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地诺单抗治疗原发性骨巨细胞瘤后的临床、放射学和病理学结果

Clinical, radiological and pathological outcomes following treatment of primary giant cell tumour of bone with Denosumab.

作者信息

Murphy Benjamin, Vodanovich Domagoj, Spelman Tim, Gullifer James, Slavin John, Powell Gerard, Pang Grant, Choong Peter

机构信息

Department of Orthopaedics and The University of Melbourne Department of Surgery, St Vincent's Hospital Melbourne, Melbourne, Victoria, Australia.

Department of Anatomical Pathology, St Vincent's Hospital, Melbourne, Victoria, Australia.

出版信息

ANZ J Surg. 2020 Dec;90(12):2553-2558. doi: 10.1111/ans.16157. Epub 2020 Aug 6.

Abstract

BACKGROUND

Giant cell tumour of bone (GCTOB) is a relatively uncommon, benign, but locally aggressive neoplasm. Denosumab is a fully human monoclonal antibody with inhibitory effects on receptor activator of nuclear factor kappa-B ligand that has shown early promise as a possible treatment adjuvant for GCTB. However, much is still unknown about its current indications, long-term effects, the potential risk for rapid relapse and its involvement in sarcomatous transformation.

METHODS

We analysed the outcomes of 154 patients with GCTOB. We assessed clinical outcomes via local recurrence free-survival, metastatic free-survival and sarcomatous transformation between those treated without Denosumab and those with neo-adjuvant Denosumab. Our radiological and pathological outcomes were assessed through independent specialist reviews.

RESULTS

Four (19.0%) patients of the neo-adjuvant group had local recurrence of disease versus 16 (12.0%) patients in the surgery alone group; this results in a 3.62 times increased likelihood of developing local recurrence (P = 0.030). The median time to local recurrence was shorter for the neo-adjuvant group (421.5 days versus 788.5 days) (P = 0.01). There was no difference between Denosumab and the surgery groups in terms of metastatic disease (P = 0.45). Two patients in our cohort with GCTOB developed sarcomatous transformation, both were treated with Denosumab.

CONCLUSION

Our use of Denosumab tended to be for those patients who had surgically difficult tumours to halt the progression and allow easier resections. Of concern we noted a trend towards increasing recurrence rates with the potential risk for rapid relapse. Furthermore, two cases experienced sarcomatous transformation, which is a growing area of concern within the literature.

摘要

背景

骨巨细胞瘤(GCTOB)是一种相对罕见的良性但具有局部侵袭性的肿瘤。地诺单抗是一种对核因子κB受体活化因子配体具有抑制作用的全人单克隆抗体,已显示出作为GCTB可能的治疗辅助药物的早期前景。然而,关于其当前适应证、长期效果、快速复发的潜在风险及其在肉瘤样转化中的作用,仍有许多未知之处。

方法

我们分析了154例GCTOB患者的治疗结果。我们通过无局部复发生存率、无转移生存率和肉瘤样转化情况,评估了未接受地诺单抗治疗的患者与接受新辅助地诺单抗治疗的患者的临床结局。我们通过独立的专家评审评估了影像学和病理学结果。

结果

新辅助治疗组中有4例(19.0%)患者出现疾病局部复发,而单纯手术组有16例(12.0%)患者出现局部复发;这导致发生局部复发的可能性增加了3.62倍(P = 0.030)。新辅助治疗组局部复发的中位时间较短(421.5天对788.5天)(P = 0.01)。地诺单抗组和手术组在转移疾病方面没有差异(P = 0.45)。我们队列中的2例GCTOB患者发生了肉瘤样转化,均接受了地诺单抗治疗。

结论

我们使用地诺单抗往往是针对那些手术难度较大的肿瘤患者,以阻止疾病进展并使切除更容易。令人担忧的是,我们注意到复发率有上升趋势,存在快速复发的潜在风险。此外,有2例发生了肉瘤样转化,这是文献中一个日益受到关注的领域。

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