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Abstract：
BACKGROUND: Survivors of childhood central nervous system (CNS) tumors can develop motor and sensory impairment from their cancer and treatment history. We estimated the prevalence of motor and sensory impairment in survivors compared with controls through clinical assessment and identified associated treatment exposures and functional, quality of life (QOL), and social outcomes.
METHODS: Survivors of childhood CNS tumors from the St. Jude Lifetime Cohort (n = 378, median [range] age 24.0 [18.0-53.0] years, 43.4% female) ≥5 years from diagnosis and controls (n = 445, median [range] age 34.0 [18.0-70.0] years, 55.7% female) completed in-person evaluation for motor and sensory impairment using the modified Total Neuropathy Score. Impairment was graded by modified Common Terminology Criteria for Adverse Events. Multivariable models estimated associations between grade ≥2 motor/sensory impairment, individual/treatment characteristics, and secondary outcomes (function by Physical Performance Test, fitness by physiologic cost index, QOL by Medical Outcomes Survey Short Form-36 physical/mental summary scores, social attainment).
RESULTS: Grade ≥2 motor or sensory impairment was more prevalent in survivors (24.1%, 95% Confidence Interval [CI] 19.8%-29.4%) than controls (2.9%, CI 1.4-4.5%). Among survivors, in multivariable models, motor impairment was associated with vinca exposure <15 mg/m2 versus none (OR 4.38, CI 1.06-18.08) and etoposide exposure >2036 mg/m2 versus none (OR 12.61, CI 2.19-72.72). Sensory impairment was associated with older age at diagnosis (OR 1.09, CI 1.01-1.16) and craniospinal irradiation versus none (OR 4.39, CI 1.68-11.50). There were lower odds of motor/sensory impairment in survivors treated in the year 2000 or later versus before 1990 (Motor: OR 0.29, CI 0.10-0.84, Sensory: OR 0.35, CI 0.13-0.96). Motor impairment was associated with impaired physical QOL (OR 2.64, CI 1.22-5.72).
CONCLUSIONS: In survivors of childhood CNS tumors, motor and sensory impairment is prevalent by clinical assessment, especially after exposure to etoposide, vinca, or craniospinal radiation. Treating motor impairment may improve survivors\' QOL.
METHODS: Survivors of childhood CNS tumors from the St. Jude Lifetime Cohort (n = 378, median [range] age 24.0 [18.0-53.0] years, 43.4% female) ≥5 years from diagnosis and controls (n = 445, median [range] age 34.0 [18.0-70.0] years, 55.7% female) completed in-person evaluation for motor and sensory impairment using the modified Total Neuropathy Score. Impairment was graded by modified Common Terminology Criteria for Adverse Events. Multivariable models estimated associations between grade ≥2 motor/sensory impairment, individual/treatment characteristics, and secondary outcomes (function by Physical Performance Test, fitness by physiologic cost index, QOL by Medical Outcomes Survey Short Form-36 physical/mental summary scores, social attainment).
RESULTS: Grade ≥2 motor or sensory impairment was more prevalent in survivors (24.1%, 95% Confidence Interval [CI] 19.8%-29.4%) than controls (2.9%, CI 1.4-4.5%). Among survivors, in multivariable models, motor impairment was associated with vinca exposure <15 mg/m2 versus none (OR 4.38, CI 1.06-18.08) and etoposide exposure >2036 mg/m2 versus none (OR 12.61, CI 2.19-72.72). Sensory impairment was associated with older age at diagnosis (OR 1.09, CI 1.01-1.16) and craniospinal irradiation versus none (OR 4.39, CI 1.68-11.50). There were lower odds of motor/sensory impairment in survivors treated in the year 2000 or later versus before 1990 (Motor: OR 0.29, CI 0.10-0.84, Sensory: OR 0.35, CI 0.13-0.96). Motor impairment was associated with impaired physical QOL (OR 2.64, CI 1.22-5.72).
CONCLUSIONS: In survivors of childhood CNS tumors, motor and sensory impairment is prevalent by clinical assessment, especially after exposure to etoposide, vinca, or craniospinal radiation. Treating motor impairment may improve survivors\' QOL.
摘要：
背景：儿童中枢神经系统（CNS）肿瘤的幸存者可以从他们的癌症和治疗史发展为运动和感觉障碍。我们通过临床评估，与对照组相比，评估了幸存者运动和感觉障碍的患病率，并确定了相关的治疗暴露和功能，生活质量（QOL），和社会结果。
方法：来自St.Jude终身队列的儿童中枢神经系统肿瘤的幸存者（n=378，年龄中位数为24.0[18.0-53.0]岁，43.4％女性）自诊断和对照组起≥5年（n=445，中位[范围]年龄34.0[18.0-70.0]岁，55.7％女性）使用改良的总神经病变评分完成了对运动和感觉障碍的亲自评估。根据修改后的不良事件通用术语标准对损害进行分级。多变量模型估计≥2级运动/感觉障碍之间的关联，个体/治疗特征，和次要结果(身体机能测试，按生理成本指数计算的健身，按医疗结果调查的QOL简短表格-36身体/心理汇总分数，社会素养)。
结果：≥2级运动或感觉障碍在幸存者中更为普遍（24.1％，95%置信区间[CI]19.8%-29.4%）比对照组（2.9%，CI1.4-4.5%）。在幸存者中，在多变量模型中，运动障碍与长春花暴露<15mg/m2和无(OR4.38,CI1.06-18.08)以及依托泊苷暴露>2036mg/m2和无(OR12.61,CI2.19-72.72)相关.感觉障碍与诊断时的年龄（OR1.09，CI1.01-1.16）和颅骨照射与无（OR4.39，CI1.68-11.50）有关。与1990年之前相比，在2000年或以后接受治疗的幸存者中，运动/感觉障碍的几率较低（运动：OR0.29，CI0.10-0.84，感觉：OR0.35，CI0.13-0.96）。运动障碍与身体生活质量受损相关（OR2.64，CI1.22-5.72）。
结论：在儿童中枢神经系统肿瘤的幸存者中，运动和感觉障碍是普遍存在的临床评估，尤其是在接触依托泊苷之后,vinca,或者颅脊髓放射.治疗运动障碍可能会改善幸存者的生活质量。
方法：来自St.Jude终身队列的儿童中枢神经系统肿瘤的幸存者（n=378，年龄中位数为24.0[18.0-53.0]岁，43.4％女性）自诊断和对照组起≥5年（n=445，中位[范围]年龄34.0[18.0-70.0]岁，55.7％女性）使用改良的总神经病变评分完成了对运动和感觉障碍的亲自评估。根据修改后的不良事件通用术语标准对损害进行分级。多变量模型估计≥2级运动/感觉障碍之间的关联，个体/治疗特征，和次要结果(身体机能测试，按生理成本指数计算的健身，按医疗结果调查的QOL简短表格-36身体/心理汇总分数，社会素养)。
结果：≥2级运动或感觉障碍在幸存者中更为普遍（24.1％，95%置信区间[CI]19.8%-29.4%）比对照组（2.9%，CI1.4-4.5%）。在幸存者中，在多变量模型中，运动障碍与长春花暴露<15mg/m2和无(OR4.38,CI1.06-18.08)以及依托泊苷暴露>2036mg/m2和无(OR12.61,CI2.19-72.72)相关.感觉障碍与诊断时的年龄（OR1.09，CI1.01-1.16）和颅骨照射与无（OR4.39，CI1.68-11.50）有关。与1990年之前相比，在2000年或以后接受治疗的幸存者中，运动/感觉障碍的几率较低（运动：OR0.29，CI0.10-0.84，感觉：OR0.35，CI0.13-0.96）。运动障碍与身体生活质量受损相关（OR2.64，CI1.22-5.72）。
结论：在儿童中枢神经系统肿瘤的幸存者中，运动和感觉障碍是普遍存在的临床评估，尤其是在接触依托泊苷之后,vinca,或者颅脊髓放射.治疗运动障碍可能会改善幸存者的生活质量。
