骨盆软骨瘤的软骨厚度能否决定恶性风险,术前活检的准确性如何?

Minna K. Laitinen, Michael C. Parry, Guy V. Morris, V. Kurisunkal, Jonathan Stevenson, L. Jeys
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Patients with symptomatic tumors that had cartilage caps ≤ 1.5 cm underwent removal without biopsy (63 patients). A total of 82 patients (63 with caps ≤ 1.5 cm and 19 with caps > 1.5 cm, whose treatment deviated from the routine at the time) had their tumors removed without biopsy. This left 97 patients who underwent biopsy before removal of peripheral cartilage tumors of the pelvis, and this was the group we used to answer research question 1. The thickness of the cartilage cap was recorded from MRI and measuring to the nearest millimeter, with measurements taken perpendicular in the plane that best allowed the greatest measurement. Patient survival rates were assessed using the Kaplan-Meier method with 95% confidence intervals as median observation times to estimate MFS, LRFS, and DSS.\n \n \n \n Of malignant tumors biopsied, in 49% (40 of 82), the biopsy result was recorded as benign (or was considered uncertain regarding malignancy). 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引用次数: 0

摘要

外周骨软骨肿瘤很常见,盆腔肿瘤的治疗具有挑战性,也是一个有争议的话题。有些人认为软骨帽的厚度可能预示着恶性的可能性,但这一推测并没有得到很好的验证。 (1) 术前活检在确定骨盆外周软骨肿瘤是良性还是恶性方面的准确性如何? (2) MRI 确定的软骨帽厚度是否与特定外周软骨肿瘤是恶性的可能性有关? (3) 骨盆外周软骨肉瘤的无局部复发生存率(LRFS)、无转移生存率(MFS)和疾病特异性生存率(DSS)如何,是否与手术切缘有关? 2005年至2022年间,289名患者被诊断为骨盆外周软骨肿瘤(有蒂或无蒂),并在一家三级肉瘤中心(英国伯明翰皇家骨科医院)接受了治疗。这些患者是从一个纵向维护的机构数据库中回顾性识别出来的。无症状、偶然发现且软骨帽≤1.5厘米的肿瘤患者(95人)已出院,剩下194名肿瘤患者要么无症状,要么软骨帽>1.5厘米。对无症状且软骨帽>1.5厘米的肿瘤患者进行为期2年的核磁共振成像随访,如果肿瘤没有生长或外观发生变化,则无需活检即可出院(15名患者)。软骨帽≤1.5厘米的无症状肿瘤患者在不进行活检的情况下进行了切除(63名患者)。共有 82 名患者(63 人的软骨帽≤1.5 厘米,19 人的软骨帽>1.5 厘米,其治疗方法与当时的常规方法不同)在不进行活组织检查的情况下切除了肿瘤。这样就有97名患者在盆腔外周软骨肿瘤切除前接受了活检,这就是我们用来回答研究问题1的一组患者。软骨帽的厚度由核磁共振成像记录并测量到最接近的毫米,测量时要垂直于最适合进行最大测量的平面。采用卡普兰-梅耶法评估患者的存活率,将95%置信区间作为中位观察时间,以估计MFS、LRFS和DSS。 在活检的恶性肿瘤中,49%(82 例中的 40 例)的活检结果被记录为良性(或被认为不确定是否为恶性)。51%的患者(82 例中的 42 例)在活检报告中正确报告了恶性诊断,如果排除恶性程度不确定的活检样本,84%的患者(50 例中的 42 例)的活检确定病变为恶性。只有 33% 的患者(82 例中有 27 例)的活检结果与切除标本记录的最终组织学分级相关。在这82例患者中,有15例活检结果低估了最终的组织学分级。所有良性骨软骨瘤的软骨帽厚度中位数为0.5厘米(范围为0.1至4.0厘米),恶性周围软骨肉瘤的软骨帽厚度中位数为8.0厘米(范围为3.0至19厘米,中位数之差为7.5厘米;P<0.01)。边缘<1毫米的恶性周围肿瘤患者3年的LRFS为49%(95% CI为35%至63%),边缘≥1毫米的恶性周围肿瘤患者3年的LRFS为97%(95% CI为92%至100%)(P<0.01)。1级软骨肉瘤3年的DSS为100%,2级软骨肉瘤3年的DSS为94%(95% CI 86%至100%),3级和5级软骨肉瘤3年和5年的DSS为73%(95% CI 47%至99%),未分化软骨肉瘤3年和5年的DSS为20%(95% CI 0%至55%)(P<0.01)。边缘<1毫米的恶性周围肿瘤患者3年后的DSS为87%(95% CI为78%至96%),边缘≥1毫米的恶性周围肿瘤患者3年后的DSS为100%(P = 0.01)。 软骨帽较薄(< 3 厘米)是良性骨软骨瘤的特征。软骨帽厚度超过 3 厘米后,软骨瘤恶变的可能性就会增加。根据我们的经验,术前活检结果与最终组织学分级或恶性程度的关系并不可靠,只有33%的患者的活检结果是准确的。因此,我们建议对骨盆骨软骨瘤患者进行为期 2 年的观察,如果瘤帽厚度小于 1.5 厘米且无相关疼痛。对于瘤帽厚度在 1.5 至 3 厘米之间的患者,我们建议密切观察 2 年或进行切除,具体取决于主治医生的决定。对于骨盆骨软骨瘤厚度增加或出现疼痛的患者,我们建议最好在软骨帽厚度达到 3 厘米之前进行切除。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Can the Cartilaginous Thickness Determine the Risk of Malignancy in Pelvic Cartilaginous Tumors, and How Accurate is the Preoperative Biopsy of These Tumors?
Peripheral osteochondral tumors are common, and the management of tumors presenting in the pelvis is challenging and a controversial topic. Some have suggested that cartilage cap thickness may indicate malignant potential, but this supposition is not well validated. (1) How accurate is preoperative biopsy in determining whether a peripheral cartilage tumor of the pelvis is benign or malignant? (2) Is the thickness of the cartilage cap as determined by MRI associated with the likelihood that a given peripheral cartilage tumor is malignant? (3) What is local recurrence-free survival (LRFS), metastasis-free survival (MFS), and disease-specific survival (DSS) in peripheral chondrosarcoma of the pelvis and is it associated with surgical margin? Between 2005 and 2022, 289 patients had diagnoses of peripheral cartilage tumors of the pelvis (either pedunculated or sessile) and were treated at one tertiary sarcoma center (the Royal Orthopaedic Hospital, Birmingham, UK). These patients were identified retrospectively from a longitudinally maintained institutional database. Those whose tumors were asymptomatic and discovered incidentally and had cartilage caps ≤ 1.5 cm were discharged (95 patients), leaving 194 patients with tumors that were either symptomatic or had cartilage caps > 1.5 cm. Tumors that were asymptomatic and had a cartilage cap > 1.5 cm were followed with MRIs for 2 years and discharged without biopsy if the tumors did not grow or change in appearance (15 patients). Patients with symptomatic tumors that had cartilage caps ≤ 1.5 cm underwent removal without biopsy (63 patients). A total of 82 patients (63 with caps ≤ 1.5 cm and 19 with caps > 1.5 cm, whose treatment deviated from the routine at the time) had their tumors removed without biopsy. This left 97 patients who underwent biopsy before removal of peripheral cartilage tumors of the pelvis, and this was the group we used to answer research question 1. The thickness of the cartilage cap was recorded from MRI and measuring to the nearest millimeter, with measurements taken perpendicular in the plane that best allowed the greatest measurement. Patient survival rates were assessed using the Kaplan-Meier method with 95% confidence intervals as median observation times to estimate MFS, LRFS, and DSS. Of malignant tumors biopsied, in 49% (40 of 82), the biopsy result was recorded as benign (or was considered uncertain regarding malignancy). A malignant diagnosis was correctly reported in biopsy reports in 51% (42 of 82) of patients, and if biopsy samples with uncertainty regarding malignancy were excluded, the biopsy identified a lesion as being malignant in 84% (42 of 50) of patients. The biopsy results correlated with the final histologic grade as recorded from the resected specimen in only 33% (27 of 82) of patients. Among these 82 patients, 15 biopsies underestimated the final histologic grade. The median cartilage cap thickness for all benign osteochondromas was 0.5 cm (range 0.1 to 4.0 cm), and the median cartilage cap thickness for malignant peripheral chondrosarcomas was 8.0 cm (range 3.0 to 19 cm, difference of medians 7.5 cm; p < 0.01). LRFS was 49% (95% CI 35% to 63%) at 3 years for patients with malignant peripheral tumors with < 1-mm margins, and LRFS was 97% (95% CI 92% to 100%) for patients with malignant peripheral tumors with ≥ 1-mm margins (p < 0.01). DSS was 100% at 3 years for Grade 1 chondrosarcomas, 94% (95% CI 86% to 100%) at 3 years for Grade 2 chondrosarcomas, 73% (95% CI 47% to 99%) at 3 and 5 years for Grade 3 chondrosarcomas, and 20% (95% CI 0% to 55%) at 3 and 5 years for dedifferentiated chondrosarcomas (p < 0.01). DSS was 87% (95% CI 78% to 96%) at 3 years for patients with malignant peripheral tumors with < 1-mm margin, and DSS was 100% at 3 years for patients with malignant peripheral tumors with ≥ 1-mm margins (p = 0.01). A thin cartilage cap (< 3 cm) is characteristic of benign osteochondroma. The likelihood of a cartilage tumor being malignant increases after the cartilage cap thickness exceeds 3 cm. In our experience, preoperative biopsy results were not reliably associated with the final histologic grade or malignancy, being accurate in only 33% of patients. We therefore recommend observation for 2 years for patients with pelvic osteochondromas in which the cap thickness is < 1.5 cm and there is no associated pain. For patients with tumors in which the cap thickness is 1.5 to 3 cm, we recommend either close observation for 2 years or resection, depending on the treating physician’s decision. We recommend excision in patients whose pelvic osteochondromas show an increase in thickness or pain, preferably before the cartilage cap thickness is 3 cm. We propose that surgical resection of peripheral cartilage tumors in which the cartilage cap exceeds 3 cm (aiming for clear margins) is reasonable without preoperative biopsy; the role of preoperative biopsy is less helpful because radiologic measurement of the cartilage cap thickness appears to be accurately associated with malignancy. Biopsy might be helpful in patients in whom there is diagnostic uncertainty or when confirming the necessity of extensive surgical procedures. Future studies should evaluate other preoperative tumor qualities in differentiating malignant peripheral cartilage tumors from benign tumors. Level III, diagnostic study.
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