Department of Neurology, 1st Faculty of Medicine and General Teaching Hospital, Charles University, Katerinska 30, 120 00, Prague 2, Czech Republic,
Curr Neurol Neurosci Rep. 2015 Jan;15(1):522. doi: 10.1007/s11910-014-0522-0.
Niemann-Pick disease type C (NP-C) is a rare and progressive autosomal recessive disease leading to disabling neurological manifestation and premature death. The disease is prone to underdiagnosis because of its highly heterogeneous presentation. NP-C is characterized by visceral, neurological, and psychiatric manifestation, and its clinical picture varies according to age at onset. Although cataplexy is one of its characteristic symptoms, particularly in the late infantile and juvenile form, sleep disturbances are described only exceptionally. A combination of splenomegaly, vertical supranuclear gaze palsy, and cataplexy creates a most useful suspicion index tool for the disease. In adolescent and adult patients, when intellectual deterioration progresses and emotional reactions become flat, cataplexy usually disappears. Pathological findings in the brainstem in NP-C mouse model are compatible with the patients' symptoms including cataplexy. The authors observed cataplexy in 5 (3 with late infantile and 2 with juvenile form) out of 22 NP-C cases followed up in the past 20 years.
尼曼-皮克病 C 型(NP-C)是一种罕见的进行性常染色体隐性遗传病,可导致进行性神经功能障碍和过早死亡。由于其表现高度异质性,该病易被漏诊。NP-C 的特征为内脏、神经和精神表现,其临床表现根据发病年龄而有所不同。尽管猝倒症是其特征性症状之一,特别是在晚婴型和少年型中,但睡眠障碍仅偶有描述。脾肿大、垂直性核上性眼球运动障碍和猝倒症的组合为该病提供了一个非常有用的可疑指数工具。在青少年和成年患者中,随着智力下降和情绪反应变得平淡,猝倒症通常会消失。NP-C 小鼠模型中的脑干病理发现与包括猝倒症在内的患者症状相符。作者在过去 20 年中观察到 22 例 NP-C 病例中有 5 例(3 例为晚婴型,2 例为少年型)出现猝倒症。