Neurosciences, Great Ormond Street Hospital NHS Foundation Trust, London, UK.
Neurosciences, Great Ormond Street Hospital NHS Foundation Trust, London, UK; Clinical Neurosciences, UCL- Institute of Child Health, London, UK.
Pediatr Neurol. 2019 Apr;93:27-33. doi: 10.1016/j.pediatrneurol.2018.11.012. Epub 2018 Nov 28.
Children with Sturge-Weber syndrome can experience severe headache with or without transient hemiparesis. Flunarizine, a calcium antagonist, has been used for migraine. The experience with flunarizine for headache in a cohort of children at a national center for Sturge-Weber syndrome is reviewed, reporting its efficacy and adverse effect in this population.
We collected data from health care professionals' documentation on headache (severity, frequency, duration) before and on flunarizine in 20 children with Sturge-Weber syndrome. Adverse effects reported during flunarizine treatment were collated. The Wilcoxon signed rank test was used to determine the significance of pre- versus post-treatment effect.
Flunarizine was used for headache alone (13) or mixed migrainous episodes and vascular events (7). The median duration of treatment was 145 days (range 43 to 1864 days). Flunarizine reduced headache severity (z = -3.354, P = 0.001), monthly frequency (z = -2.585, P = 0.01), and duration (z = -2.549, P = 0.01). Flunarizine was discontinued owing to intolerable adverse effects in a minority (2). Sedation and weight gain were the most common side effects. There were no reports of behavior change or extrapyramidal features.
The most effective management for headaches in patients with Sturge-Weber syndrome has not been established. This retrospective observational study found benefit of flunarizine prophylaxis on headache severity, frequency, and duration in children with Sturge-Weber syndrome without severe side effects. Flunarizine is not licensed for use in the United Kingdom, but these data support its off-license specialist use for headache prophylaxis in Sturge-Weber syndrome.
患有 Sturge-Weber 综合征的儿童可能会出现严重头痛，伴有或不伴有短暂偏瘫。氟桂利嗪是一种钙拮抗剂，已用于偏头痛的治疗。本研究回顾了全国性 Sturge-Weber 综合征中心使用氟桂利嗪治疗头痛的儿童队列的经验，报告了其在该人群中的疗效和不良反应。
我们从医疗保健专业人员记录的头痛（严重程度、频率、持续时间）中收集了 20 例 Sturge-Weber 综合征患儿使用氟桂利嗪治疗前后的数据。整理了使用氟桂利嗪治疗期间报告的不良反应。采用 Wilcoxon 符号秩检验比较治疗前后的效果。
氟桂利嗪单独用于头痛（13 例）或混合偏头痛发作和血管事件（7 例）。中位治疗时间为 145 天（43 至 1864 天）。氟桂利嗪降低了头痛的严重程度（z=-3.354，P=0.001）、每月发作频率（z=-2.585，P=0.01）和持续时间（z=-2.549，P=0.01）。少数患者因不良反应不能耐受而停用氟桂利嗪（2 例）。镇静和体重增加是最常见的副作用。无行为改变或锥体外系表现的报告。
Sturge-Weber 综合征患者头痛的最佳治疗方法尚未确定。本回顾性观察性研究发现，氟桂利嗪预防治疗可有效减轻 Sturge-Weber 综合征患儿头痛的严重程度、频率和持续时间，且无严重不良反应。氟桂利嗪在英国未获得许可用于治疗头痛，但这些数据支持其在 Sturge-Weber 综合征中的专科应用。
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