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巨细胞丰富型骨肉瘤:11 例新病例的配对分析及 56 例患者的文献复习。

Giant cell-rich osteosarcoma: A match pair analysis of 11 new cases and literature review of 56 patients.

机构信息

Department of Orthopedic Surgery, Musculoskeletal Oncology Service, Massachusetts General Hospital, Harvard Medical School, Boston, Massachusetts, USA.

Department of Orthopaedic Surgery, University Medical Center Groningen, University of Groningen, Groningen, The Netherlands.

出版信息

J Surg Oncol. 2023 Oct;128(5):877-890. doi: 10.1002/jso.27368. Epub 2023 Jun 9.

DOI:10.1002/jso.27368
PMID:37292033
Abstract

BACKGROUND

Limited remains known on giant cell-rich osteosarcoma (GCRO) with current studies being case reports or smaller series. This investigation compared GCRO and conventional osteoblastic osteosarcoma (OOS) with regard to demographics and survival.

METHODS

An institutional tumor registry was used to identify 11 patients (six males) treated for GCRO. Mean age was 43 years. Staging showed American Joint Committee on Cancer (AJCC) stages IIA in four and IIB in seven patients. Mean follow-up was 14 years. Study initiatives were: (1) Comparison of demographics between GCRO and 167 OOS from our institutional registry, (2) Differences in survival between GCRO and 33 OOS case controls (based on sex and AJCC stage), as well as 10 OOS using an age-based propensity match, and (3) Summary of all GCRO cases reported in the literature.

RESULTS

(1) Sex (p = 0.53), grading (p = 0.56), AJCC stage (p = 0.42), and chemotherapeutic response rate (p = 0.67) did not differ between groups. Age was significantly increased in GCRO (p = 0.001). (2) Case-control and propensity-matched groups revealed no difference in disease-free survival, local recurrence, and distant disease-free survival at 2 years (p > 0.05). (3) Mean age of 56 patients (50% males) reported in the literature was 26 years. After merging with our 11 cases, the 2-year disease-free survival was 66%.

CONCLUSIONS

GCRO remains a rare disease with high short-term mortality. Although affecting older patients more than conventional osteosarcoma, GCRO should not be viewed as a predictor of survival compared to OOS.

摘要

背景

目前关于巨细胞丰富型骨肉瘤(GCRO)的研究较少,仅有病例报告或小系列研究。本研究比较了 GCRO 和传统成骨型骨肉瘤(OOS)在人口统计学和生存方面的差异。

方法

利用机构肿瘤登记处,我们共确定了 11 例(男 6 例)GCRO 患者。患者平均年龄为 43 岁。分期显示美国癌症联合委员会(AJCC)分期 IIA 期 4 例,IIB 期 7 例。平均随访时间为 14 年。研究举措包括:(1)将 GCRO 与本机构登记处的 167 例 OOS 患者的人口统计学资料进行比较;(2)GCRO 与 33 例 OOS 病例对照(基于性别和 AJCC 分期)以及 10 例基于年龄倾向匹配的 OOS 患者的生存差异;(3)对文献中报道的所有 GCRO 病例进行总结。

结果

(1)性别(p=0.53)、分级(p=0.56)、AJCC 分期(p=0.42)和化疗反应率(p=0.67)两组间无差异。GCRO 患者年龄显著增加(p=0.001)。(2)病例对照和倾向匹配组在 2 年无病生存率、局部复发率和远处无病生存率方面无差异(p>0.05)。(3)文献中报道的 56 例患者(50%为男性)的平均年龄为 26 岁。将这些患者与我们的 11 例患者合并后,2 年无病生存率为 66%。

结论

GCRO 仍是一种罕见疾病,短期死亡率较高。虽然 GCRO 比传统的骨肉瘤更常发生在老年患者,但与 OOS 相比,GCRO 不应被视为生存的预测因素。

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