Birca Ala, D'Anjou Guy, Carmant Lionel
1Department of Neurology, Sainte Justine Hospital (CHU Sainte-Justine), University of Montreal, Montreal, Quebec, Canada.
J Child Neurol. 2014 May;29(5):695-7. doi: 10.1177/0883073813483901. Epub 2013 Apr 11.
Infantile spasms constitute a severe epileptic encephalopathy of infancy with poor long-term developmental outcome. Many diverse etiologies have been associated with infantile spasms, but the pathophysiological process is still not fully understood. We describe 2 cases of previously healthy 1- and 3-month-old infants who suffered a nonaccidental head injury with extensive cerebral lesions. Both presented with acute focal seizures rapidly controlled with phenobarbital. Nevertheless, they developed infantile spasms after a latency period of 3-4 months. Spasms were rapidly controlled with vigabatrin. Both children manifested with developmental delay, either exacerbated (case 1) or elicited (case 2) by infantile spasms. Our report highlights nonaccidental head injury as a risk factor for developing infantile spasms following a seizure-free latency period. A better understanding of the pathophysiology linking accidental brain trauma with infantile spasms could lead to more effective neuroprotective strategies. In the meantime, increased awareness and follow-up are warranted.
婴儿痉挛症是一种严重的婴儿期癫痫性脑病,长期发育预后较差。婴儿痉挛症与多种不同病因相关,但病理生理过程仍未完全明了。我们描述了2例既往健康的1个月和3个月大婴儿,他们遭受了非意外性头部损伤并伴有广泛脑损伤。二者均出现急性局灶性癫痫发作,用苯巴比妥迅速控制。然而,在3 - 4个月的潜伏期后,他们发展为婴儿痉挛症。用vigabatrin迅速控制了痉挛。两名儿童均表现出发育迟缓,婴儿痉挛症使其加重(病例1)或引发(病例2)。我们的报告强调非意外性头部损伤是在无癫痫发作潜伏期后发生婴儿痉挛症的一个危险因素。更好地理解将意外脑外伤与婴儿痉挛症联系起来的病理生理学,可能会带来更有效的神经保护策略。与此同时,有必要提高认识并进行随访。