University of Groningen, University Medical Center Groningen, Department of Neurology, Groningen, The Netherlands.
University of Groningen, University Medical Center Groningen, Department of Clinical Neurophysiology, Groningen, The Netherlands.
Mov Disord. 2018 Dec;33(12):1844-1856. doi: 10.1002/mds.27484. Epub 2018 Nov 28.
Inborn errors of metabolism in adults are still largely unexplored. Despite the fact that adult-onset phenotypes have been known for many years, little attention is given to these disorders in neurological practice. The adult-onset presentation differs from childhood-onset phenotypes, often leading to considerable diagnostic delay. The identification of these patients at the earliest stage of disease is important, given that early treatment may prevent or lessen further brain damage. Neurological and psychiatric symptoms occur more frequently in adult forms. Abnormalities of eye movements are also common and can be the presenting sign. Eye movement disorders can be classified as central or peripheral. Central forms are frequently observed in lysosomal storage disorders, whereas peripheral forms are a key feature of mitochondrial disease. Furthermore, oculogyric crisis is an important feature in disorders affecting dopamine syntheses or transport. Ocular motor disorders are often not reported by the patient, and abnormalities can be easily overlooked in a general examination. In adults with unexplained psychiatric and neurological symptoms, a special focus on examination of eye movements can serve as a relatively simple clinical tool to detect a metabolic disorder. Eye movements can be easily quantified and analyzed with video-oculography, making them a valuable biomarker for following the natural course of disease or the response to therapies. Here, we review, for the first time, eye movement disorders that can occur in inborn errors of metabolism, with a focus on late-onset forms. We provide a step-by-step overview that will help clinicians to examine and interpret eye movement disorders. © 2018 The Authors. Movement Disorders published by Wiley Periodicals, Inc. on behalf of International Parkinson and Movement Disorder Society.
成人先天性代谢缺陷仍然在很大程度上未被探索。尽管多年来人们已经知道成人发病表型,但神经科实践中很少关注这些疾病。成人发病表现与儿童发病表型不同,常常导致相当大的诊断延迟。鉴于早期治疗可能预防或减轻进一步的脑损伤,因此在疾病的最早阶段识别这些患者非常重要。神经系统和精神症状在成人形式中更为常见。眼球运动异常也很常见,并且可能是首发症状。眼球运动障碍可分为中枢性或周围性。中枢形式常发生在溶酶体贮积症中,而周围形式是线粒体疾病的一个关键特征。此外,动眼危象是影响多巴胺合成或转运的疾病的重要特征。眼球运动障碍通常不会被患者报告,并且在一般检查中很容易忽略异常。对于原因不明的精神和神经系统症状的成年人,特别关注眼球运动检查可以作为一种相对简单的临床工具,以检测代谢紊乱。可以使用视频眼动描记术轻松地对眼球运动进行量化和分析,使其成为跟踪疾病自然病程或对治疗反应的有价值的生物标志物。在这里,我们首次回顾了可发生于先天性代谢缺陷的眼球运动障碍,重点关注迟发性疾病。我们提供了一个逐步概述,将帮助临床医生检查和解释眼球运动障碍。© 2018 作者。运动障碍由 Wiley 期刊出版公司代表国际帕金森和运动障碍协会出版。