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资源有限环境下儿童和青少年骨肉瘤的管理:改善治疗结果的质量改进考虑因素。

The management of osteosarcoma in children and adolescents in a resource-limited setting: quality improvement considerations to improve treatment outcomes.

机构信息

Department of Paediatrics and Child Health, Lira University, P.O. Box 1035, Lira, Uganda.

Department of Paediatric Oncology, Uganda Cancer Institute, P.O. Box 3935, Kampala, Uganda.

出版信息

BMC Cancer. 2024 Aug 28;24(1):1061. doi: 10.1186/s12885-024-12786-6.

Abstract

BACKGROUND

The survival rates for children and adolescents with osteosarcoma in low-income countries are poor. Insufficient data regarding the challenges of managing osteosarcoma in resource-limited settings has been published. We evaluated the treatment of osteosarcoma in children and adolescents with the aim of improving the health system and management outcomes.

METHODS

We sourced data on children under 18 years treated for osteosarcoma at the Uganda Cancer Institute between January 2016 and December 2020. Descriptive statistics and Kaplan-Meier survival analysis were used.

RESULTS

Seventy-four osteosarcoma cases were identified, with a median age of 13 years (IQR 9.8-15). Referrals were made after a median of 28 days (range 1-147). Before appropriate referral, more than a quarter (26%) had undergone invasive procedures that could compromise tumour integrity and outcome. Half (50%) of the patients had metastatic disease at diagnosis, primarily to the lungs (n = 43; 92%). Only 14 (33%) patients received neoadjuvant chemotherapy. Forty-three (58.1%) patients underwent limb amputation surgery, including 25 localized tumours and 18 patients with distant metastatic disease. No metastatectomies were performed. Adjuvant chemotherapy was delayed for longer than 21 days in 26 (61%) patients. No pathology reports described the status of resection margins or the degree of chemotherapy-induced necrosis. Twenty-six (35%) patients abandoned treatment, mainly due to pending radical surgery (n = 18/26; 69%). Only 18% (n = 13) were still alive; 46% (n = 34) had died; and 37% (n = 27) had an unknown status. The median overall survival was 1.1 years, and was significantly negatively affected by disease metastasis, timing of adjuvant therapy, and treatment abandonment.

CONCLUSIONS

Osteosarcoma outcomes for children and adolescents at the Uganda Cancer Institute are extremely poor. The quality of care can be improved by addressing delayed referrals, high rates of prior manipulative therapy, metastatic disease, treatment abandonment, surgical challenges, and delayed resumption of adjuvant chemotherapy.

摘要

背景

在低收入国家,儿童和青少年骨肉瘤的生存率较差。关于资源有限环境下骨肉瘤管理挑战的数据发表不足。我们评估了儿童和青少年骨肉瘤的治疗,旨在改善卫生系统和管理结果。

方法

我们从乌干达癌症研究所 2016 年 1 月至 2020 年 12 月治疗的 18 岁以下骨肉瘤患儿中获取数据。采用描述性统计和 Kaplan-Meier 生存分析。

结果

共确定了 74 例骨肉瘤病例,中位年龄为 13 岁(IQR 9.8-15)。中位转诊时间为 28 天(范围 1-147)。在适当转诊之前,超过四分之一(26%)的患者接受了可能影响肿瘤完整性和结果的侵入性操作。一半(50%)的患者在诊断时已经有转移病灶,主要是肺部(n=43;92%)。仅有 14(33%)例患者接受新辅助化疗。43(58.1%)例患者接受了肢体截肢手术,包括 25 例局限性肿瘤和 18 例远处转移疾病患者。未行转移切除术。26(61%)例患者辅助化疗延迟超过 21 天。没有病理报告描述了切缘状态或化疗诱导的坏死程度。26(35%)例患者放弃治疗,主要是由于等待根治性手术(n=18/26;69%)。仅有 18%(n=13)例患者仍然存活;46%(n=34)例患者死亡;37%(n=27)例患者状态未知。中位总生存期为 1.1 年,疾病转移、辅助治疗时机和治疗放弃显著负性影响生存。

结论

乌干达癌症研究所儿童和青少年骨肉瘤的治疗结果极差。通过解决延迟转诊、高比例的前期有创治疗、转移疾病、治疗放弃、手术挑战和辅助化疗延迟恢复等问题,可以提高治疗质量。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c3f9/11351074/b87a345a2618/12885_2024_12786_Fig1_HTML.jpg

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