Myoclonic involuntary movement associated with chronic manganese poisoning

doi:10.1016/S0022-510X(02)00111-9

Journal of the Neurological Sciences

Volume 199, Issues 1–2, 15 July 2002, Pages 93-96

https://doi.org/10.1016/S0022-510X(02)00111-9 Get rights and content

Abstract

We report a 17-year-old man showing myoclonic involuntary movement (IVM) associated with chronic manganese (Mn) poisoning. The patient, a welder, showed myoclonic IVM mainly in the right upper and lower extremities, elevated levels of Mn in the blood and hair and high-intensity signals in the globus pallidus on T1-weighted MR images. Chelation therapy resulted in improvement of the myoclonic IVM and MRI abnormalities. This is the first report of Mn poisoning characterized by myoclonic IVM without parkinsonism.

Introduction

The most characteristic manifestations of manganese (Mn) poisoning are extrapyramidal tract syndrome, primarily parkinsonism [2], [4], [7], [10]. We recently encountered a patient with chronic Mn poisoning due to electric arc welding, who showed myoclonic involuntary movement (IVM), but no parkinsonism.

Section snippets

Case report

The patient was a 17-year-old man with chief complaints of IVM of the right upper and lower extremities. His past or family history was not remarkable. He had been engaged in electric arc welding for about 2 years.

On February 14, 2000, the patient's right arm began to tremble while he was welding. The trembling subsided in about one hour. Thereafter, the patient became to frequently suffer from the trembling of the right arm and leg, and he stopped his work of welding. The drug therapies

Discussion

Our patient was characterized by myoclonic IVM mainly in the right upper and lower extremities, hyperintense globus pallidus on T1-weighted MR images, and elevated Mn levels in the blood and hair. The chelation therapy resulted in excretion of Mn in the urine, normalization of Mn levels in the blood and remarkable improvement of the myoclonic symptoms and MRI abnormalities. These findings indicate that the patient had been suffering from Mn poisoning and that the myoclonic IVM was associated

References (14)

E.P. Brouillet et al.
Manganese injection into the rat striatum produces excitotoxic lesions by impairing energy metabolism
Exp. Neurol.
(1993)
N.A. Larsen et al.
Distribution of arsenic, manganese, and selenium in the human brain in chronic renal insufficiency, Parkinson's disease, and amyotrophic lateral sclerosis
J. Neurol. Sci.
(1981)
M.C. Newland et al.
Visualizing manganese in the primate basal ganglia with magnetic resonance imaging
Exp. Neurol.
(1989)
R.G. Feldman
Manganese
S. Ikeda et al.
Manganese deposition in the globus pallidus in patients with biliary atresia
Transplantation
(2000)
N. Inoue et al.
Neurological aspects in human exposures to manganese
Y. Kim et al.
Increase in signal on T1-weighted magnetic resonance images in asymptomatic manganese-exposed workers
Neurotoxicology
(1999)

There are more references available in the full text version of this article.

Cited by (71)

Copper(II) complexes of aminopolycarboxylate ligands with N<inf>2</inf>O<inf>2</inf>, N<inf>2</inf>O<inf>3</inf> and N<inf>2</inf>O<inf>4</inf> donor sets. The relationship between the ligand structure and molecular geometry of the complex
2021, Journal of Molecular Structure
The article presents an overview of the results achieved during the last several decades on the synthesis and structural characterization of copper(II) complexes with different aminopolycarboxylates. Considering the structural varieties in designing aminopolycarboxylate ligands and significant stereochemical flexibility of the corresponding copper(II) complexes to adopt a wide range of coordination geometries, this review is mainly focused on the results related to the crystallographic characterization of the synthesized complexes. The ligands used for the synthesis of copper(II) complexes described in this article contain two amine nitrogen atoms and different number of the carboxylate oxygen donor atoms (N₂O₂, N₂O₃ and N₂O₄-type of ligands). Giving the importance to the biological relevance of the Cu(II) ion and broad aspects of potential application of aminopolycarboxylate ligands in human practice, the presented results could have some important implications for coordination chemists dealing with structural properties of aminopolycarboxylate-copper(II) complexes and their potential medicinal and biological applications.
The impact of manganese on neurotransmitter systems
2020, Journal of Trace Elements in Medicine and Biology
Citation Excerpt :
Humans are easily exposed to Mn through air, soil and, consequently, water, being the main source of intoxication identified due to occupational exposure [9,55]. Industry professionals in battery production, welding and mining operations are constantly exposed to Mn by inhalation, with reported neurotoxicity in these workers [27,56–59]. Neurological damage with Mn accumulation in various areas of the midbrain such as globus pallidus, substantia nigra, subthalamic nucleus, putamen, caudate nucleus and dentate nucleus has been described [31,60,61].
Manganese (Mn) is a metal ubiquitously present in nature and essential for many living organisms. As a trace element, it is required in small amounts for the proper functioning of several important enzymes, and reports of Mn deficiency are indeed rare.
This mini-review will cover aspects of Mn toxicokinetics and its impact on brain neurotransmission, as well as its Janus-faced effects on humans and other animal’s health.
The estimated safe upper limit of intracellular Mn for physiological function is in anarrow range of 20–53 μM.Therefore, intake of higher levels of Mn and the outcomes, especially to the nervous system, have been well documented.
The metal affects mostly the brain by accumulating in specific areas, altering cognitive functions and locomotion, thus severely impacting the health of the exposed organisms.
Ethanol increases manganese—Induced spatial learning and memory deficits via oxidative/nitrosative stress induced p53 dependent/independent hippocampal apoptosis
2019, Toxicology
Citation Excerpt :
Early symptoms of Mn toxicity are compulsive behavior, emotional instability, slowing motor functions, intellectual and memory impairment (Mergler and Baldwin, 1997; Blecharz-Klin et al., 2012; Lucchini et al., 2012). Environmental and occupational exposure to Mn occurs mainly in Mn alloys mining, steel welding, smelters (Crossgrove and Zheng, 2004; Ono et al., 2002) and in the production of dye, paints, fertilizers, lacquers, fireworks, dry cell batteries, fungicides, wood preservatives and rubber (Blecharz-Klin et al., 2012) and other industries. It has been reported that Mn induced neurotoxicity involved diminished learning abilities and lower scores in many neurobehavioral tests (He et al., 1994; Zhang et al., 1995; Menezes-Filho et al., 2009).
Manganese (Mn) is an essential nutrient element. However, Mn is causing great environmental and occupational exposure health risk concern globally, even high rate of alcohol consumption. There is dearth of scientific information on the interaction of manganese (Mn) and ethanol (EtOH) on hippocampal functions. This study was designed to investigate the effect of EtOH on Mn – induced hippocampal toxicity with special reference to spatial learning and memory and its underlying mechanism in adults male Wistar Rats. Rats were exposed to Mn alone at 30 mg/kg or co-expose with EtOH at 1.25 and 5 g/kg body weight by oral gavage for 35 consecutive days. Morris Water Maze task was used to assessed spatial learning and memory. Subsequently, oxidative/nitrosative stress, neuro-inflammation (myeloperoxidase and cyclooxygenase-2) and protein expression of apoptotic proteins (p53 and Bax), active executioner caspase (caspase-3) and B – cell lymphoma – 2 (Bcl – 2) markers in the hippocampus were investigated. The results indicate that Mn and EtOH exposure induces spatial learning and memory deficits, increase oxidative/nitrosative stress, neuro-inflammation resulting in enhanced hippocampal apoptosis. Moreover, the results indicated that Mn co-exposure with EtOH at 1.25 and 5 g/kg body weight further exacerbates neurotoxicity in rat hippocampus when compared with single dose of Mn and EtOH alone. Collectively, EtOH increases Mn – induced oxidative/nitrosative stress, neuro-inflammation and hippocampal apoptosis via mechanism involving oxidative damages of cellular constituents, neuronal inflammation and subsequent upregulation of Bax and caspase-3 and downregulation of Bcl-2 protein expression via p53 dependent/independent pathways to induced hippocampal apoptosis associated with impaired spatial learning and memory.
Effect of flash flood on trace metal pollution in the groundwater - Wadi Baysh Basin, western Saudi Arabia
2018, Journal of African Earth Sciences
This study focused on the impact of flash flood (FF) on trace metal pollution in the groundwater, Wadi Baysh Basin, Western Saudi Arabia using spatial interpolations, integrating pollution indices and statistical analysis. Groundwater (n = 182) is neutral to alkaline in nature and the order of mean metals before and after FF are Sr > Ba > B > Fe > Al > V > Mn > Cr > Cu=Pb > Mo > Zn and Sr > Al > B > Fe > V > Ba > Cr > Cu > Pb > Mn > Mo > Zn > Ni > Cd, respectively. Enhancement (Al, Cr, Cu, Mn, Mo, Pb and V) and depletion (B, Ba, Fe, Sr) of trace metals are noticed in the groundwater after FF, which is due to dilution, dissolution/desorption, flushing of weathered layer, saline sources and surface input. Before and after FF, 57–63%(Pb), 55–56%(V), 43-16%(B), 27-0%(Ba), 15-5%(Sr), 6-3%(Fe), 0–16%(Al), 0–9%(Cr) and 1–2%(Mn) of groundwater samples, respectively, exceeded the drinking water standards. Coastal wells have elevated B, Sr and Mn concentration due to saline water intrusion. Integrated pollution evaluation indices (Heavy metal pollution index (HPI), Contamination index (C_d) and Heavy metal evaluation index (HEI)) reveal that number of samples in the unsuitable classes are increased after FF event (HPI: 13%–55%; C_d: 11%–38%; HEI: 5%–19%) and groundwater quality is degraded due to the trace metals enhancement after FF especially in the downstream wells. Wells in the upstream is flushed by FF recharge and groundwater flow accumulated the metals in the downstream and coastal region. This study demonstrates that metal accumulation and mobility are regulated by the groundwater flow in the study region. Pearson correlation analysis and Principle component analysis also justify that water chemistry is influenced by the saline sources, flushing of weathered layer, natural processes and anthropogenic inputs. Continuous groundwater monitoring is an important task to protect and manage valuable groundwater resources in the arid region.
Nonmotor Symptoms in Vascular and Other Secondary Parkinsonism
2017, International Review of Neurobiology
Citation Excerpt :
The doses of Mn and exposure time that produce them are not precisely known (Jain & Ferrando, 2011; Sikk et al., 2011). Repeated courses of calcium and sodium EDTA that accelerates manganese excretion may cause regress of neurological symptoms (Herrero Hernandez et al., 2006; Ono et al., 2002). In patients with ephedrone encephalopathy, CaNa2EDTA decreases a probability of the further disease progress, though do not contribute significantly to symptoms regress (Levin, 2005; Selikhova et al., 2008).
Vascular parkinsonism (VP) is a relatively frequent variant of secondary parkinsonism caused by ischemic or hemorrhagic lesions of basal ganglia, midbrain, or their links with frontal cortex. According to different investigations, various forms of cerebrovascular disease cause 1%–15% of parkinsonism cases. Nonmotor symptoms are frequently found in VP and may negatively influence on quality of life. However, nonmotor symptoms such as hallucinations, orthostatic hypotension, REM-sleep behavior disorder, and anosmia are rarely revealed in VP, which may be noted to another diagnosis or mixed pathology. Clinical value of nonmotor symptoms in normal pressure hydrocephalus, toxic, and drug-induced parkinsonism is also discussed.
Myoclonus in the critically ill: Diagnosis, management, and clinical impact
2016, Clinical Neurophysiology
Myoclonus is the second most common involuntary non-epileptic movement in intensive care units following tremor-like gestures. Although there are several types of myoclonus, they remain underappreciated, and their diagnostic and prognostic associations are largely ignored.
This review discusses clinical, electrophysiological, neuroanatomical, and neuroimaging characteristics of different types of myoclonus in critically ill adults along with their prognostic impact and treatment options.
Myoclonus is characterized by a sudden, brief, and sometimes repetitive muscle contraction of body parts, or a brief and sudden cessation of tonic muscle innervation followed by a rapid recovery of tonus. Myoclonus can resemble physiologic and other pathologic involuntary movements. Neurologic injuries, anesthetics, and muscle relaxants interfere with the typical appearance of myoclonus. Identifying “real myoclonus” and determining the neuroanatomical origin are important, as treatment responses depend on the involved neuroanatomical structures. The identification of the type of myoclonus, the involved neuroanatomical structures, and the associated illnesses is essential to direct treatment.
In conclusion, the combined clinical, electrophysiological, and neuroradiological examination reliably uncovers the neuroanatomical sources and the pathophysiology of myoclonus. Recognizing cortical myoclonus is critical, as it is treatable and may progress to generalized convulsive seizures or status epilepticus.

View all citing articles on Scopus

View full text

Short communicationMyoclonic involuntary movement associated with chronic manganese poisoning

Abstract

Introduction

Section snippets

Case report

Discussion

Exp. Neurol.

J. Neurol. Sci.

Exp. Neurol.

Manganese

Manganese deposition in the globus pallidus in patients with biliary atresia

Transplantation

Neurological aspects in human exposures to manganese

Increase in signal on T1-weighted magnetic resonance images in asymptomatic manganese-exposed workers

Neurotoxicology

Short communication
Myoclonic involuntary movement associated with chronic manganese poisoning