Khan Alisha, Hingre Jonathan, Dhamoon Amit S
Department of Medicine, SUNY Upstate Medical University, Syracuse, NY, USA.
College of Medicine, SUNY Upstate Medical University, Syracuse, NY, USA.
Case Rep Neurol Med. 2020 Apr 23;2020:9484028. doi: 10.1155/2020/9484028. eCollection 2020.
Manganese accumulation in the central nervous system creates clinical symptoms of cognitive dysfunction, behavioral changes, and movement disorders resembling Parkinson's disease. Radiographic features of this rare clinical entity include symmetric T1 hyperintensities in the bilateral globus pallidi, with corresponding hypointensities on T2-weighted images. Total parenteral nutrition (TPN) is an increasingly used potentially lifesaving therapy for patients who cannot tolerate enteral nutrition. However, when used over a period of several weeks to months, its associated risks and complications carry significant morbidity and mortality. One of the more rare complications of TPN use is manganese toxicity. We provided care for a 38-year-old female on chronic TPN who presented to the hospital with Parkinsonian features, confusion, falls, and lethargy. MRI brain showed T1 hyperintensities in the bilateral globus pallidi, which were attributed to manganese toxicity from chronic TPN use. Supporting evidence for this rare entity included decreased signal intensity in the bilateral globus pallidi on T2-weighted images and T1 hyperintensities in the substantia nigra. With antifungal treatment and permanent cessation of TPN, her mentation and neurological symptoms began to improve within a week. Repeat MRI brain performed one month after discontinuation of TPN revealed improvement of the T1 hyperintensities in the bilateral globus pallidi. Our objective in presenting this case is to highlight manganese neurotoxicity as a rare complication of TPN in a patient without known hepatic dysfunction and to emphasize the importance of routinely monitoring patients for the possible adverse effects of chronic TPN. Our case is among the handful of published cases in which a patient without known liver dysfunction, which is the primary organ responsible for manganese elimination from the body, developed manganese neurotoxicity.
锰在中枢神经系统中的蓄积会引发认知功能障碍、行为改变以及类似帕金森病的运动障碍等临床症状。这种罕见临床病症的影像学特征包括双侧苍白球对称性T1高信号,在T2加权图像上相应区域呈低信号。全胃肠外营养(TPN)是一种越来越多地用于无法耐受肠内营养患者的潜在救命疗法。然而,当持续使用数周或数月时,其相关风险和并发症会带来显著的发病率和死亡率。TPN使用中较为罕见的并发症之一是锰中毒。我们为一名38岁长期接受TPN治疗的女性提供了护理,她因出现帕金森病特征、意识模糊、跌倒和嗜睡而入院。脑部MRI显示双侧苍白球T1高信号,这归因于长期使用TPN导致的锰中毒。支持这一罕见病症的证据包括T2加权图像上双侧苍白球信号强度降低以及黑质T1高信号。经过抗真菌治疗并永久停用TPN后,她的精神状态和神经症状在一周内开始改善。停用TPN一个月后复查脑部MRI显示双侧苍白球T1高信号有所改善。我们呈现此病例的目的是强调锰神经毒性作为TPN在无已知肝功能障碍患者中罕见并发症的情况,并强调常规监测患者慢性TPN可能产生的不良反应的重要性。我们的病例是少数已发表的病例之一,即一名无已知肝功能障碍(肝功能障碍是身体清除锰的主要器官)的患者发生了锰神经毒性。