FRINK Linked Data Fragments server

Matches in SemOpenAlex for { <https://semopenalex.org/work/W2149476005> ?p ?o ?g. }

• W2149476005 endingPage "2330" @default.
• W2149476005 startingPage "2320" @default.
• W2149476005 abstract "Allan-Herndon-Dudley syndrome (AHDS) is an inherited disorder of brain development characterized by severe psychomotor retardation. This X-linked disease is caused by mutations in the monocarboxylate transporter 8 (MCT8), an important thyroid hormone transporter in brain neurons. MCT8-knockout mice lack the 2 major neurological symptoms of AHDS, namely locomotor problems and cognitive impairment. The pathological mechanism explaining the symptoms is still obscure, and no cure for this condition is known. The development of an animal model that carries MCT8-related neurological symptoms is warranted. We have employed morpholino-based gene knockdown to create zebrafish deficient for mct8. Knockdown of mct8 results in specific symptoms in the thyroid axis and brain. The mct8-morphants showed impaired locomotor behavior and brain development. More specifically, we observed maldevelopment of the cerebellum and mid-hindbrain boundary and apoptotic clusters in the zebrafish brain. The mRNA expression of zebrafish orthologs of mammalian TSH, thyroid hormone transporters, and deiodinases was altered in mct8 morphants. In particular, deiodinase type 3 gene expression was consistently up-regulated in zebrafish mct8 morphants. The thyroid hormone metabolite tetrac, but not T3, partly ameliorated the affected phenotype and locomotion disability of morphant larvae. Our results show that mct8 knockdown in zebrafish larvae results in disturbances in the thyroid axis, brain, and locomotion behavior, which is congruent with the clinical aspect of impaired locomotion and cognition in patients with AHDS. Taken together, the zebrafish is a suitable animal model for the study of the pathophysiology of AHDS." @default.
• W2149476005 created "2016-06-24" @default.
• W2149476005 creator A5000563716 @default.
• W2149476005 creator A5000648294 @default.
• W2149476005 creator A5018867215 @default.
• W2149476005 creator A5031373547 @default.
• W2149476005 creator A5056342793 @default.
• W2149476005 creator A5080801101 @default.
• W2149476005 date "2014-06-01" @default.
• W2149476005 modified "2023-10-15" @default.
• W2149476005 title "Knockdown of Monocarboxylate Transporter 8 (mct8) Disturbs Brain Development and Locomotion in Zebrafish" @default.
• W2149476005 cites W1963976116 @default.
• W2149476005 cites W1965744141 @default.
• W2149476005 cites W1968953868 @default.
• W2149476005 cites W1976810009 @default.
• W2149476005 cites W1984924011 @default.
• W2149476005 cites W1985685797 @default.
• W2149476005 cites W1991454487 @default.
• W2149476005 cites W1992268803 @default.
• W2149476005 cites W2006137582 @default.
• W2149476005 cites W2008868853 @default.
• W2149476005 cites W2009635392 @default.
• W2149476005 cites W2017632004 @default.
• W2149476005 cites W2019419796 @default.
• W2149476005 cites W2030509668 @default.
• W2149476005 cites W2030833458 @default.
• W2149476005 cites W2033805763 @default.
• W2149476005 cites W2035087361 @default.
• W2149476005 cites W2046152275 @default.
• W2149476005 cites W2051673849 @default.
• W2149476005 cites W2062347722 @default.
• W2149476005 cites W2071232430 @default.
• W2149476005 cites W2078333270 @default.
• W2149476005 cites W2080817589 @default.
• W2149476005 cites W2086609663 @default.
• W2149476005 cites W2088566177 @default.
• W2149476005 cites W2089164938 @default.
• W2149476005 cites W2099824652 @default.
• W2149476005 cites W2100049277 @default.
• W2149476005 cites W2101816987 @default.
• W2149476005 cites W2103080150 @default.
• W2149476005 cites W2111575083 @default.
• W2149476005 cites W2114513291 @default.
• W2149476005 cites W2115582509 @default.
• W2149476005 cites W2121125354 @default.
• W2149476005 cites W2126991953 @default.
• W2149476005 cites W2127859037 @default.
• W2149476005 cites W2133877174 @default.
• W2149476005 cites W2147843272 @default.
• W2149476005 cites W2149097868 @default.
• W2149476005 cites W2149995096 @default.
• W2149476005 cites W2152278958 @default.
• W2149476005 cites W2156416721 @default.
• W2149476005 cites W2160166542 @default.
• W2149476005 cites W2164991389 @default.
• W2149476005 cites W2186384656 @default.
• W2149476005 doi "https://doi.org/10.1210/en.2013-1962" @default.
• W2149476005 hasPubMedId "https://pubmed.ncbi.nlm.nih.gov/24693966" @default.
• W2149476005 hasPublicationYear "2014" @default.
• W2149476005 type Work @default.
• W2149476005 sameAs 2149476005 @default.
• W2149476005 citedByCount "36" @default.
• W2149476005 countsByYear W21494760052015 @default.
• W2149476005 countsByYear W21494760052016 @default.
• W2149476005 countsByYear W21494760052017 @default.
• W2149476005 countsByYear W21494760052018 @default.
• W2149476005 countsByYear W21494760052019 @default.
• W2149476005 countsByYear W21494760052020 @default.
• W2149476005 countsByYear W21494760052021 @default.
• W2149476005 countsByYear W21494760052022 @default.
• W2149476005 countsByYear W21494760052023 @default.
• W2149476005 crossrefType "journal-article" @default.
• W2149476005 hasAuthorship W2149476005A5000563716 @default.
• W2149476005 hasAuthorship W2149476005A5000648294 @default.
• W2149476005 hasAuthorship W2149476005A5018867215 @default.
• W2149476005 hasAuthorship W2149476005A5031373547 @default.
• W2149476005 hasAuthorship W2149476005A5056342793 @default.
• W2149476005 hasAuthorship W2149476005A5080801101 @default.
• W2149476005 hasBestOaLocation W21494760051 @default.
• W2149476005 hasConcept C104317684 @default.
• W2149476005 hasConcept C126322002 @default.
• W2149476005 hasConcept C134018914 @default.
• W2149476005 hasConcept C149011108 @default.
• W2149476005 hasConcept C169760540 @default.
• W2149476005 hasConcept C173396325 @default.
• W2149476005 hasConcept C2776878037 @default.
• W2149476005 hasConcept C2781217356 @default.
• W2149476005 hasConcept C54355233 @default.
• W2149476005 hasConcept C71924100 @default.
• W2149476005 hasConcept C86803240 @default.
• W2149476005 hasConceptScore W2149476005C104317684 @default.
• W2149476005 hasConceptScore W2149476005C126322002 @default.
• W2149476005 hasConceptScore W2149476005C134018914 @default.
• W2149476005 hasConceptScore W2149476005C149011108 @default.
• W2149476005 hasConceptScore W2149476005C169760540 @default.
• W2149476005 hasConceptScore W2149476005C173396325 @default.
• W2149476005 hasConceptScore W2149476005C2776878037 @default.
• W2149476005 hasConceptScore W2149476005C2781217356 @default.

• next