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慢性点头综合征的神经学

The neurology of chronic nodding syndrome.

作者信息

Olum Sam, Hardy Charlotte, Obol James, Scolding Neil

机构信息

Faculty of Medicine, Gulu University, c/o Guest House, Gulu, Uganda.

出版信息

Brain Commun. 2022 Jun 6;4(3):fcac126. doi: 10.1093/braincomms/fcac126. eCollection 2022.

Abstract

Nodding syndrome is an uncommon disorder of childhood onset and unknown cause, presenting with nodding seizures, and which appears to occur exclusively in clusters in sub-Saharan Africa. An endemic pattern of disease was initially described in Tanzania and in Liberia; epidemic occurrences were later reported in South Sudan and northern Uganda. Not the least significant of the many questions remaining about nodding syndrome concerns the common presence or otherwise of neurological features other than seizures-clearly relevant to the core issue of whether this is a focal, primary epileptic disease, or a multi-system CNS disorder, with, in turn implications for its aetiology. We had the opportunity to interview and clinically to examine 57 affected individuals in rural northern Uganda some 10 years after onset. In this observational cross-sectional study, nodding onset was invariably between the ages of 5 and 14, presenting with food-triggered nodding attacks in over 75% of cases; 86% went on to develop other seizure types. In 53 of 57 nodding syndrome individuals (93%), there was a definite history of the child and his or her family having resided in or been fed from an internally displaced person camp for some time prior to the onset of nodding. A half of nodding syndrome sufferers (28/57) had focal neurological abnormalities-mainly pyramidal signs (92%), often asymmetric, some with extrapyramidal abnormalities. Many individuals (28/57) were severely functionally disabled, ranging from 'sometimes can dig' to 'can do nothing at home' or 'cannot even feed herself'. Such sufferers tended more frequently to have significant burns, and clear cognitive impairment. We conclude that nodding syndrome is a unique multisystem CNS disorder of childhood onset and then slow progression over several years often followed by spontaneous stabilisation, consistent with an underlying self-limiting neurodegenerative process. We discuss the possibility that this might be triggered by food-related mycotoxins, within a fixed window of CNS vulnerability during childhood.

摘要

点头综合征是一种罕见的儿童期起病且病因不明的疾病,表现为点头样癫痫发作,且似乎仅在撒哈拉以南非洲呈聚集性出现。该疾病的地方性模式最初在坦桑尼亚和利比里亚被描述;后来在南苏丹和乌干达北部报告了其流行情况。关于点头综合征仍存在的诸多问题中,最关键的一点涉及除癫痫发作外是否普遍存在其他神经学特征——这显然与该疾病是局灶性原发性癫痫疾病还是多系统中枢神经系统疾病这一核心问题相关,进而对其病因学也有影响。在发病约10年后,我们有机会对乌干达北部农村地区的57名患者进行访谈和临床检查。在这项观察性横断面研究中,点头发作年龄均在5至14岁之间,超过75%的病例表现为食物诱发的点头发作;86%的患者随后发展出其他癫痫发作类型。在57名点头综合征患者中有53名(93%),明确有儿童及其家庭在点头发作前曾在境内流离失所者营地居住或从该营地获取食物一段时间的病史。一半的点头综合征患者(28/57)有局灶性神经学异常——主要是锥体束征(92%),通常不对称,部分伴有锥体外系异常。许多患者(28/57)存在严重的功能障碍,从“有时能挖掘”到“在家什么都做不了”或“甚至无法自己进食”。这些患者更容易出现严重烧伤和明显的认知障碍。我们得出结论,点头综合征是一种独特的儿童期起病的多系统中枢神经系统疾病,随后数年缓慢进展,常自发稳定,符合潜在的自限性神经退行性过程。我们讨论了在儿童期中枢神经系统易损的特定窗口期内,这可能由与食物相关的霉菌毒素引发的可能性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/442e/9178964/e63ed34bda25/fcac126ga1.jpg

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