[庞贝氏症早期治疗和酶替代疗法定量评估的重要性:阿糖苷酶α上市后监测补充分析]。

Q4 Medicine Clinical Neurology Pub Date : 2024-11-21 DOI:10.5692/clinicalneurol.cn-001894
Yoshinori Sunaga, Tatsuro Sakashita, Tadashi Koga, Takayuki Sawada, Shiho Yamane, Mitsunobu Ikeda
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引用次数: 0

摘要

我们利用阿糖苷酶α治疗庞贝氏症的上市后监测数据进行了额外分析。我们的目的是根据总体改善情况调查预测强迫生命容量百分比(%FVC)的变化和 6 分钟步行测试(6MWT)距离的变化,并使用线性混合模型根据从症状发作到调查登记的持续时间(较短/较长组别)调查%FVC 的变化。分别有 37 名和 18 名调查参与者提供了 %FVC 和 6MWT 数据;在总体改善被评为 "相对改善 "的患者中,分别有 71.4% 和 66.7% 的患者的 %FVC 和 6MWT 出现恶化。使用线性混合模型估算的调查登记时的 FVC 百分比,短程组明显高于长程组(P = 0.0413)。估计的 FVC 百分比斜率,短程组明显低于长程组(P = 0.0051)。这些结果表明,尽早开始治疗并对每种症状进行定量评估非常重要。
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[Importance of early treatment and quantitative evaluation of enzyme replacement therapy for Pompe disease: alglucosidase alfa post-marketing surveillance additional analysis].

We conducted an additional analysis using the data from the post-marketing surveillance of Alglucosidase alfa for Pompe disease. We aimed to investigate the changes in the percentage of predicted forced vital capacity (%FVC) and the changes in the distance of the 6-min walk test (6MWT) by overall improvement and to investigate the %FVC change by the duration from symptom onset to survey registration (shorter/longer groups) using a linear mixed model. Thirty-seven and eighteen survey participants had %FVC and 6MWT data available, respectively; of the patients whose overall improvement was rated as "relatively improved," %FVC and 6MWT worsened in 71.4% and 66.7%, respectively. The %FVC at the survey registration estimated using a linear mixed model was significantly higher in the shorter group than in the longer group (P = 0.0413). The estimated slope of %FVC was significantly lower in the shorter group than in the longer group (P = 0.0051). These results suggest the importance of early treatment initiation and quantitative evaluation of each symptom.

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来源期刊
Clinical Neurology
Clinical Neurology Medicine-Neurology (clinical)
CiteScore
0.30
自引率
0.00%
发文量
147
期刊最新文献
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