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地诺单抗:寰椎动脉瘤样骨囊肿的一种潜在治疗选择。

Denosumab: a potential treatment option for aneurysmal bone cyst of the atlas.

作者信息

Patel Ravish Shammi, Dhamne Chetan Anil, Gopinathan Anil, Kumar Nishant, Kumar Naresh

机构信息

Department of Orthopaedic Surgery, National University Hospital, University Orthopaedics, Hand and Reconstructive Microsurgery Cluster, 1E Kent Ridge Road, NUHS Tower Block, Level 11, Singapore, 119228, Singapore.

Department of Paediatric Haematology and Oncology, National University Hospital, Singapore, Singapore.

出版信息

Eur Spine J. 2018 Jul;27(Suppl 3):494-500. doi: 10.1007/s00586-018-5528-x. Epub 2018 Feb 22.

DOI:10.1007/s00586-018-5528-x
PMID:29470716
Abstract

PURPOSE

Aneurysmal bone cysts (ABCs) of spine are conventionally treated with en-bloc resection or intralesional excision/curettage and reconstruction or filling of defects with bone cement. For the treatment of upper cervical ABCs, en-bloc resections are often not desirable considering the risk/benefit ratio while the risk of recurrence after intralesional excision is high. Hence, alternative management options are often necessary. We describe our clinical experience with one such treatment alternative-denosumab for the treatment of ABC of Atlas.

METHODS AND RESULTS

We present a case of 16-year-old boy who presented with neck pain and restriction of neck movements. A large lytic lesion with multiple fluid-fluid interfaces involving vertebral arch of atlas was identified on further imaging. There was destruction of right lateral mass and the lesion was found encasing the right vertebral artery. Core needle biopsy confirmed the diagnosis of ABC. With no visible CT response after first session of intra-lesional injection of Calcitonin and Methylprednisolone, the patient was treated with denosumab (120 mg SC once-a-month) for a period of 12 months. His symptoms resolved within 7 months of onset of treatment and serial CT scans over 12-month treatment period showed complete ossification of the lesion. Further there was no evidence of recurrence at 12 months after completion of treatment.

CONCLUSION

Our case report contributes to the accruing evidence on the effectiveness of denosumab for the treatment of spinal ABCs. However, long-term safety, risk of recurrence, optimal duration of treatment and consistency of denosumab are yet to be determined.

摘要

目的

脊柱动脉瘤样骨囊肿(ABCs)传统上采用整块切除或病损内切除/刮除并重建或用骨水泥填充缺损的方法进行治疗。对于上颈椎ABCs的治疗,考虑到风险/获益比,整块切除往往不可取,而病损内切除后复发风险较高。因此,通常需要其他治疗选择。我们描述了一种这样的治疗选择——地诺单抗治疗寰椎ABC的临床经验。

方法与结果

我们报告一例16岁男孩,因颈部疼痛和颈部活动受限就诊。进一步影像学检查发现寰椎椎弓有一个大的溶骨性病变,伴有多个液-液平面。右侧侧块破坏,病变包绕右侧椎动脉。芯针活检确诊为ABC。在病损内注射降钙素和甲基强的松龙第一疗程后未见明显CT反应,该患者接受地诺单抗(皮下注射120mg,每月一次)治疗12个月。治疗开始后7个月内其症状缓解,在12个月的治疗期间,系列CT扫描显示病变完全骨化。此外,治疗完成后12个月没有复发迹象。

结论

我们的病例报告为地诺单抗治疗脊柱ABCs的有效性积累了证据。然而,地诺单抗的长期安全性、复发风险、最佳治疗持续时间和一致性尚待确定。

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Pediatr Neurosurg. 2017;52(4):219-224. doi: 10.1159/000475820. Epub 2017 Jun 13.
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Efficacy and Safety of Selective Arterial Embolization in the Treatment of Aneurysmal Bone Cyst of the Mobile Spine: A Retrospective Observational Study.选择性动脉栓塞治疗活动脊柱骨囊肿的疗效与安全性:一项回顾性观察研究
Spine (Phila Pa 1976). 2017 Aug 1;42(15):1130-1138. doi: 10.1097/BRS.0000000000002017.
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Denosumab: non-surgical treatment option for selective arterial embolization resistant aneurysmal bone cyst of the spine and sacrum. Case report.
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J Spine Surg. 2024 Dec 20;10(4):745-757. doi: 10.21037/jss-24-16. Epub 2024 Dec 3.
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Vertebral Aneurysmal Bone Cyst Mimicking Osteosarcoma: Case Report and Review of the Literature.酷似骨肉瘤的椎体动脉瘤样骨囊肿:病例报告及文献复习
Cureus. 2023 Feb 15;15(2):e35033. doi: 10.7759/cureus.35033. eCollection 2023 Feb.
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An aneurysmal bone cyst ruptured and compressed the spinal cord: a case report.动脉瘤样骨囊肿破裂并压迫脊髓:一例报告
Transl Cancer Res. 2020 Aug;9(8):5008-5014. doi: 10.21037/tcr-20-1124.
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