一名患有杜氏肌营养不良症的男孩晚期用阿特洛仑治疗后反应良好:以前的轻度病程会影响结果吗?

Q3 Medicine
Acta Myologica Pub Date : 2022-09-30 eCollection Date: 2022-01-01 DOI:10.36185/2532-1900-078
Ludovica Pasca, Alice Gardani, Matteo Paoletti, Daniele Velardo, Angela Berardinelli
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引用次数: 1

摘要

杜氏肌营养不良症(DMD)是一种严重的进行性x连锁隐性遗传病,由肌营养不良蛋白缺失引起。目前还没有一种治疗DMD的有效方法。首个被批准用于无义突变DMD患者的药物是ataluren,被批准用于治疗≥2岁的儿童,似乎可以有效减缓疾病进展。较早地引入阿塔鲁伦似乎会产生更好的效果。我们报告一名14岁的DMD患者,外显子70无义突变,仍然不能走动,他在12岁时开始服用阿他仑,并在接下来的两年保持稳定。患者从6岁开始服用类固醇,效果良好。在两年的随访中,观察到最佳的疾病演变,与肌酸激酶血液水平的持续下降有关。尽管治疗开始较晚,但ataluren似乎对该患者功能状态的稳定做出了重大贡献,尽管不能排除结果可能受到先前良好病程的影响。然而,应该计划在年龄相近的患者中进行进一步的研究,以更好地评估与疾病自然病程相比的治疗结果。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

Good response to the late treatment with ataluren in a boy with Duchenne muscular dystrophy: could the previous mild course of the disease have affected the outcome?

Good response to the late treatment with ataluren in a boy with Duchenne muscular dystrophy: could the previous mild course of the disease have affected the outcome?

Good response to the late treatment with ataluren in a boy with Duchenne muscular dystrophy: could the previous mild course of the disease have affected the outcome?

Duchenne muscular dystrophy (DMD) is a severe, progressive X-linked recessive disorder, caused by the absence of the dystrophin protein. A resolutive therapy for DMD is not yet available. The first approved drug for DMD patients with nonsense mutations is ataluren, approved for the treatment of children aged ≥ 2 yrs, that seems effective in slowing the disease progression. An earlier introduction of ataluren seems to give better results. We report the case of a 14-year-old DMD patient with a nonsense mutation in exon 70, still ambulant, who started taking ataluren at 12 years and remained stable for the following two years. The patient was on steroid since the age of 6, with beneficial effects. At two-years follow-up, an optimal disease evolution was observed, associated with a constant decrease of creatine kinase blood levels. Despite the late start of the treatment, ataluren seems to have significantly contributed to the stabilization of the functional status in this patient though it cannot be excluded that the result may have been influenced by the previous favorable course of the disease. However, further studies should be planned in patients with similar age treated with ataluren to better evaluate the treatment's results compared to the natural course of the disease.

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来源期刊
Acta Myologica
Acta Myologica Medicine-Cardiology and Cardiovascular Medicine
CiteScore
3.70
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