Publication: Loss of slc39a14 causes simultaneous manganese hypersensitivity and deficiency in zebrafish
Loss of slc39a14 causes simultaneous manganese hypersensitivity and deficiency in zebrafish
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Tuschl, K., White, R. J., Trivedi, C., Valdivia, L. E., Niklaus, S., Bianco, I. H., Dadswell, C., González-Méndez, R., Sealy, I. M., Neuhauss, S. C. F., Houart, C., Rihel, J., Wilson, S. W., & Busch-Nentwich, E. M. (2022). Loss of slc39a14 causes simultaneous manganese hypersensitivity and deficiency in zebrafish. Disease Models & Mechanisms, 15(6), dmm044594. https://doi.org/10.1242/dmm.044594
Abstract
Abstract
Abstract
Manganese neurotoxicity is a hallmark of Hypermanganesemia with Dystonia 2, an inherited manganese transporter defect caused by mutations in SLC39A14. To identify novel potential targets of manganese neurotoxicity we performed transcriptome analysis of slc39a14-/- mutant zebrafish unexposed and exposed to MnCl2. Differentially expressed genes mapped to the central nervous system and eye, and pathway analysis suggested that calcium dyshomeostasis and activation of the unfolded protein response are key features of manganese neurotoxicit
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- Tuschl, Karin
- White, Richard J
- Trivedi, Chintan
- Valdivia, Leonardo E
- Niklaus, Stephanie
- Bianco, Isaac H
- Dadswell, Chris
- González-Méndez, Ramón
- Sealy, Ian M
- Houart, Corinne
- Rihel, Jason
- Wilson, Stephen W
- Busch-Nentwich, Elisabeth M
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Tuschl, K., White, R. J., Trivedi, C., Valdivia, L. E., Niklaus, S., Bianco, I. H., Dadswell, C., González-Méndez, R., Sealy, I. M., Neuhauss, S. C. F., Houart, C., Rihel, J., Wilson, S. W., & Busch-Nentwich, E. M. (2022). Loss of slc39a14 causes simultaneous manganese hypersensitivity and deficiency in zebrafish. Disease Models & Mechanisms, 15(6), dmm044594. https://doi.org/10.1242/dmm.044594
