Response to Denosumab in 2 Children With Recurrent Giant Cell Tumor of the Bone With Pulmonary Metastasis

Reddy, Kiranmye; Ramirez, Lorimar; Kukreja, Kamlesh; Venkatramani, Rajkumar

doi:10.1097/mph.0000000000001654

Cited by 5 publications

(4 citation statements)

References 31 publications

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“…A critical review of these published studies identified 60 studies that met the inclusion criteria for the present meta-analysis ( Table 1 ). 11 , 12 , 44 , 60 - 116 The www.clinicaltrials.gov website showed 7 ongoing studies of denosumab in the treatment of GCTB. The majority of the studies examined for this meta-analysis were case reports, case series studies, and nonrandomized, uncontrolled, open-label phase II studies ( Figure 2 ).…”

Section: Resultsmentioning

confidence: 99%

“…44 An important point to note is that 9 cases of malignant transformation of GCTB during treatment with denosumab, without prior radiation treatment, have been reported in the literature. 118 Inhibition of RANKL has been reported to Chawla et al 61 (2019) Palmerini et al 86 (2017) X X 0% 20% 40% 60% 80% 100%…”

Section: Proportion Of Participants In Each Study Design Typementioning

confidence: 93%

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Denosumab for Effective Tumor Size Reduction in Patients With Giant Cell Tumors of the Bone: A Systematic Review and Meta-Analysis

Yayan

2020

Cancer Control

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Background: Denosumab is a human monoclonal antibody that has been used successfully in the treatment of giant cell tumors of bone. These tumors are rare and, in principle, benign, but they are highly aggressive, locally advanced, osteolytic bone tumors that can metastasize to the lungs. Denosumab is an effective treatment when these tumors cannot be surgically removed or when surgical resection is likely to lead to severe morbidity (eg, loss of limbs or joints). The aim of this systematic review and meta-analysis was to investigate patients with giant cell tumors of bone who experienced tumor progression during treatment with denosumab and to compare them with patients who experienced reduction of their giant cell tumors of bone during treatment with denosumab. Methods: Embase, Cochrane Library, and MEDLINE/PubMed databases were searched for trials submitted by January 7, 2020, that reported the efficacy and safety of denosumab in patients with giant cell tumors of bone. Results: Sixty studies were reviewed, involving a total of 1074 patients who had giant cell tumors of bone and were treated with denosumab. Of the 60 studies, 58% of the patients were from case series studies, 39% from open-label phase II studies, and 3% from case reports. The response rate for denosumab as a treatment for giant cell tumors of bone was 97.5%, with statistical significance ( P < .0001). Pain in the limbs was statistically the most common adverse event for denosumab treatment in case series studies ( P < .0001). No treatment-related deaths occurred in the reviewed studies. Conclusion: Cumulative evidence supports the addition of surgery to optimal medical therapy with denosumab to reduce tumor size, clinical symptoms, and mortality among patients with giant cell tumors of bone.

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Section: Resultsmentioning

confidence: 99%

Section: Proportion Of Participants In Each Study Design Typementioning

confidence: 93%

Denosumab for Effective Tumor Size Reduction in Patients With Giant Cell Tumors of the Bone: A Systematic Review and Meta-Analysis

Yayan

2020

Cancer Control

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“…In 2021, Reddy et al reported their experience with denosumab in 2 children with GCTB and pulmonary metastasis, achieving satisfactory results (33). During the denosumab treatment period, a reduction in the size of the primary lesions and lung metastatic lesions was observed, and pain was relieved.…”

Section: Giant Cell Tumor Of Bone (Gctb)mentioning

confidence: 99%

Denosumab in pediatric bone disorders and the role of RANKL blockade: a narrative review

Wang¹,

Tang²,

Shi³

et al. 2023

Transl Pediatr

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Background and Objective: Denosumab is a valuable and safe therapy for skeletal disorders in adults and has received regulatory approval to treat osteoporosis and bone metastases. However, denosumab is not licensed for pediatric use due to a lack of high-quality prospective research on children. This study aimed to describe and discuss the benefits and disadvantages of denosumab in treating bone diseases in children and to summarize the current understanding of the role of denosumab therapy in children.Methods: A narrative review was conducted using the literature retrieved from the PubMed, Embase, and Cochrane Library databases.

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“…Although rare below the age of 18, Reddy et al reported two children with recurrent GCTB with pulmonary metastases [63]. Both were treated with denosumab and imaging demonstrated resolved, decreased or residual but stable pulmonary nodules during follow-up.…”

Section: Metastatic Diseasementioning

confidence: 99%

Updated concepts in treatment of giant cell tumor of bone

Heijden¹,

Lipplaa²,

Langevelde³

et al. 2022

Current Opinion in Oncology

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Purpose of reviewGiant cell tumors of bone (GCTB) are intermediate, locally aggressive primary bone tumors. For conventional GCTB, surgery remains treatment of choice. For advanced GCTB, a more important role came into play for systemic therapy including denosumab and bisphosphonates over the last decade.Recent findingsIn diagnostics, focus has been on H3F3A (G34) driver mutations present in GCTB. The most frequent mutation (G34W) can be detected using immunohistochemistry and is highly specific in differentiating GCTB from other giant cell containing tumors. PD-L1 expression can be used as biological marker to predict higher recurrence risks in GCTB patients.The use of bisphosphonate-loaded bone cement is under investigation in a randomized controlled trial. A new technique consisting of percutaneous microwave ablation and bisphosphonate-loaded polymethylmethacrylate cementoplasty was proposed for unresectable (pelvic) GCTB.Increased experience with use of denosumab raised concern on elevated recurrence rates. However, conclusions of meta-analyses should be interpreted with risk of indication bias in mind. Several small studies are published with short-course denosumab (varying from 3 to 6 doses). One small trial directly compared denosumab and zoledronic acid, with no statistical differences in radiological and clinical outcome, and nonsignificantly higher recurrence rate after denosumab. As bisphosphonates directly target neoplastic stromal cells in GCTB, larger directly comparative trials are still warranted.SummaryNeoadjuvant denosumab is highly effective for advanced GCTB, and a short-course is advised to facilitate surgery, whereas increased recurrence rates remain of concern. Randomized controlled trials are conducted on bisphosphonate-loaded bone cement and on optimal dose and duration of neoadjuvant denosumab. PD-L1 could be a potential new therapy target in GCTB.

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Response to Denosumab in 2 Children With Recurrent Giant Cell Tumor of the Bone With Pulmonary Metastasis

Cited by 5 publications

References 31 publications

Denosumab for Effective Tumor Size Reduction in Patients With Giant Cell Tumors of the Bone: A Systematic Review and Meta-Analysis

Denosumab for Effective Tumor Size Reduction in Patients With Giant Cell Tumors of the Bone: A Systematic Review and Meta-Analysis

Denosumab in pediatric bone disorders and the role of RANKL blockade: a narrative review

Updated concepts in treatment of giant cell tumor of bone

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