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一名茶商因高锰血症引发的舞蹈症和认知功能衰退

Hypermanganesemia Induced Chorea and Cognitive Decline in a Tea Seller.

作者信息

Ghosh Ritwik, Dubey Souvik, Chatterjee Subhankar, Ghosh Mrinalkanti, Ray Biman Kanti, Benito-León Julián

机构信息

Department of General Medicine, Burdwan Medical College and Hospital, Burdwan, West Bengal, IN.

Department of Neuromedicine, Bangur Institute of Neurosciences, Kolkata, IN.

出版信息

Tremor Other Hyperkinet Mov (N Y). 2020 Oct 20;10:45. doi: 10.5334/tohm.537.

Abstract

BACKGROUND

Manganese associated neurotoxicity and neurodegeneration is quite rare yet established neurological disorder. This neurotoxic element has predilection for depositing in basal ganglia structures, manifesting mainly as parkinsonian and dystonic movement disorders with behavioral abnormalities.

CASE REPORT

We report a 40-year-old man who presented with a subacute onset bilateral, asymmetric hyperkinetic movement disorder (predominantly left sided chorea) with multi-domain cognitive impairment, dysarthria, and generalized rigidity. Clinical history and examination yielded multiple differential diagnoses including deposition and metabolic disorders, autoimmune and paraneoplastic encephalitis involving basal ganglia, and neurodegenerative disorders with chorea and cognitive impairment. However, magnetic resonance imaging was suggestive of paramagnetic substance deposition, which came out to be manganese after laboratory investigations. History, clinical examinations, and investigation results pointed towards a diagnosis of acquired hypermanganesemia due to over-ingestion of manganese containing substance (i.e., black tea). He was treated symptomatically and with chelation therapy (calcium disodium edetate). At the sixth month of follow-up, complete resolution of chorea, dysarthria and partial amelioration of rigidity were observed. His cognitive decline and behavioral abnormalities improved.

DISCUSSION

This is probably the first reported case of acquired hypermanganesemia that presented as a combination of asymmetric chorea and cognitive dysfunction with atypical imaging characteristics. The clinical picture mimicked that of Huntington's disease. We highlight the potential deleterious effects of an apparently "benign" non-alcoholic beverage (i.e., black tea) on cerebral metabolism.

摘要

背景

锰相关的神经毒性和神经退行性变是一种相当罕见但已确定的神经系统疾病。这种神经毒性元素倾向于沉积在基底神经节结构中,主要表现为帕金森氏症和肌张力障碍性运动障碍以及行为异常。

病例报告

我们报告一名40岁男性,他出现亚急性起病的双侧不对称性运动增多症(主要为左侧舞蹈症),伴有多领域认知障碍、构音障碍和全身僵硬。临床病史和检查得出了多种鉴别诊断,包括沉积和代谢紊乱、累及基底神经节的自身免疫性和副肿瘤性脑炎,以及伴有舞蹈症和认知障碍的神经退行性疾病。然而,磁共振成像提示有顺磁性物质沉积,实验室检查后发现是锰。病史、临床检查和检查结果指向因过量摄入含锰物质(即红茶)导致的获得性高锰血症诊断。他接受了对症治疗和螯合疗法(依地酸钙钠)。在随访的第六个月,观察到舞蹈症完全缓解、构音障碍消失且僵硬部分改善。他的认知衰退和行为异常有所改善。

讨论

这可能是第一例报告的获得性高锰血症病例,表现为不对称舞蹈症和认知功能障碍的组合,并具有非典型影像学特征。临床表现类似于亨廷顿舞蹈症。我们强调了一种看似“良性”的非酒精饮料(即红茶)对脑代谢的潜在有害影响。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2633/7597573/6975f33b1793/tohm-10-1-537-g1.jpg

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