Giant cell tumour of the distal radius/ulna: response to pre-operative treatment with short-term denosumab.

Clinical Sarcoma Research Pub Date : 2017-11-30 eCollection Date: 2017-01-01 DOI:10.1186/s13569-017-0085-3
Catherine L McCarthy, Christopher L M H Gibbons, Kevin M Bradley, A Bass Hassan, Henk Giele, Nicholas A Athanasou
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引用次数: 19

Abstract

Background: Treatment of giant cell tumour of bone (GCTB) of the distal radius/ulna poses a surgical challenge, as complex reconstructive surgery may be required. This study evaluates the clinical, radiological and pathological findings in five cases of GCTB of the distal forearm where a 3 month course of denosumab was given prior to surgery.

Methods: Patients with biopsy proven distal forearm GCTB, treated for 3 months with denosumab, followed by salvage surgery (curettage and cementation) were included. Wrist pain and function were assessed using the modified Mayo Wrist Score (MMWS). Plain radiographs, MRI and PET/CT were performed pre-treatment and 2 months after initiation of denosumab therapy. Histological comparison was made between the original biopsy and surgical curettage specimens.

Results: Five patients with an average age of 25 years were included in the study. Improvement in wrist pain and function was seen in all patients with the average MMWS increasing from 30 pre-treatment to 85 at 3 months. Plain radiographs demonstrated marginal sclerosis in all cases with reconstitution of cortical and subarticular bone by 2 months; internal matrix sclerosis and osseous consolidation was more variable. Increased tumour heterogeneity and low signal were observed on T2-weighted MR images. PET/CT revealed a decrease in average SUV from 14.8 pre-treatment to 4.7 at 2 months. Histology showed disappearance of osteoclasts and increased fibro-osseous tissue. Denosumab treatment has the potential to facilitate salvage surgery, thus avoiding bone resection and graft reconstruction. A good outcome was achieved apart from local recurrence in one case. Follow up ranged from 17 to 54 months.

Conclusion: Distal forearm GCTB responds clinically, radiologically and histologically to a short course of pre-operative denosumab therapy, which has the potential to facilitate salvage surgery.

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桡骨/尺骨远端巨细胞瘤:对术前短期地诺单抗治疗的反应
背景:桡骨/尺骨远端骨巨细胞瘤(GCTB)的治疗是一个外科挑战,因为可能需要复杂的重建手术。本研究评估了5例前臂远端GCTB患者的临床、影像学和病理表现,这些患者术前接受了3个月的地诺单抗治疗。方法:纳入活检证实前臂远端GCTB的患者,用denosumab治疗3个月,然后进行挽救性手术(刮除和骨水泥)。采用改良的Mayo腕关节评分(MMWS)评估腕关节疼痛和功能。在治疗前和开始地诺单抗治疗2个月后进行x线平片、MRI和PET/CT检查。将原始活检标本与手术刮除标本进行组织学比较。结果:5例患者被纳入研究,平均年龄25岁。所有患者的腕关节疼痛和功能均有改善,平均MMWS从治疗前的30增加到3个月时的85。x线平片显示所有病例的边缘硬化,2个月时皮质骨和关节下骨重建;内基质硬化和骨实变变化较大。在t2加权MR图像上观察到肿瘤异质性增加和低信号。PET/CT显示平均SUV从治疗前的14.8下降到2个月时的4.7。组织学显示破骨细胞消失,纤维骨组织增多。Denosumab治疗有可能促进补救性手术,从而避免骨切除和移植物重建。除1例局部复发外,治疗效果良好。随访时间为17至54个月。结论:前臂远端GCTB在临床、影像学和组织学上对短疗程的术前denosumab治疗有反应,有可能促进挽救性手术。
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期刊介绍: Clinical Sarcoma Research considers for publication articles related to research on sarcomas, including both soft tissue and bone. The journal publishes original articles and review articles on the diagnosis and treatment of sarcomas along with new insights in sarcoma research, which may be of immediate or future interest for diagnosis and treatment. The journal also considers negative results, especially those from studies on new agents, as it is vital for the medical community to learn whether new agents have been proven effective or ineffective within subtypes of sarcomas. The journal also aims to offer a forum for active discussion on topics of major interest for the sarcoma community, which may be related to both research results and methodological topics.
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