Kell David, Yang Daniel, Lee Juliana, Orellana Kevin, Wetzel Sarah, Arkader Alexandre
Department of Orthopaedics, The Children's Hospital of Philadelphia, Philadelphia, PA, USA.
Clin Orthop Relat Res. 2025 Apr 1;483(4):748-758. doi: 10.1097/CORR.0000000000003299. Epub 2024 Oct 30.
The published reports examining socioeconomic factors and their relationship to osteosarcoma presentation and treatment suggest an association between lower socioeconomic status and a worse response to chemotherapy and lower survivorship. However, the driving factors behind these disparities are unclear. The Child Opportunity Index was developed by diversitydatakids.org ( https://www.diversitydatakids.org/ ) in 2014 to cumulatively quantify social determinants of health in an index specifically tailored toward a pediatric population and organized by census tract. The Childhood Opportunity Index can be used to explore the relationship between a patient's socioeconomic background and disparities in osteosarcoma presentation, treatment, and outcomes.
QUESTION/PURPOSES: Are differences in a child's Childhood Opportunity Index score associated with differences in (1) time from symptom onset to first office visit for osteosarcoma, (2) timing of chemotherapy or timing and type of surgical resection, or (3) initial disease severity, development of metastatic disease, or overall survival?
A retrospective therapeutic study was conducted using data drawn from the institutional records of a large pediatric tertiary cancer center located in the Mid-Atlantic region of the United States from the years 2006 to 2022. Our main site is in an urban setting, with ample access to public transit. Patients were excluded from analysis if they were seeking a second opinion or our institution was not the main point of orthopaedic care (20% [45 of 223]). Of the remaining patients, those with incomplete electronic medical records (24% [43 of 178]), resided in an international country (5% [9 of 178]), presented after relapse (4% [8 of 178]), or lacked 2 years of follow-up at our institution (3% [5 of 178]) were excluded as well. A total of 113 pediatric patients (children younger than 18 years) met the inclusion criteria. The Child Opportunity Index is a composite index derived from three domains (education, health and environment, and social and economic) and 29 indicators within the domains that serve to capture the cumulative effect of disparities on child well-being. National Childhood Opportunity scores were collected and scored from 1 to 100. Each score represents an equal proportion of the US population of children 18 years of age or younger. A higher number indicates higher levels of socioeconomic opportunity. The overall Childhood Opportunity Index score was then broken down into three groups representative of the child's relative socioeconomic opportunity: lowest tertile for scores < 34, middle tertile for scores between 34 and 66, and highest tertile for scores > 66. Means, ranges, medians, IQRs, and percentages were used to describe the study sample. Data analysis was conducted across the three groups (lowest tertile, middle, and highest), assessing differences in time to presentation, treatment variations, disease severity, and overall survivorship. Chi-square and Fisher exact tests were applied to compare categorical variables. Mann-Whitney U tests compared continuous data. Kaplan-Meier survival analysis, stratified by Childhood Opportunity Index tertile, was performed for a 5-year period to evaluate the development of metastatic disease and overall survivorship. A log-rank test was applied to evaluate statistical significance. Due to the small sample size, we were unable to control potential confounders such as race and insurance. However, the three domains (education, health and environment, and social and economic) encapsulated by the Childhood Opportunity Index data indirectly account for disparities related to race and insurance status.
There was no association between lower levels of socioeconomic opportunity, as expressed by the lack of difference between the Childhood Opportunity Index tertiles for the interval between symptom onset and first office visit (mean ± SD lowest tertile 77 ± 67 days [95% confidence interval (CI) 60 to 94], middle tertile 69 ± 94 days [95% CI 50 to 89], and highest tertile 56 ± 58 days [95% CI 41 to 71]; p = 0.3). Similarly, we found no association between lower levels of socioeconomic opportunity, as expressed by the lack of difference between the Childhood Opportunity Index tertiles and the time elapsed from the first office visit to the first chemotherapy session (lowest tertile 19 ± 12 days [95% CI 12 to 26], middle 19 ± 14 days [95% CI 11 to 26], and highest 15 ± 9.7 days [95% CI 8.4 to 21]; p = 0.31), the time to surgical resection (lowest tertile 99 ± 35 days [95% CI 87 to 111], middle 88 ± 28 days [95% CI 77 to 99], and highest 102 ± 64 days [95% CI 86 to 118]; p = 0.24), or the type of surgical resection (limb-sparing versus amputation: 84% [21 of 25] in lowest tertile, 83% [24 of 29] in the middle tertile, and 81% [48 of 59] in the highest tertile received limb-sparing surgery; p = 0.52). Finally, we found no differences in terms of disease-free survival at 5 years (lowest tertile 27% [95% CI 7.8% to 43%], middle 44% [95% CI 23% to 59%], and highest 56% [95% CI 40% to 67%]; p = 0.22), overall survival (lowest 74% [95% CI 58% to 95%], middle 82% [95% CI 68% to 98%], and highest 64% [95% CI 52% to 78%]; p = 0.27), or in terms of survivorship of the cohort, excluding patients who presented with metastatic disease (lowest 84% [95% CI 68% to 100%], middle 91% [95% CI 80% to 100%], and highest 68% [95% CI 55% to 83%]; p = 0.10).
In our single-center retrospective study of 113 children who presented with osteosarcoma, we did not find an association between a patient's national socioeconomic opportunity and their time to presentation, chemotherapy treatment, time to and type of surgical resection, or disease-free and overall survival. Prior work has shown an association between socioeconomic background and disparities in osteosarcoma treatment. It is possible that these findings will be similar to those from other hospitals and geographic areas, but based on our findings, we believe that proximity to providers, access to public transit, and regional insurance policies may help diminish these disparities. Future multicenter studies are needed to further explore the role that regional variations and the aforementioned factors may play in osteosarcoma treatment to help inform the direction of public policy.
Level III, therapeutic study.
已发表的研究报告探讨了社会经济因素及其与骨肉瘤表现和治疗的关系,提示社会经济地位较低与化疗反应较差及生存率较低之间存在关联。然而,这些差异背后的驱动因素尚不清楚。儿童机会指数由diversitydatakids.org(https://www.diversitydatakids.org/ )于2014年制定,用于在一个专门针对儿科人群并按普查区组织的指数中累积量化健康的社会决定因素。儿童机会指数可用于探讨患者的社会经济背景与骨肉瘤表现、治疗及结果差异之间的关系。
问题/目的:儿童机会指数得分的差异是否与以下方面的差异相关:(1)从症状出现到首次因骨肉瘤就诊的时间;(2)化疗时间或手术切除的时间及类型;(3)初始疾病严重程度、转移性疾病的发生或总体生存率?
采用回顾性治疗研究,数据来源于美国中大西洋地区一家大型儿科三级癌症中心2006年至2022年的机构记录。我们的主要院区位于城市环境,公共交通便利。如果患者寻求二次诊断或我们的机构不是骨科护理的主要地点,则将其排除在分析之外(20%[223例中的45例])。在其余患者中,那些电子病历不完整的患者(24%[178例中的43例])、居住在国外的患者(5%[178例中的9例])、复发后就诊的患者(4%[178例中的8例])或在我们机构缺乏2年随访的患者(3%[178例中的5例])也被排除。共有113名儿科患者(18岁以下儿童)符合纳入标准。儿童机会指数是一个综合指数,源自三个领域(教育、健康与环境以及社会与经济)以及这些领域内的29项指标,用于捕捉差异对儿童福祉的累积影响。收集全国儿童机会得分并从1到100进行评分。每个得分代表美国18岁及以下儿童人口的相等比例。得分越高表明社会经济机会水平越高。然后将儿童机会指数的总体得分分为三组,代表儿童相对的社会经济机会:得分<34为最低三分位数,得分在34至66之间为中间三分位数,得分>66为最高三分位数。使用均值、范围、中位数、四分位数间距和百分比来描述研究样本。对三组(最低三分位数、中间三分位数和最高三分位数)进行数据分析,评估就诊时间、治疗差异、疾病严重程度和总体生存率的差异。应用卡方检验和Fisher精确检验比较分类变量。Mann-Whitney U检验比较连续数据。按儿童机会指数三分位数分层进行Kaplan-Meier生存分析,为期5年,以评估转移性疾病的发生和总体生存率。应用对数秩检验评估统计学意义。由于样本量较小,我们无法控制种族和保险等潜在混杂因素。然而,儿童机会指数数据所涵盖的三个领域(教育、健康与环境以及社会与经济)间接考虑了与种族和保险状况相关的差异。
社会经济机会水平较低与症状出现至首次就诊间隔时间的儿童机会指数三分位数之间无差异(最低三分位数平均±标准差77±67天[95%置信区间(CI)60至94],中间三分位数69±94天[95%CI 50至89],最高三分位数56±58天[95%CI 41至71];p = 0.3)。同样地,我们发现社会经济机会水平较低与儿童机会指数三分位数和首次就诊至首次化疗疗程的时间间隔之间无差异(最低三分位数19±12天[95%CI 12至26],中间三分位数19±14天[95%CI 11至26],最高三分位数15±9.7天[95%CI 8.4至21];p = 0.31),手术切除时间(最低三分位数99±35天[95%CI 87至111],中间三分位数88±28天[95%CI 77至99],最高三分位数102±64天[95%CI 86至118];p = 0.24),或手术切除类型(保肢与截肢:最低三分位数中84%[25例中的21例],中间三分位数中83%[29例中的24例],最高三分位数中81%[59例中的48例]接受保肢手术;p = 0.52)。最后,我们发现5年无病生存率(最低三分位数27%[95%CI 7.8%至43%],中间三分位数44%[95%CI 23%至59%],最高三分位数56%[95%CI 40%至67%];p = 0.22)、总体生存率(最低三分位数74%[95%CI 58%至95%],中间三分位数82%[95%CI 68%至98%],最高三分位数64%[95%CI 52%至78%];p = 0.27),或在排除出现转移性疾病的患者后的队列生存率方面(最低三分位数84%[95%CI 68%至100%],中间三分位数91%[95%CI 80%至100%],最高三分位数68%[95%CI 55%至83%];p = 0.10)均无差异。
在我们对113例骨肉瘤患儿的单中心回顾性研究中,我们未发现患者的全国社会经济机会与其就诊时间、化疗治疗、手术切除时间及类型、或无病生存率和总体生存率之间存在关联。先前的研究表明社会经济背景与骨肉瘤治疗差异之间存在关联。这些发现可能与其他医院和地理区域的发现相似,但基于我们的研究结果,我们认为靠近医疗服务提供者、公共交通便利以及地区保险政策可能有助于减少这些差异。未来需要多中心研究进一步探讨区域差异和上述因素在骨肉瘤治疗中可能发挥的作用,以指导公共政策的方向。
III级,治疗性研究。