Slonim Alfred E, Bulone Linda, Goldberg Teresia, Minikes Jennifer, Slonim Efrat, Galanko Joseph, Martiniuk Frank
Department of Pediatrics, Division of Molecular Genetics, Columbia University Medical College, 3959 Broadway BH6N, Room 601A, New York, New York 10032, USA.
Muscle Nerve. 2007 Jan;35(1):70-7. doi: 10.1002/mus.20665.
Adult-onset acid maltase deficiency is an inherited lysosomal skeletal-muscle disease characterized by progressive myopathy and respiratory failure, for which there is no known therapy. In an uncontrolled, prospective study, we evaluated whether adherence to high-protein and low-carbohydrate nutrition and exercise therapy (NET) can slow the progressive deterioration of muscle function in this disease. Thirty-four patients have been treated with NET for periods of 2-10 years (mean 4.5 +/- 2.5). Pre-NET rate of muscle function deterioration, as measured by the Walton scale, was compared to post-NET rate. Twenty-six patients were deemed to be consistently compliant with NET. Difference between pre-NET slope of muscle function deterioration to that of post-NET slope in compliant patients was -0.29 (95% CI -0.19, 0.39) (P < 0.0001). We conclude that compliance with NET can slow deterioration of muscle function and improve the natural history of adult-onset acid maltase deficiency. Muscle Nerve, 2006.
成人型酸性麦芽糖酶缺乏症是一种遗传性溶酶体骨骼肌疾病,其特征为进行性肌病和呼吸衰竭,目前尚无已知的治疗方法。在一项非对照前瞻性研究中,我们评估了坚持高蛋白和低碳水化合物营养及运动疗法(NET)是否能减缓该疾病中肌肉功能的进行性恶化。34例患者接受NET治疗2至10年(平均4.5±2.5年)。通过沃尔顿量表测量,将NET治疗前的肌肉功能恶化率与NET治疗后的进行比较。26例患者被认为始终坚持NET治疗。在坚持治疗的患者中,NET治疗前肌肉功能恶化斜率与NET治疗后斜率的差异为-0.29(95%可信区间-0.19,0.39)(P<0.0001)。我们得出结论,坚持NET治疗可减缓肌肉功能恶化,并改善成人型酸性麦芽糖酶缺乏症的自然病程。《肌肉与神经》,2006年。