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多中心性巨细胞瘤的系统评价及三例长期随访病例报告

A systematic review of multicentric giant cell tumour with the presentation of three cases at long-term follow-up.

作者信息

Trovarelli Giulia, Pala Elisa, Angelini Andrea, Ruggieri Pietro

机构信息

Department of Orthopedics and Orthopedic Oncology, University of Padova, Padova, Italy.

出版信息

Bone Joint J. 2022 Dec;104-B(12):1352-1361. doi: 10.1302/0301-620X.104B12.BJJ-2022-0401.R1.

DOI:10.1302/0301-620X.104B12.BJJ-2022-0401.R1
PMID:36453049
Abstract

AIMS

We performed a systematic literature review to define features of patients, treatment, and biological behaviour of multicentric giant cell tumour (GCT) of bone.

METHODS

The search terms used in combination were "multicentric", "giant cell tumour", and "bone". Exclusion criteria were: reports lacking data, with only an abstract; papers not reporting data on multicentric GCT; and papers on multicentric GCT associated with other diseases. Additionally, we report three patients treated under our care.

RESULTS

A total of 52 papers reporting on 104 patients were included in the analysis, with our addition of three patients. Multicentric GCT affected predominantly young people at a mean age of 22 years (10 to 62), manifesting commonly as metachronous tumours. The mean interval between the first and subsequent lesions was seven years (six months to 27 years). Synchronous lesions were observed in one-third of the patients. Surgery was curettage in 63% of cases (163 lesions); resections or amputation were less frequent. Systemic treatments were used in 10% (n = 14) of patients. Local recurrence and distant metastases were common.

CONCLUSION

Multicentric GCT is rare, biologically aggressive, and its course is unpredictable. Patients with GCT should be followed indefinitely, and referred promptly if new symptoms, particularly pain, emerge. Denosumab can have an important role in the treatment.Cite this article:  2022;104-B(12):1352-1361.

摘要

目的

我们进行了一项系统的文献综述,以明确多中心骨巨细胞瘤(GCT)患者的特征、治疗方法及生物学行为。

方法

联合使用的检索词为“多中心”“巨细胞瘤”和“骨”。排除标准为:缺乏数据的报告(仅摘要);未报告多中心GCT数据的论文;以及关于与其他疾病相关的多中心GCT的论文。此外,我们报告了在我们治疗下的3例患者。

结果

分析纳入了共52篇报告104例患者的论文,外加我们的3例患者。多中心GCT主要影响年轻人,平均年龄22岁(10至62岁),通常表现为异时性肿瘤。首个病灶与后续病灶之间的平均间隔为7年(6个月至27年)。三分之一的患者观察到同步病灶。63%的病例(163个病灶)采用刮除术;切除术或截肢术较少见。10%(n = 14)的患者使用了全身治疗。局部复发和远处转移很常见。

结论

多中心GCT罕见,具有生物学侵袭性,其病程不可预测。GCT患者应长期随访,若出现新症状,尤其是疼痛,应及时转诊。地诺单抗在治疗中可发挥重要作用。引用本文:2022;104-B(12):1352-1361。

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