Excessive premature mortality among children with cerebral palsy in rural Uganda: a longitudinal, population-based study

Background
Studies from high-income countries reported reduced life expectancy in children with cerebral palsy (CP), while no population-based study has evaluated mortality of children with CP in sub-Saharan Africa. This study aimed to estimate the mortality rate (MR) of children with CP in a rural region of Uganda and identify risk factors and causes of death (CODs).
Methods and Findings
This population-based, longitudinal cohort study was based on data from Iganga-Mayuge Health and Demographic Surveillance System in eastern Uganda. We identified 97 children (aged 2–17 years) with CP in 2015, whom we followed to 2019. They were compared with an age-matched cohort from the general population (n=41 319). MRs, MR ratios (MRRs), hazard ratios (HRs), and immediate CODs were determined.
MR was 3952 per 100 000 person years (95% CI 2212–6519) in children with CP and 137 per 100 000 person years (95% CI 117–159) in the general population. Standardized MRR was 25·3 in the CP cohort, compared with the general population. In children with CP, risk of death was higher in those with severe gross motor impairments than in those with milder impairments (HR 6·8; p=0·007) and in those with severe malnutrition than in those less malnourished (HR=3·7; p=0·052). MR was higher in females in the CP cohort, with a higher MRR in females (53·0; 95% CI 26·4–106·3) than in males (16·3; 95% CI 7·2–37·2). Age had no significant effect on MR in the CP cohort, but MRR was higher at 10–18 years (39·6; 95% CI 14·2–110·0) than at 2–6 years (21·0; 95% CI 10·2–43·2). Anaemia, malaria, and other infections were the most common CODs in the CP cohort.
Conclusions
Risk of premature death was excessively high in children with CP in rural sub-Saharan Africa, especially in those with severe motor impairments or malnutrition. While global childhood mortality has significantly decreased during recent decades, this observed excessive mortality is a hidden humanitarian demand that needs to be addressed.

The dataset contains of the following files:
- CP_cohort–Children_and_youth_at_the_IM-HDSS.csv
- CoD–General_population_of_children_and_youth_IM-HDSS.csv
- Variable_list.pdf

Details about the variables in the tables can be found in the variable list.

背景
高收入国家的相关研究已证实，脑性瘫痪（cerebral palsy, CP）患儿的预期寿命有所缩短，但目前尚无基于人群的研究对撒哈拉以南非洲地区CP患儿的死亡率展开评估。本研究旨在估算乌干达某农村地区CP患儿的死亡率（mortality rate, MR），并明确其死亡危险因素与死亡原因（causes of death, CODs）。

方法与结果
本项基于人群的纵向队列研究以乌干达东部伊甘加-马尤盖健康与人口监测系统（Iganga-Mayuge Health and Demographic Surveillance System, IM-HDSS）的数据为基础。研究人员于2015年纳入97名2~17岁的CP患儿，并对其随访至2019年；同时以41319名来自普通人群的年龄匹配队列作为对照。本研究统计了两组的死亡率、死亡率比（mortality rate ratio, MRR）、风险比（hazard ratio, HR）以及直接死亡原因。

CP患儿组的死亡率为3952/10万人年（95%置信区间：2212~6519），普通人群对照组的死亡率为137/10万人年（95%置信区间：117~159）。与普通人群相比，CP队列的标准化死亡率比为25.3。在CP患儿中，粗大运动功能重度障碍者的死亡风险高于轻度障碍者（HR=6.8；P=0.007），重度营养不良者的死亡风险高于营养状况较好者（HR=3.7；P=0.052）。CP队列中女性患儿的死亡率更高，女性的死亡率比为53.0（95%置信区间：26.4~106.3），显著高于男性的16.3（95%置信区间：7.2~37.2）。年龄对CP队列的死亡率无显著影响，但10~18岁组的死亡率比（39.6；95%置信区间：14.2~110.0）高于2~6岁组（21.0；95%置信区间：10.2~43.2）。贫血、疟疾及其他感染是CP队列最常见的死亡原因。

结论
撒哈拉以南非洲农村地区的CP患儿过早死亡风险极高，尤其是存在重度运动障碍或营养不良的患儿。尽管近几十年来全球儿童死亡率已显著下降，但本研究观察到的过度死亡现象仍是一项亟待解决的被忽视的人道主义需求。

本数据集包含以下文件：
- CP_cohort–Children_and_youth_at_the_IM-HDSS.csv
- CoD–General_population_of_children_and_youth_IM-HDSS.csv
- Variable_list.pdf
表格中变量的详细说明可查阅变量列表文档。