Neurological Department, Charles Nicolle Hospital, Boulevard 9 Avril, 1006, Tunis, Tunisia.
Neurol Sci. 2009 Aug;30(4):315-8. doi: 10.1007/s10072-009-0102-2. Epub 2009 Jun 5.
The objective of this study is to report cases of unexpected deaths in Unverricht-Lundborg disease (ULD) patients, a comparatively benign form of progressive myoclonus epilepsy. We performed a multicentric study of the circumstances of death in ULD patients seen in the last 16 years. We assessed age, sex, severity and duration of disease, antiepileptic drugs, circumstances and presumed cause of death. Nineteen observations (12 females, 7 males) were collected from four centers (Tunis, Marseille, Milan, Belgrade). The most common causes of death are (1) SUDEP (six cases, all female), with 4/6 on phenobarbital alone, and (2) complications of severe ULD (six cases). Two patients committed suicide. Only one death was clearly unrelated to ULD (car accident), while four patients died of stroke, drowning, complications of chronic alcoholism and Wernicke encephalopathy, respectively. In conclusion, although the prognosis of ULD has progressed, there are still spontaneously severe forms and high risk of early death, including SUDEP.
本研究旨在报告 Unverricht-Lundborg 病(ULD)患者意外死亡的病例,ULD 是一种相对良性的进行性肌阵挛癫痫。我们对过去 16 年中在四个中心(突尼斯、马赛、米兰、贝尔格莱德)观察到的 ULD 患者的死亡情况进行了一项多中心研究。我们评估了年龄、性别、疾病的严重程度和持续时间、抗癫痫药物、死亡的情况和推测原因。从四个中心共收集了 19 例观察结果(12 例女性,7 例男性)。最常见的死亡原因是(1)SUDEP(6 例,均为女性),其中 4/6 例仅用苯巴比妥治疗,以及(2)严重 ULD 的并发症(6 例)。两名患者自杀。只有 1 例死亡显然与 ULD 无关(车祸),而另外 4 例分别死于中风、溺水、慢性酒精中毒并发症和 Wernicke 脑病。总之,尽管 ULD 的预后有所改善,但仍存在自发性严重形式和早逝的高风险,包括 SUDEP。