Weber Amanda, Cole Justin W, Mytinger John R
Division of Pediatric Neurology, Department of Pediatrics, Nationwide Children's Hospital and The Ohio State University, Columbus, Ohio.
Department of Pharmacy, Nationwide Children's Hospital, Columbus, Ohio.
Pediatr Neurol. 2015 Aug;53(2):130-4. doi: 10.1016/j.pediatrneurol.2015.05.003. Epub 2015 May 9.
Infantile spasms are seizures typical of an age-related epileptic encephalopathy. Although evidence supporting topiramate for infantile spasms is lacking, many clinicians use it for this indication. The aim of this study was to determine the rate of infantile spasm remission with topiramate at our institution. A low rate of infantile spasm remission was hypothesized.
This was a single-center retrospective medical record review of patients treated with topiramate for infantile spasms between January 2009 and September 2013. Records were reviewed for accuracy of diagnosis and outcome. Clinical remission of infantile spasms was defined as resolution for at least 28 days at any time during treatment with topiramate. For patients with clinical remission, posttreatment electroencephalographs were reviewed to assess for electrographic remission. To assess for confounding variables affecting remission rate, demographics and outcomes were compared with patients treated with adrenocorticotropic hormone within the same period using the same criteria for remission.
Three of 31 (9.7%) patients achieved clinical remission with topiramate, two of whom also experienced electrographic remission. The third patient had electrographic remission with previous adrenocorticotropic hormone treatment but infantile spasm remission only after receiving topiramate. All three of these patients experienced subsequent electroclinical relapse during topiramate therapy. Although there were no significant demographic differences between the topiramate and adrenocorticotropic hormone cohorts, more adrenocorticotropic hormone patients achieved clinical remission (9.7% versus 56%; P < 0.001).
Remission of infantile spasms with topiramate was uncommon and no patient experienced persistent electroclinical remission. These findings suggest that infantile spasms respond poorly to topiramate.
婴儿痉挛是一种与年龄相关的癫痫性脑病所特有的发作形式。尽管缺乏托吡酯治疗婴儿痉挛的证据，但许多临床医生仍将其用于这一适应症。本研究的目的是确定在我们机构中使用托吡酯治疗婴儿痉挛的缓解率。我们假设婴儿痉挛缓解率较低。
这是一项单中心回顾性病历审查，研究对象为2009年1月至2013年9月期间接受托吡酯治疗婴儿痉挛的患者。审查病历以确保诊断和结果的准确性。婴儿痉挛的临床缓解定义为在使用托吡酯治疗期间的任何时间至少28天无发作。对于临床缓解的患者，复查治疗后的脑电图以评估脑电图缓解情况。为评估影响缓解率的混杂变量，将人口统计学和结果与同期使用相同缓解标准接受促肾上腺皮质激素治疗的患者进行比较。
31例患者中有3例（9.7%）使用托吡酯后达到临床缓解，其中2例也出现脑电图缓解。第3例患者在先前接受促肾上腺皮质激素治疗时出现脑电图缓解，但仅在接受托吡酯治疗后婴儿痉挛才缓解。这3例患者在托吡酯治疗期间均随后出现电临床复发。尽管托吡酯组和促肾上腺皮质激素组在人口统计学上无显著差异，但更多接受促肾上腺皮质激素治疗的患者实现了临床缓解（9.7%对56%；P<0.001）。
托吡酯治疗婴儿痉挛缓解并不常见，且无患者实现持续的电临床缓解。这些发现表明婴儿痉挛对托吡酯反应不佳。
