Maximen Julien, Robin François, Tronchot Alexandre, Rossetti Adrien, Ropars Mickaël, Guggenbuhl Pascal
Department of Orthopedic surgery, Pontchaillou University Hospital, 2, rue Henri Le Guilloux, 35033 Rennes, France.
Inserm, Univ Rennes, INRA, Rheumatology department CHU Rennes, Institut NUMECAN (Nutrition Metabolisms and Cancer), 35000 Rennes, France.
Joint Bone Spine. 2022 Jan;89(1):105260. doi: 10.1016/j.jbspin.2021.105260. Epub 2021 Sep 2.
Aneurysmal bone cyst (ABC) is a benign, locally aggressive tumour that arises predominantly in long bones and spine. Following the encouraging results of denosumab use in Giant Cell Tumors (GCT) and the histological similarities between ABC and GCT, the interest on the role of denosumab in the therapeutic arsenal of the most advanced ABC is growing. The purpose of this literature review is to investigate the current state of knowledge about the use of denosumab in ABCs.
A literature research was conducted through PUBMED, COCHRANE and GOOGLE SCHOLAR using the keywords "aneurysmal bone cyst" AND "denosumab". Seventeen articles were included.
A total of 43 cases were reported in the literature. There were 23 males, 20 females. The mean age was 15,9±8,1 year. Pain relief and neurological improvement were rapid and sustained. Radiological assessment showed ossification and/or volume reduction in 36/39 patients. Eight patients (18,6%) presented a recurrence after or during denosumab therapy of whom 7 were adults. Adverse events occurred in 11 patients, 5 of them were admitted to the intensive care unit due to hypercalcemia.
Denosumab use in non-surgical ABCs has shown a positive impact in pain and neurological symptoms. The oncological outcome remains unclear with a recurrence rate of 18,6% during/after denosumab therapy, mostly in adults. However, regarding the potential clinical benefits, its use might be discussed in the most advanced cases. Further research and clinical trials are mandatory to precise its belonging in the therapeutic arsenal.
骨动脉瘤样囊肿（ABC）是一种良性、具有局部侵袭性的肿瘤，主要发生于长骨和脊柱。鉴于地诺单抗治疗骨巨细胞瘤（GCT）取得了令人鼓舞的结果，且ABC与GCT在组织学上存在相似性，人们对地诺单抗在最晚期ABC治疗手段中的作用的兴趣日益浓厚。本文献综述的目的是研究关于地诺单抗在ABC治疗中应用的当前知识状况。
通过PUBMED、COCHRANE和谷歌学术进行文献检索，使用关键词“骨动脉瘤样囊肿”和“地诺单抗”。共纳入17篇文章。
文献中共报道了43例病例。其中男性23例，女性20例。平均年龄为15.9±8.1岁。疼痛缓解和神经功能改善迅速且持久。影像学评估显示，39例患者中有36例出现骨化和/或体积缩小。8例患者（18.6%）在接受地诺单抗治疗期间或治疗后复发，其中7例为成年人。11例患者出现不良事件，其中5例因高钙血症入住重症监护病房。
在非手术ABC中使用地诺单抗已显示出对疼痛和神经症状有积极影响。肿瘤学结局仍不明确，地诺单抗治疗期间/治疗后复发率为18.6%，主要发生在成年人中。然而，考虑到其潜在的临床益处，在最晚期病例中可能会讨论其应用。需要进一步的研究和临床试验来明确其在治疗手段中的地位。
