely employed treatment is intralesional curettage. However, tumor recurrence with curettage alone is common and has driven some to propose a multitude of adjuvants with varying efficacy and risk profiles. Historically, therapies such as en bloc resection or radiation therapy were utilized as an alternative to decrease the recurrence rate, but these therapies imposed high morbidity. As a result, modern techniques now seek to simultaneously reduce morbidity and recurrence, the pursuit of which has produced preliminary study into minimally invasive percutaneous treatments and medical management.

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“…It was shown that the clinical effect of denosumab observed in a considerable part of the patients was accompanied by more than 90% decrease of the number of tumor-associated giant cells and a decrease of the number of stromal cells [18]. RANK/RANKL interaction inhibitors are also considered as new approaches to the treatment of chordoma [19] and some benign bone lesions [10]. And an effect of denosumab combination with protein kinase inhibitor sorafenib in a patient with unresectable chemorefractory osteoblastic osteosarcoma was reported [20].…”

Section: Discussionmentioning

confidence: 99%

“…High expression of factors responsible for osteoclastogenesis (RANKL, M-CSF) by osteosarcoma cell lines MG63 and Saos-2 was demonstrated in in vitro experiments [5,6], and a clinical study has demonstrated a significant relationship between high RANKL expression in the tumor and a poor response of osteosarcoma patients to neoadjuvant chemotherapy: high RANKL expression levels were associated with low 5-years' overall survival [7]. Increased expression of RANK/RANKL/OPG system components was also found in chondrosarcoma [8], Ewing sarcoma [9], and some bone tissue benign tumor-like lesions [10].…”

Section: Introductionmentioning

confidence: 97%

Receptor Activator of Nuclear Transcription Factor Nf-b (Rank), Its Ligand (Rankl) and Natural Inhibitor Osteoprotegerin (Opg) in Blood Serum of Primary Bone Tumor Patients: Association With Clinicopathological Features and Inflammatory Cytokines Levels

Kushlinskii¹,

Es²,

Timofeev³

et al. 2017

RAD Conference Proceedings

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Abstract. RANK/RANKL/OPG system (the key regulator of bone homeostasis) component levels were measured in blood serum of 199 patients with primary bone tumors and tumor-like lesions: 121 with bone sarcomas (53 osteosarcoma, 46 chondrosarcoma, 12 chordoma, 8 Ewing sarcoma), 32 with borderline giant cell bone tumor (GCBT), 46 with benign bone neoplasms; 131 persons comprised the control group. OPG, sRANKL, sRANK, IL-6, 8, 16 serum levels were measured by standard ELISA kits. Giant-cell bone tumor (GCBT) manifesting high osteoclastogenic and osteolytic activities was characterized by high serum content of all three components studied and the highest sRANKL/OPG ratio. The group of patients with various benign bone tumors and tumor-like lesions displayed similar to GCBT, but lower indices. Malignant bone tumor patients could be divided into 2 subgroups with opposite characteristics: osteosarcoma and Ewing sarcoma patients demonstrated low sRANK and high sRANKL levels, while chondrosarcoma and chordoma patients, on the contrary -high sRANK and low sRANKL levels. The highest IL-6 levels were revealed in GCBT patients, while serum IL-8 and IL-16 did not differ between groups. Thus, disturbances in osteolysis activators and inhibitors balance in blood serum of primary bone tumor patients were revealed. Their extent depended on neoplasm character (malignant, borderline, or benign) and histological structure of malignant sarcomas. Most prominent changes were found in GCBT characterized by active bone destruction and an accepted target of anti-RANKL antibody denosumab treatment. Hence, the proteins studied can be regarded as promising serologic markers and therapeutic targets in this rare disease.

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Targeting receptor-activator of nuclear kappaB ligand in aneurysmal bone cysts: verification of target and therapeutic response

Cited by 70 publications

References 68 publications

Primary Aneurysmal Bone Cyst of Long Bones Treated with a Single Dose of Calcitonin and Methylprednisolone Percutaneous Intralesional Injection: A Case Series and Literature Review

Primary Aneurysmal Bone Cyst of Long Bones Treated with a Single Dose of Calcitonin and Methylprednisolone Percutaneous Intralesional Injection: A Case Series and Literature Review

Current management of aneurysmal bone cysts

Receptor Activator of Nuclear Transcription Factor Nf-b (Rank), Its Ligand (Rankl) and Natural Inhibitor Osteoprotegerin (Opg) in Blood Serum of Primary Bone Tumor Patients: Association With Clinicopathological Features and Inflammatory Cytokines Levels

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