<> <http://www.w3.org/2000/01/rdf-schema#comment> "The repository administrator has not yet configured an RDF license."^^<http://www.w3.org/2001/XMLSchema#string> . <> <http://xmlns.com/foaf/0.1/primaryTopic> <https://discovery.ucl.ac.uk/id/eprint/10148179> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://www.w3.org/1999/02/22-rdf-syntax-ns#type> <http://purl.org/ontology/bibo/AcademicArticle> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://www.w3.org/1999/02/22-rdf-syntax-ns#type> <http://purl.org/ontology/bibo/Article> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/dc/terms/title> "Loss of slc39a14 causes simultaneous manganese hypersensitivity and deficiency in zebrafish"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/ontology/bibo/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 neurotoxicity. Consistent with this interpretation, MnCl2 exposure led to decreased whole animal calcium levels, locomotor defects and changes in neuronal activity within the telencephalon and optic tectum. In accordance with reduced tectal activity, slc39a14-/- zebrafish showed changes in visual phototransduction gene expression, absence of visual background adaptation and a diminished optokinetic reflex. Finally, numerous differentially expressed genes in mutant larvae normalised upon MnCl2 treatment indicating that, in addition to neurotoxicity, manganese deficiency is present either subcellularly or in specific cells or tissues. Overall, we assembled a comprehensive set of genes that mediate manganese-systemic responses and found a highly correlated and modulated network associated with calcium dyshomeostasis and cellular stress."^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/dc/terms/date> "2022-05-06" . <https://discovery.ucl.ac.uk/id/document/1469778> <http://www.w3.org/1999/02/22-rdf-syntax-ns#type> <http://purl.org/ontology/bibo/Document> . <https://discovery.ucl.ac.uk/id/org/ext-d2596156ee130bb0a805ff1203c24d37> <http://www.w3.org/1999/02/22-rdf-syntax-ns#type> <http://xmlns.com/foaf/0.1/Organization> . <https://discovery.ucl.ac.uk/id/org/ext-d2596156ee130bb0a805ff1203c24d37> <http://xmlns.com/foaf/0.1/name> "The Company of Biologists"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/dc/terms/publisher> <https://discovery.ucl.ac.uk/id/org/ext-d2596156ee130bb0a805ff1203c24d37> . <https://discovery.ucl.ac.uk/id/publication/ext-17548403> <http://www.w3.org/1999/02/22-rdf-syntax-ns#type> <http://purl.org/ontology/bibo/Collection> . <https://discovery.ucl.ac.uk/id/publication/ext-17548403> <http://xmlns.com/foaf/0.1/name> "Disease Models and Mechanisms"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/dc/terms/isPartOf> <https://discovery.ucl.ac.uk/id/publication/ext-17548403> . <https://discovery.ucl.ac.uk/id/publication/ext-17548403> <http://www.w3.org/2002/07/owl#sameAs> <urn:issn:17548403> . <https://discovery.ucl.ac.uk/id/publication/ext-17548403> <http://purl.org/ontology/bibo/issn> "17548403" . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/ontology/bibo/status> <http://purl.org/ontology/bibo/status/forthcoming> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/dc/terms/creator> <https://discovery.ucl.ac.uk/id/person/ext-e1edf44db53ace21b6d048d31db719e9> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/ontology/bibo/authorList> <https://discovery.ucl.ac.uk/id/eprint/10148179#authors> . <https://discovery.ucl.ac.uk/id/eprint/10148179#authors> <http://www.w3.org/1999/02/22-rdf-syntax-ns#_1> <https://discovery.ucl.ac.uk/id/person/ext-e1edf44db53ace21b6d048d31db719e9> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/dc/terms/creator> <https://discovery.ucl.ac.uk/id/person/ext-a3502a0856d93fcda51c9edf335565e1> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/ontology/bibo/authorList> <https://discovery.ucl.ac.uk/id/eprint/10148179#authors> . <https://discovery.ucl.ac.uk/id/eprint/10148179#authors> <http://www.w3.org/1999/02/22-rdf-syntax-ns#_2> <https://discovery.ucl.ac.uk/id/person/ext-a3502a0856d93fcda51c9edf335565e1> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/dc/terms/creator> <https://discovery.ucl.ac.uk/id/person/ext-512e8f72aa7a47280d82561c5c24d6d5> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/ontology/bibo/authorList> <https://discovery.ucl.ac.uk/id/eprint/10148179#authors> . <https://discovery.ucl.ac.uk/id/eprint/10148179#authors> <http://www.w3.org/1999/02/22-rdf-syntax-ns#_3> <https://discovery.ucl.ac.uk/id/person/ext-512e8f72aa7a47280d82561c5c24d6d5> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/dc/terms/creator> <https://discovery.ucl.ac.uk/id/person/ext-e717bfee70d466f43e7c382b16b1afe8> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/ontology/bibo/authorList> <https://discovery.ucl.ac.uk/id/eprint/10148179#authors> . <https://discovery.ucl.ac.uk/id/eprint/10148179#authors> <http://www.w3.org/1999/02/22-rdf-syntax-ns#_4> <https://discovery.ucl.ac.uk/id/person/ext-e717bfee70d466f43e7c382b16b1afe8> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/dc/terms/creator> <https://discovery.ucl.ac.uk/id/person/ext-a38a1ff719652980c0476defe6187e0b> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/ontology/bibo/authorList> <https://discovery.ucl.ac.uk/id/eprint/10148179#authors> . <https://discovery.ucl.ac.uk/id/eprint/10148179#authors> <http://www.w3.org/1999/02/22-rdf-syntax-ns#_5> <https://discovery.ucl.ac.uk/id/person/ext-a38a1ff719652980c0476defe6187e0b> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/dc/terms/creator> <https://discovery.ucl.ac.uk/id/person/ext-18d50c177ebce4532f7b966da18d9ce0> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/ontology/bibo/authorList> <https://discovery.ucl.ac.uk/id/eprint/10148179#authors> . <https://discovery.ucl.ac.uk/id/eprint/10148179#authors> <http://www.w3.org/1999/02/22-rdf-syntax-ns#_6> <https://discovery.ucl.ac.uk/id/person/ext-18d50c177ebce4532f7b966da18d9ce0> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/dc/terms/creator> <https://discovery.ucl.ac.uk/id/person/ext-da3ecec396db0b7fc95eff1cff43f0ed> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/ontology/bibo/authorList> <https://discovery.ucl.ac.uk/id/eprint/10148179#authors> . <https://discovery.ucl.ac.uk/id/eprint/10148179#authors> <http://www.w3.org/1999/02/22-rdf-syntax-ns#_7> <https://discovery.ucl.ac.uk/id/person/ext-da3ecec396db0b7fc95eff1cff43f0ed> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/dc/terms/creator> <https://discovery.ucl.ac.uk/id/person/ext-c2326a931f24b9811790d384c36081a0> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/ontology/bibo/authorList> <https://discovery.ucl.ac.uk/id/eprint/10148179#authors> . <https://discovery.ucl.ac.uk/id/eprint/10148179#authors> <http://www.w3.org/1999/02/22-rdf-syntax-ns#_8> <https://discovery.ucl.ac.uk/id/person/ext-c2326a931f24b9811790d384c36081a0> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/dc/terms/creator> <https://discovery.ucl.ac.uk/id/person/ext-d710290beae6c3cb6f7159feab089528> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/ontology/bibo/authorList> <https://discovery.ucl.ac.uk/id/eprint/10148179#authors> . <https://discovery.ucl.ac.uk/id/eprint/10148179#authors> <http://www.w3.org/1999/02/22-rdf-syntax-ns#_9> <https://discovery.ucl.ac.uk/id/person/ext-d710290beae6c3cb6f7159feab089528> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/dc/terms/creator> <https://discovery.ucl.ac.uk/id/person/ext-cab6786dd67031a4c4e30bf146b1b9ed> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/ontology/bibo/authorList> <https://discovery.ucl.ac.uk/id/eprint/10148179#authors> . <https://discovery.ucl.ac.uk/id/eprint/10148179#authors> <http://www.w3.org/1999/02/22-rdf-syntax-ns#_10> <https://discovery.ucl.ac.uk/id/person/ext-cab6786dd67031a4c4e30bf146b1b9ed> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/dc/terms/creator> <https://discovery.ucl.ac.uk/id/person/ext-41fc32431e029428fbb42d2b8d16901c> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/ontology/bibo/authorList> <https://discovery.ucl.ac.uk/id/eprint/10148179#authors> . <https://discovery.ucl.ac.uk/id/eprint/10148179#authors> <http://www.w3.org/1999/02/22-rdf-syntax-ns#_11> <https://discovery.ucl.ac.uk/id/person/ext-41fc32431e029428fbb42d2b8d16901c> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/dc/terms/creator> <https://discovery.ucl.ac.uk/id/person/ext-5f4894d08972ca656e8d8d7d2c04f5ba> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/ontology/bibo/authorList> <https://discovery.ucl.ac.uk/id/eprint/10148179#authors> . <https://discovery.ucl.ac.uk/id/eprint/10148179#authors> <http://www.w3.org/1999/02/22-rdf-syntax-ns#_12> <https://discovery.ucl.ac.uk/id/person/ext-5f4894d08972ca656e8d8d7d2c04f5ba> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/dc/terms/creator> <https://discovery.ucl.ac.uk/id/person/ext-dc5eb2fe4dc4ecfe82ccf1cc41ee795d> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/ontology/bibo/authorList> <https://discovery.ucl.ac.uk/id/eprint/10148179#authors> . <https://discovery.ucl.ac.uk/id/eprint/10148179#authors> <http://www.w3.org/1999/02/22-rdf-syntax-ns#_13> <https://discovery.ucl.ac.uk/id/person/ext-dc5eb2fe4dc4ecfe82ccf1cc41ee795d> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/dc/terms/creator> <https://discovery.ucl.ac.uk/id/person/ext-e278d249dcd9d0f739978e7ad2c9b5c0> . <https://discovery.ucl.ac.uk/id/eprint/10148179> <http://purl.org/ontology/bibo/authorList> <https://discovery.ucl.ac.uk/id/eprint/10148179#authors> . <https://discovery.ucl.ac.uk/id/eprint/10148179#authors> <http://www.w3.org/1999/02/22-rdf-syntax-ns#_14> <https://discovery.ucl.ac.uk/id/person/ext-e278d249dcd9d0f739978e7ad2c9b5c0> . <https://discovery.ucl.ac.uk/id/person/ext-512e8f72aa7a47280d82561c5c24d6d5> <http://www.w3.org/1999/02/22-rdf-syntax-ns#type> <http://xmlns.com/foaf/0.1/Person> . <https://discovery.ucl.ac.uk/id/person/ext-512e8f72aa7a47280d82561c5c24d6d5> <http://xmlns.com/foaf/0.1/givenName> "Chintan"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-512e8f72aa7a47280d82561c5c24d6d5> <http://xmlns.com/foaf/0.1/familyName> "Trivedi"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-512e8f72aa7a47280d82561c5c24d6d5> <http://xmlns.com/foaf/0.1/name> "Chintan Trivedi"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-5f4894d08972ca656e8d8d7d2c04f5ba> <http://www.w3.org/1999/02/22-rdf-syntax-ns#type> <http://xmlns.com/foaf/0.1/Person> . <https://discovery.ucl.ac.uk/id/person/ext-5f4894d08972ca656e8d8d7d2c04f5ba> <http://xmlns.com/foaf/0.1/givenName> "Jason"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-5f4894d08972ca656e8d8d7d2c04f5ba> <http://xmlns.com/foaf/0.1/familyName> "Rihel"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-5f4894d08972ca656e8d8d7d2c04f5ba> <http://xmlns.com/foaf/0.1/name> "Jason Rihel"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-cab6786dd67031a4c4e30bf146b1b9ed> <http://www.w3.org/1999/02/22-rdf-syntax-ns#type> <http://xmlns.com/foaf/0.1/Person> . <https://discovery.ucl.ac.uk/id/person/ext-cab6786dd67031a4c4e30bf146b1b9ed> <http://xmlns.com/foaf/0.1/givenName> "Stephan CF"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-cab6786dd67031a4c4e30bf146b1b9ed> <http://xmlns.com/foaf/0.1/familyName> "Neuhauss"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-cab6786dd67031a4c4e30bf146b1b9ed> <http://xmlns.com/foaf/0.1/name> "Stephan CF Neuhauss"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-da3ecec396db0b7fc95eff1cff43f0ed> <http://www.w3.org/1999/02/22-rdf-syntax-ns#type> <http://xmlns.com/foaf/0.1/Person> . <https://discovery.ucl.ac.uk/id/person/ext-da3ecec396db0b7fc95eff1cff43f0ed> <http://xmlns.com/foaf/0.1/givenName> "Chris"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-da3ecec396db0b7fc95eff1cff43f0ed> <http://xmlns.com/foaf/0.1/familyName> "Dadswell"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-da3ecec396db0b7fc95eff1cff43f0ed> <http://xmlns.com/foaf/0.1/name> "Chris Dadswell"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-41fc32431e029428fbb42d2b8d16901c> <http://www.w3.org/1999/02/22-rdf-syntax-ns#type> <http://xmlns.com/foaf/0.1/Person> . <https://discovery.ucl.ac.uk/id/person/ext-41fc32431e029428fbb42d2b8d16901c> <http://xmlns.com/foaf/0.1/givenName> "Corinne"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-41fc32431e029428fbb42d2b8d16901c> <http://xmlns.com/foaf/0.1/familyName> "Houart"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-41fc32431e029428fbb42d2b8d16901c> <http://xmlns.com/foaf/0.1/name> "Corinne Houart"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-a3502a0856d93fcda51c9edf335565e1> <http://www.w3.org/1999/02/22-rdf-syntax-ns#type> <http://xmlns.com/foaf/0.1/Person> . <https://discovery.ucl.ac.uk/id/person/ext-a3502a0856d93fcda51c9edf335565e1> <http://xmlns.com/foaf/0.1/givenName> "Richard J"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-a3502a0856d93fcda51c9edf335565e1> <http://xmlns.com/foaf/0.1/familyName> "White"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-a3502a0856d93fcda51c9edf335565e1> <http://xmlns.com/foaf/0.1/name> "Richard J White"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-a38a1ff719652980c0476defe6187e0b> <http://www.w3.org/1999/02/22-rdf-syntax-ns#type> <http://xmlns.com/foaf/0.1/Person> . <https://discovery.ucl.ac.uk/id/person/ext-a38a1ff719652980c0476defe6187e0b> <http://xmlns.com/foaf/0.1/givenName> "Stephanie"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-a38a1ff719652980c0476defe6187e0b> <http://xmlns.com/foaf/0.1/familyName> "Niklaus"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-a38a1ff719652980c0476defe6187e0b> <http://xmlns.com/foaf/0.1/name> "Stephanie Niklaus"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-e1edf44db53ace21b6d048d31db719e9> <http://www.w3.org/1999/02/22-rdf-syntax-ns#type> <http://xmlns.com/foaf/0.1/Person> . <https://discovery.ucl.ac.uk/id/person/ext-e1edf44db53ace21b6d048d31db719e9> <http://xmlns.com/foaf/0.1/givenName> "Karin"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-e1edf44db53ace21b6d048d31db719e9> <http://xmlns.com/foaf/0.1/familyName> "Tuschl"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-e1edf44db53ace21b6d048d31db719e9> <http://xmlns.com/foaf/0.1/name> "Karin Tuschl"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-dc5eb2fe4dc4ecfe82ccf1cc41ee795d> <http://www.w3.org/1999/02/22-rdf-syntax-ns#type> <http://xmlns.com/foaf/0.1/Person> . <https://discovery.ucl.ac.uk/id/person/ext-dc5eb2fe4dc4ecfe82ccf1cc41ee795d> <http://xmlns.com/foaf/0.1/givenName> "Stephen W"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-dc5eb2fe4dc4ecfe82ccf1cc41ee795d> <http://xmlns.com/foaf/0.1/familyName> "Wilson"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-dc5eb2fe4dc4ecfe82ccf1cc41ee795d> <http://xmlns.com/foaf/0.1/name> "Stephen W Wilson"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-e717bfee70d466f43e7c382b16b1afe8> <http://www.w3.org/1999/02/22-rdf-syntax-ns#type> <http://xmlns.com/foaf/0.1/Person> . <https://discovery.ucl.ac.uk/id/person/ext-e717bfee70d466f43e7c382b16b1afe8> <http://xmlns.com/foaf/0.1/givenName> "Leonardo E"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-e717bfee70d466f43e7c382b16b1afe8> <http://xmlns.com/foaf/0.1/familyName> "Valdivia"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-e717bfee70d466f43e7c382b16b1afe8> <http://xmlns.com/foaf/0.1/name> "Leonardo E Valdivia"^^<http://www.w3.org/2001/XMLSchema#string> . <https://discovery.ucl.ac.uk/id/person/ext-e278d249dcd9d0f739978e7ad2c9b5c0> <http://www.w3.org/1999/02/22-rdf-syntax-ns#type> 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