@article{213d90d945e84ab084e067c8e920a386,
title = "Maintaining Translational Relevance in Animal Models of Manganese Neurotoxicity",
abstract = "Manganese is an essential metal, but elevated brain Mn concentrations produce a parkinsonian-like movement disorder in adults and fine motor, attentional, cognitive, and intellectual deficits in children. Human Mn neurotoxicity occurs owing to elevated exposure from occupational or environmental sources, defective excretion (e.g., due to cirrhosis), or loss-of-function mutations in the Mn transporters solute carrier family 30 member 10 or solute carrier family 39 member 14. Animal models are essential to study Mn neurotoxicity, but in order to be translationally relevant, such models should utilize environmentally relevant Mn exposure regimens that reproduce changes in brain Mn concentrations and neurological function evident in human patients. Here, we provide guidelines for Mn exposure in mice, rats, nematodes, and zebrafish so that brain Mn concentrations and neurobehavioral sequelae remain directly relatable to the human phenotype.",
keywords = "Caenorhabditis elegans, SLC30A10, SLC39A14, ZIP14, ZnT10, animal models, manganese, neurotoxicity, zebrafish",
author = "Taylor, {Cherish A.} and Karin Tuschl and Nicolai, {Merle M.} and Julia Bornhorst and Priscila Gubert and Var{\~a}o, {Alexandre M.} and Michael Aschner and Smith, {Donald R.} and Somshuvra Mukhopadhyay",
note = "Funding Information: Supported by National Institute of Environmental Health Sciences grants R01-ES024812 and R01-ES024812S1 (to SM) and R01-ES07331 and R01-ES10563 (to MA); National Institute for Health Research, the Academy of Medical Sciences, Action Medical Research, and Great Ormond Street Hospital Children{\textquoteright}s Charity UK (to KT); German Research Foundation grant BO 4103/2-1 and DFG Research Unit TraceAge FOR 2558 (to MMN and JB). Author disclosures: The authors report no conflicts of interest. CAT and KT contributed equally to this work. Address correspondence to KT (e-mail: k.tuschl@ucl.ac.uk) or SM (e-mail: som@austin.utexas.edu). Abbreviations used: CRISPR, clustered regularly interspaced short palindromic repeats; DAergic, dopaminergic; dpf, days postfertilization; HMNDYT, Hyper-manganesemia with Dystonia; ICP-MS, inductively coupled plasma mass spectrometry; PND, post natal day; SLC30A10, solute carrier family 30 member 10; SLC39A14, solute carrier family 39 member 14; TH, tyrosine hydroxylase. Publisher Copyright: {\textcopyright} The Author(s) 2020.",
year = "2020",
month = jun,
day = "1",
doi = "10.1093/jn/nxaa066",
language = "English (US)",
volume = "150",
pages = "1360--1369",
journal = "Journal of Nutrition",
issn = "0022-3166",
publisher = "American Society for Nutrition",
number = "6",
}