McMaster Children's Hospital, Hamilton, Ontario, Canada.
Cancer Research Program, ICES, Toronto, Ontario, Canada.
Pediatr Blood Cancer. 2022 Oct;69(10):e29829. doi: 10.1002/pbc.29829. Epub 2022 Jun 8.
Children with Down syndrome (DS) and acute lymphoblastic leukemia (ALL) are at increased risk of treatment-related morbidity and mortality compared to non-DS-ALL, requiring increased supportive care. We examined the healthcare utilization and costs in DS-ALL patients to inform future evaluations of novel therapies.
A provincial registry identified all children (1-17 years) diagnosed with B-lineage ALL in Ontario, Canada between 2002 and 2012. Detailed demographic, disease, treatment, and outcome data were abstracted. Linkage to population-based health services databases identified all outpatient and emergency department (ED) visits, hospitalizations, and physician billings. Healthcare utilization costs were available for patients diagnosed during 2006-2012 using validated algorithms (2018 Canadian dollars). Healthcare utilization rates and costs were compared between DS and non-DS patients using regression models, adjusting for all covariates.
Of 711 patients, 28 (3.9%) had DS. Adjusting for all covariates, children with DS-ALL experienced substantially higher rates of ED visits (rate ratio [RR] 1.5, 95% confidence interval [95% CI]: 1.2-2.0; p = .001) and inpatient days (RR 2.5, 95% CI: 1.4-4.5; p = .002) compared to non-DS children. Outpatient visit rates were similar (RR 1.1, 95% CI: 0.9-1.3; p = .41). Among patients with available cost data (N = 533, DS = 19), median 5-year healthcare utilization cost was $247,700 among DS patients (interquartile range [IQR]: 200,900-354,500) and $196,200 among non-DS patients (IQR: 148,900-280,300; p = .02). In adjusted analyses, DS-associated costs were 50% higher (RR 1.5, 95% CI: 1.2-1.9; p < .002).
Healthcare utilization and treatment costs of DS-ALL patients are substantially higher than those of non-DS-ALL. Our data provide a baseline for future DS-specific cost-effectiveness studies.
与非唐氏综合征急性淋巴细胞白血病（ALL）患儿相比，唐氏综合征（DS）患儿 ALL 患儿的治疗相关发病率和死亡率更高，需要更多的支持性护理。我们检查了 DS-ALL 患儿的医疗保健利用情况和成本，以了解新型治疗方法的未来评估。
一个省级登记处确定了 2002 年至 2012 年间在加拿大安大略省诊断为 B 系 ALL 的所有 1-17 岁儿童。详细的人口统计学、疾病、治疗和结果数据被提取出来。与基于人群的健康服务数据库的链接确定了所有的门诊和急诊（ED）就诊、住院和医生账单。利用经过验证的算法（2018 加元），可获得 2006-2012 年期间诊断的患者的医疗保健利用成本。使用回归模型比较 DS 和非 DS 患者的医疗保健利用率和成本，调整所有协变量。
在 711 名患者中，有 28 名（3.9%）患有 DS。调整所有协变量后，DS-ALL 患儿的 ED 就诊率（率比 [RR] 1.5，95%置信区间 [95%CI]：1.2-2.0；p=0.001）和住院天数（RR 2.5，95%CI：1.4-4.5；p=0.002）均显著高于非 DS 患儿。门诊就诊率相似（RR 1.1，95%CI：0.9-1.3；p=0.41）。在可获得成本数据的患者中（N=533，DS=19），DS 患者的 5 年医疗保健利用总成本中位数为 247700 加元（四分位距 [IQR]：200900-354500），非 DS 患者为 196200 加元（IQR：148900-280300；p=0.02）。在调整后的分析中，DS 相关成本增加了 50%（RR 1.5，95%CI：1.2-1.9；p<0.002）。
DS-ALL 患儿的医疗保健利用情况和治疗费用明显高于非 DS-ALL 患儿。我们的数据为未来针对 DS 的成本效益研究提供了基线。
