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Effect of Chelation Therapy on a Korean Patient With Brain Manganese Deposition Resulting From a Compound Heterozygous Mutation in the SLC39A14 Gene
Jae-Hyeok Lee, Jin-Hong Shin
J Mov Disord. 2022;15(2):171-174.   Published online March 22, 2022
DOI: https://doi.org/10.14802/jmd.21143
  • 3,832 View
  • 201 Download
  • 9 Web of Science
  • 8 Crossref
AbstractAbstract PDF
Mutations in the manganese transporter gene SLC39A14 lead to inherited disorders of manganese metabolism. Chelation therapy with edetate calcium disodium (CaNa2EDTA) is known to effectively reduce manganese deposition. We describe the first identified Korean case of SLC39A14-associated manganism and the treatment response to a 5-year chelation therapy. An 18-year-old female presented with childhood-onset dystonia. Magnetic resonance imaging showed T1 hyperintensity throughout the basal ganglia, brainstem, cerebellum, cerebral and cerebellar white matter, and pituitary gland. Blood manganese levels were elevated, and whole-exome sequencing revealed compound heterozygous mutations in SLC39A14. Treatment with intravenous CaNa2EDTA led to a significant reduction in serum manganese levels and T1 hyperintensities. However, her dystonia improved insignificantly. Hence, early diagnosis of this genetic disorder is essential because it is potentially treatable. Even though our treatment did not significantly reverse the establish deficits, chelation therapy could have been more effective if it was started at an earlier stage of the disease.

Citations

Citations to this article as recorded by  
  • Magnetic Resonance Imaging and Manganism: A Narrative Review and Laboratory Recommendations
    Michal Majewski, Karolina Piwko, Michal Ordak, Elzbieta Muszynska, Tadeusz Nasierowski, Magdalena Bujalska-Zadrozny
    Journal of Clinical Medicine.2024; 13(10): 2823.     CrossRef
  • Expression of Manganese Transporters ZIP8, ZIP14, and ZnT10 in Brain Barrier Tissues
    Shannon Morgan McCabe, Ningning Zhao
    International Journal of Molecular Sciences.2024; 25(19): 10342.     CrossRef
  • Recent progress toward understanding the role of ZIP14 in regulating systemic manganese homeostasis
    Shannon McCabe, Kirsten Limesand, Ningning Zhao
    Computational and Structural Biotechnology Journal.2023; 21: 2332.     CrossRef
  • Metal-ion transporter SLC39A8 is required for brain manganese uptake and accumulation
    Qingli Liu, Supak Jenkitkasemwong, Tamanna Afrin Prami, Shannon Morgan McCabe, Ningning Zhao, Shintaro Hojyo, Toshiyuki Fukada, Mitchell D. Knutson
    Journal of Biological Chemistry.2023; 299(8): 105078.     CrossRef
  • Loss of slc39a14 causes simultaneous manganese hypersensitivity and deficiency in zebrafish
    Karin Tuschl, Richard J. White, Chintan Trivedi, Leonardo E. Valdivia, Stephanie Niklaus, Isaac H. Bianco, Chris Dadswell, Ramón González-Méndez, Ian M. Sealy, Stephan C. F. Neuhauss, Corinne Houart, Jason Rihel, Stephen W. Wilson, Elisabeth M. Busch-Nent
    Disease Models & Mechanisms.2022;[Epub]     CrossRef
  • Hereditary Disorders of Manganese Metabolism: Pathophysiology of Childhood-Onset Dystonia-Parkinsonism in SLC39A14 Mutation Carriers and Genetic Animal Models
    Alexander N. Rodichkin, Tomás R. Guilarte
    International Journal of Molecular Sciences.2022; 23(21): 12833.     CrossRef
  • Pathophysiological studies of aging Slc39a14 knockout mice to assess the progression of manganese-induced dystonia-parkinsonism
    Alexander N. Rodichkin, Melissa K. Edler, Jennifer L. McGlothan, Tomás R. Guilarte
    NeuroToxicology.2022; 93: 92.     CrossRef
  • Mechanisms of manganese-induced neurotoxicity and the pursuit of neurotherapeutic strategies
    Edward Pajarillo, Ivan Nyarko-Danquah, Alexis Digman, Harpreet Kaur Multani, Sanghoon Kim, Patric Gaspard, Michael Aschner, Eunsook Lee
    Frontiers in Pharmacology.2022;[Epub]     CrossRef
Review Article
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Manganese and Movement Disorders: A Review
Dinkar Kulshreshtha, Jacky Ganguly, Mandar Jog
J Mov Disord. 2021;14(2):93-102.   Published online April 6, 2021
DOI: https://doi.org/10.14802/jmd.20123
  • 13,219 View
  • 608 Download
  • 24 Web of Science
  • 27 Crossref
AbstractAbstract PDF
Scientific and technological advances achieved with industrial expansion have led to an ever-increasing demand for heavy metals. This demand has, in turn, led to increased contamination of soil, water and air with these metals. Chronic exposure to metals may be detrimental not only to occupational workers but also to the nonoccupational population exposed to these metals. Manganese (Mn), a commonly used heavy metal, is an essential cofactor for many enzymatic processes that drive biological functions. However, it is also a potential source of neurotoxicity, particularly in the field of movement disorders. The typical manifestation of Mn overexposure is parkinsonism, which may be difficult to differentiate from the more common idiopathic Parkinson’s disease. In addition to environmental exposure to Mn, other potential etiologies causing hypermanganesemia include systemic health conditions, total parenteral nutrition and genetic mutations causing Mn dyshomeostasis. In this review, we critically analyze Mn and discuss its sources of exposure, pathophysiology and clinical manifestations. We have highlighted the global public health impact of Mn and emphasize that movement disorder specialists should record a detailed social and occupational history to ensure that a toxic etiology is not misdiagnosed as a neurodegenerative disease. In the absence of a definite therapeutic option, early diagnosis and timely institution of preventive measures are the keys to managing its toxic effects.

Citations

Citations to this article as recorded by  
  • The cGAS−STING-mediated ROS and ferroptosis are involved in manganese neurotoxicity
    Zhimin Zhang, Jirui Yang, Qiongli Zhou, Shiyin Zhong, Jingjing Liu, Xin Zhang, Xuhong Chang, Hui Wang
    Journal of Environmental Sciences.2025; 152: 71.     CrossRef
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    Oluwaseun Mary Oladeji, Boikanyo Genneyrolter Kopaopa, Liziwe Lizbeth Mugivhisa, Joshua Oluwole Olowoyo
    Biological Trace Element Research.2024; 202(2): 778.     CrossRef
  • Neuroprotective Strategies and Cell-Based Biomarkers for Manganese-Induced Toxicity in Human Neuroblastoma (SH-SY5Y) Cells
    Catherine M. Cahill, Sanjan S. Sarang, Rachit Bakshi, Ning Xia, Debomoy K. Lahiri, Jack T. Rogers
    Biomolecules.2024; 14(6): 647.     CrossRef
  • Update: Protective and risk factors for Parkinson disease
    Nikolas Grotewold, Roger L. Albin
    Parkinsonism & Related Disorders.2024; 125: 107026.     CrossRef
  • Sex-specific associations of a ferroalloy metal mixture with motor function in Italian adolescents
    Alexa Friedman, Samantha Schildroth, Victoria Fruh, Maxine H. Krengel, Yorghos Tripodis, Donatella Placidi, Roberta F. White, Roberto G. Lucchini, Donald R. Smith, Robert O. Wright, Megan K. Horton, Birgit Claus Henn
    Environmental Epidemiology.2024; 8(4): e321.     CrossRef
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    Norlinah Mohamed Ibrahim, Chin Hsien Lin
    Parkinsonism & Related Disorders.2024; 129: 107100.     CrossRef
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    Edward Pajarillo, Sanghoon Kim, Alexis Digman, Itunu Ajayi, Ivan Nyarko-Danquah, Deok-Soo Son, Michael Aschner, Eunsook Lee
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    Frontiers in Neurology.2024;[Epub]     CrossRef
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    Veronica Manescu (Paltanea), Iulian Antoniac, Gheorghe Paltanea, Iosif Vasile Nemoianu, Aurel George Mohan, Aurora Antoniac, Julietta V. Rau, Stefan Alexandru Laptoiu, Petruta Mihai, Horia Gavrila, Abdel Rahim Al-Moushaly, Alin Danut Bodog
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  • Vitaminlerin ve Metallerin Çift Yönlü Etkileri: Sağlık, Mutajenite ve Antimutajenite Üzerine Bir İnceleme
    Eda Delik, Burcu Emine Tefon Öztürk, Bülent Kaya
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  • Effect of fish-heavy metals contamination on the generation of reactive oxygen species and its implications on human health: a review
    Yuxia Wang, Anwar Noman, Chao Zhang, Wedad Q. AL-Bukhaiti, Sherif M. Abed
    Frontiers in Marine Science.2024;[Epub]     CrossRef
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    Suetmui Yu, Ningning Zhao
    International Journal of Molecular Sciences.2023; 24(6): 5962.     CrossRef
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    Mohiudeen Alikunju, Nafeesathu Misiriyyah , Shaikh Sayeed Iqbal, Maria Khan
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  • Associations of ambient manganese exposure with brain gray matter thickness and white matter hyperintensities
    Shinyoung Woo, Young Noh, Sang-Baek Koh, Seung-Koo Lee, Jung il Lee, Ho Hyun Kim, Sun- Young Kim, Jaelim Cho, Changsoo Kim
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    Muxue Lu, Ping Deng, Lingling Yang, Xue Wang, Xiang Mei, Chao Zhou, Mengyan Chen, Zhou Zhou, Huifeng Pi, Lichuan Wu, Zhengping Yu
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    David C. Dorman
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    Qifeng Zhu, Fan Jiang, Yuanbo Song, Lili Lu, Fajian He, Shuqi Huang, Zhaoying Huang, Jing Yao, Ningning Lei, Jianmin Huang, Shijin Lu
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  • Trace Elements Open a New Direction for the Diagnosis of Atherosclerosis
    Heyu Meng, Jianjun Ruan, Yanqiu Chen, Zhaohan Yan, Jinsha Liu, Xue Wang, Xin Meng, Jingru Wang, Qiang Zhang, Xiangdong Li, Fanbo Meng
    Reviews in Cardiovascular Medicine.2023;[Epub]     CrossRef
  • Differentiating Wild and Apiary Honey by Elemental Profiling: a Case Study from Mangroves of Indian Sundarban
    Tanushree Gaine, Praveen Tudu, Somdeep Ghosh, Shouvik Mahanty, Madhurima Bakshi, Nabanita Naskar, Souparna Chakrabarty, Subarna Bhattacharya, Swati Gupta Bhattacharya, Kashinath Bhattacharya, Punarbasu Chaudhuri
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  • Environmental Impact on the Epigenetic Mechanisms Underlying Parkinson’s Disease Pathogenesis: A Narrative Review
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    Brain Sciences.2022; 12(2): 175.     CrossRef
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    Journal of Clinical Medicine.2022; 11(4): 1147.     CrossRef
  • Can therapeutic plasma exchange be life-saving in life-threatening manganese intoxication?
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    Transfusion and Apheresis Science.2022; 61(4): 103417.     CrossRef
  • Manganese‐induced parkinsonism responsive to intranasal insulin: A case report
    Mehri Salari, Masoud Etemadifar, Leila Dargahi, Neda Valian, Malihe Rezaee
    Clinical Case Reports.2022;[Epub]     CrossRef
  • The potential convergence of NLRP3 inflammasome, potassium, and dopamine mechanisms in Parkinson’s disease
    Adrianne F. Pike, Ildikò Szabò, Robert Veerhuis, Luigi Bubacco
    npj Parkinson's Disease.2022;[Epub]     CrossRef
  • Effect of Chelation Therapy on a Korean Patient With Brain Manganese Deposition Resulting From a Compound Heterozygous Mutation in the SLC39A14 Gene
    Jae-Hyeok Lee, Jin-Hong Shin
    Journal of Movement Disorders.2022; 15(2): 171.     CrossRef
  • Manganese chloride (MnCl2) induced novel model of Parkinson’s disease in adult Zebrafish; Involvement of oxidative stress, neuroinflammation and apoptosis pathway
    Abhishek.P.R. Nadig, Bader Huwaimel, Ahmed Alobaida, El-Sayed Khafagy, Hadil Faris Alotaibi, Afrasim Moin, Amr Selim Abu Lila, Suman, Sahyadri. M, K.L. Krishna
    Biomedicine & Pharmacotherapy.2022; 155: 113697.     CrossRef
  • Çalışma Yaşamında Manganez Maruz Kalımının Sağlık Etkileri ve Parkinsonizm
    Zehra GÖK METİN, Abdulsamet SANDAL, Ali Naci YILDIZ
    Karaelmas İş Sağlığı ve Güvenliği Dergisi.2021; 5(2): 147.     CrossRef

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