Development of high-grade osteosarcoma in a patient with recurrent giant cell tumor of the ischium while receiving treatment with denosumab

Tsukamoto, Shinji; Righi, Alberto; Vanel, D.; Honoki, Kanya; Donati, Davide María; Errani, Costantino

doi:10.1093/jjco/hyx112

Cited by 50 publications

(37 citation statements)

References 21 publications

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“…Malignant transformation of giant cell tumour of the bone is an extremely rare disease that has been defined mainly by pathological features. A review of literature shows cases of giant cell tumour that have undergone malignant transformation to osteosarcoma are being reported 9–11. Our case would add to these growing body of cases.…”

Section: Discussionmentioning

confidence: 65%

Comprehensive molecular imaging of malignant transformation of giant cell tumour of bone reveals diverse disease biology

Kairemo

Wang

Subbiah³

2019

BMJ Case Rep

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Malignant transformation of giant cell tumour of the bone is extremely rare. In addition, bone transformation in giant cell tumour may occur in different phases. With conventional X-rays, CT scans or MRIs, it may be challenging to distinguish among different phases of bone transformation, normal bone, soft tissue disease and bone disease (benign vs malignant lesions) and changes in multiple organs such as lung, liver and lymph nodes unless every lesion is biopsied, which is not practical. Molecular imaging with different isotopes (Tc-99m phosphonate, 2-deoxy-2-(18F)fluoro-d-glucose and sodium fluoride-18) may help to better characterise the disease. We hypothesised that molecular imaging could offer qualitative and quantitative characterisation of all stages of bone formation, destruction, reactivity or neoplasia in a patient with giant cell tumour of the bone, and we present the first case of molecular imaging where bone formation was seen in multiple soft tissues, such as lungs, muscles, lymph nodes and liver.

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Section: Discussionmentioning

confidence: 65%

Comprehensive molecular imaging of malignant transformation of giant cell tumour of bone reveals diverse disease biology

Kairemo

Wang

Subbiah³

2019

BMJ Case Rep

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“…Seven months after surgical excision, the patient developed an osteosarcoma in the same site, along with pulmonary metastasis. Tsukamoto et al ( 59 ) demonstrated a 25-year-old woman with recurrent GCTB in her left ischium developed a high grade osteosarcoma. She received denosumab therapy for 6 months.…”

Section: Denosumab May Arouse the Malignant Transformation Of Gctbmentioning

confidence: 99%

“…The potential mechanisms of sarcomatous transformations of GCTB following denosumab therapy are probably associated with its actions against RANKL ( 59 ). Although the exact molecular basis is poorly defined, three possible hypotheses are proposed.…”

Section: Denosumab May Arouse the Malignant Transformation Of Gctbmentioning

confidence: 99%

Denosumab in Giant Cell Tumor of Bone: Current Status and Pitfalls

Gao

et al. 2020

Front. Oncol.

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Denosumab is a monoclonal antibody against RANK ligand for treatment of giant cell tumor of bone (GCTB). Clinical trials and case series have demonstrated that denosumab is relevant to beneficial tumor response and surgical down-staging in patients of GCTB. However, these trials or case series have limitations with a short follow-up. Recent increasing studies revealed that denosumab probably increased the local recurrence risk in patients treated with curettage. This may be caused by the thicken bone margin of tumor that trapped tumor cells from curettage. The direct bone formation by tumor cells in the margin after denosumab treatment also contributed to the local recurrence. in vitro studies showed denosumab resulted in a cytostatic instead of a true cytotoxic response on neoplastic stromal cells. More importantly, denosumab-treated GCTB exhibited morphologic overlap with malignancy, and a growing number of patients of malignant transformation of GCTB during denosumab treatment have been reported. The optimal duration, long term safety, maintenance dose, and optimum indications remain to be elucidated. With these concerns in mind, this review warns that the denosumab therapy of GCTB should be applied with caution.

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“…The efficacy of denosumab has been demonstrated in patients with unresectable or recurrent GCT of bone, according to the NCCN guideline and previous studies [43]. However, denosumab did not show any effect on reducing the recurrence rate [44], and complications such as sarcomatous transformation should be considered [45]. Fourthly, our findings are only based on the respective data from our institution, this implies a study selection bias that must be acknowledged, and which might only reflect surgeon or patient preference.…”

Section: Comparison Of Complications With Henderson Classificationmentioning

confidence: 99%

The functional outcomes and complications of different reconstruction methods for Giant cell tumor of the distal radius: comparison of Osteoarticular allograft and three-dimensional-printed prosthesis

Wang

et al. 2020

BMC Musculoskelet Disord

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Background: En bloc excision has been increasingly used for the management of giant cell tumors (GCTs) in the distal radius. An osteoarticular allograft has been used extensively for decades, and custom-made prosthesis reconstruction has been more recently applied. We aimed to compare the clinical outcomes of the two procedures. Methods: We retrospectively analyzed 30 patients with Campanacci III or recurrent GCTs of the distal radius for follow-up at a mean of 33.2 months. In total, 15 underwent osteoarticular allograft reconstruction (allograft group) and 15 received cementless three-dimensional (3D)-printed prosthesis reconstruction (prosthesis group) between March 18, 2013, and May 20, 2018. All patients underwent by clinical and radiological examinations, including pre-and postoperative active range of motion (ROM) of the wrist, VAS score, grip strength, degenerative change of wrist, Mayo wrist score and Musculoskeletal Tumor Society (MSTS) score. Complications were evaluated using the Henderson classification. Results: Both groups showed significantly increased ROM, grip strength, Mayo score and MSTS score postoperatively. Furthermore, the extension, flexion, MSTS, and Mayo score were significantly higher in the prosthesis group. There was no significant difference in grip strength and VAS between the groups. In allograft group, one patient had a late infection one had resorption of allograft without allograft bone fracture. and four had wrist subluxation. All patients had degenerative changes (mean 9 months). In the prosthesis group, three patients developed wrist subluxation, three had separation of the distal radioulnar joint, and none of the patients developed wrist degeneration. Conclusions: Our study compared the objective functional outcomes and complications of two reconstructive methods for Campanacci III or recurrent GCT in the distal radius. 3D-printed prosthesis replacement can partially preserve wrist function better than allograft reconstruction in the short-term. During the design of 3D-printed prosthesis, preoperative morphological assessment of the affected proximal row carpal is helpful to control postoperative dislocation. After allograft reconstruction, wrist degeneration, which has been demonstrated in all patients, severely influence their wrist function. Therefore, compared to allograft reconstruction, 3D-printed prosthesis reconstruction has irreplaceable advantages at early-stage application, especially in wrist function, however, further studied with a larger number of cases and longer follow-up.

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Development of high-grade osteosarcoma in a patient with recurrent giant cell tumor of the ischium while receiving treatment with denosumab

Cited by 50 publications

References 21 publications

Comprehensive molecular imaging of malignant transformation of giant cell tumour of bone reveals diverse disease biology

Comprehensive molecular imaging of malignant transformation of giant cell tumour of bone reveals diverse disease biology

Denosumab in Giant Cell Tumor of Bone: Current Status and Pitfalls

The functional outcomes and complications of different reconstruction methods for Giant cell tumor of the distal radius: comparison of Osteoarticular allograft and three-dimensional-printed prosthesis

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