John Walton Muscular Dystrophy Research Centre, Newcastle University International Centre for Life, Newcastle upon Tyne, United Kingdom.
Institute of Myology, Pitié Salpêtrière University Hospital, Paris, France.
Neuromuscul Disord. 2023 Feb;33(2):208-217. doi: 10.1016/j.nmd.2022.12.008. Epub 2022 Dec 15.
Although mexiletine effectively treats myotonia, supply disruptions affected Europe between 2008-2018. MyoPath was a mixed-methods, cross-sectional, market research survey conducted January-June 2018 to evaluate consequences of limited access to/awareness of mexiletine in people with myotonia. Part A: qualitative structured interviews (clinicians; advocates for adult patients); Part B: quantitative online questionnaire completed by people with self-reported history of myotonia. Part A: Interviews (clinicians, n=12; patient advocates, n=5; 12 countries) indicated poor mexiletine awareness among general neurologists. Patients chose between living with myotonia (other treatments were generally unsatisfactory) or importing mexiletine. Part B: Questionnaire respondents, myotonic dystrophy (DM)1, n=213; DM2, n=128; non-dystrophic myotonia (NDM), n=41; other n=8; (11 countries). Of the respondents, 76/390 (20%) people with awareness of/access to mexiletine described profound improvements in myotonia and health-related quality of life following treatment. Respondents with NDM had greatest mexiletine experience (n=28/41). Mexiletine was associated with fewer falls, less muscle stiffness, increased mobility. Treatment interruptions worsened myotonia and were associated with fatigue, pain, dysphagia, breathing difficulty, impaired digestion, poor sleep. However, 36/54 (67%) of currently treated people expressed anxiety about mexiletine's availability: this finding was expected (MyoPath was undertaken before mexiletine's approval in NDM). MyoPath provides the largest European exploration of patients' views regarding impact of mexiletine on myotonia. Anticipated effects of mexiletine differ between people with different myotonic disorders: myotonia is the main symptom in NDM but one of many potential symptoms affecting those with DM. Nevertheless, findings indicate substantial harm caused to people with myotonia when mexiletine awareness/access is limited.
尽管美西律有效地治疗肌强直,但 2008 年至 2018 年间,药物供应中断影响了欧洲。MyoPath 是一项混合方法、横断面、市场研究调查,于 2018 年 1 月至 6 月进行,旨在评估肌强直患者对美西律获取/认知受限的后果。部分 A:定性结构访谈(临床医生;成人患者的倡导者);部分 B:通过自我报告有肌强直病史的人完成的在线定量问卷。部分 A:访谈(临床医生,n=12;患者倡导者,n=5;12 个国家)表明,普通神经科医生对美西律的认知度较低。患者在忍受肌强直(其他治疗方法通常不尽如人意)或进口美西律之间做出选择。部分 B:问卷受访者,肌强直性营养不良 1 型(DM1),n=213;DM2,n=128;非肌强直性肌强直(NDM),n=41;其他,n=8;(11 个国家)。在回答者中,76/390(20%)对美西律有认知/有获取途径的人表示,治疗后肌强直和健康相关生活质量有显著改善。NDM 患者的美西律经验最多(n=28/41)。美西律与较少的跌倒、较少的肌肉僵硬、增加的活动度相关。治疗中断使肌强直恶化,并与疲劳、疼痛、吞咽困难、呼吸困难、消化不良、睡眠不佳相关。然而,54 名目前接受治疗的人中,有 36 名(67%)对美西律的供应表示焦虑:这一发现是意料之中的(MyoPath 在 NDM 批准之前进行)。MyoPath 是对欧洲患者对美西律对肌强直影响的看法进行的最大规模探索。不同肌强直性疾病患者对美西律的预期影响不同:NDM 中的主要症状是肌强直,但影响 DM 患者的许多潜在症状之一。然而,研究结果表明,当美西律的认知/获取受到限制时,肌强直患者会受到严重伤害。