title: Zebrafish - An emerging model to explore thyroid hormone transporters and psychomotor retardation.
abstract: Thyroid hormones (THs) regulate a variety of fundamental physiological processes, including the development and maintenance of the brain. For decades, it was thought that THs enter the cells by passive diffusion. However, it is now clear that TH transport across the cell membrane requires specific transporter proteins that facilitate the uptake and efflux of THs. Several thyroid hormone transmembrane transporters (THTTs) have been identified, including monocarboxylate transporter 8 (MCT8), MCT10, and organic anion transporting polypeptide 1C1 (OATP1C1). The critical role of THTTs in regulating metabolism and brain function is demonstrated in the Allan-Herndon-Dudley syndrome (AHDS), an X-linked psychomotor retardation associated with mutations in the MCT8/SLC16A2 gene. In addition to traditional research on humans, cell-lines, and rodents, the zebrafish has recently emerged as an attractive model to study THTTs and neuroendocrinological-related disorders. In this review, we describe the unique contribution of zebrafish studies to the understanding of the functional role of THTTs in live animals, and how this transparent vertebrate model can be used for translational studies on TH-related disorders.
authors.affiliation: The Faculty of Life Sciences and the Multidisciplinary Brain Research Center, Bar-Ilan University, Ramat-Gan 5290002, Israel.
authors.forename: David
authors.initials: D
authors.lastname: Zada
authors.affiliation: The Faculty of Life Sciences and the Multidisciplinary Brain Research Center, Bar-Ilan University, Ramat-Gan 5290002, Israel.
authors.forename: Einat
authors.initials: E
authors.lastname: Blitz
authors.affiliation: The Faculty of Life Sciences and the Multidisciplinary Brain Research Center, Bar-Ilan University, Ramat-Gan 5290002, Israel. Electronic address: lior.appelbaum@biu.ac.il.
authors.forename: Lior
authors.initials: L
authors.lastname: Appelbaum
mesh_terms: D000818:Animals; D001921:Brain / Q000378:metabolism / Q000473:pathology; D002352:Carrier Proteins / Q000235:genetics* / Q000378:metabolism; D004195:Disease Models, Animal; D018507:Gene Expression Regulation, Developmental; D055786:Gene Knockout Techniques; D006801:Humans; D007814:Larva / Q000235:genetics / Q000254:growth & development / Q000378:metabolism; D038901:X-Linked Intellectual Disability / Q000235:genetics* / Q000378:metabolism / Q000473:pathology; D027501:Monocarboxylic Acid Transporters / Q000172:deficiency / Q000235:genetics*; D009123:Muscle Hypotonia / Q000235:genetics* / Q000378:metabolism / Q000473:pathology; D009133:Muscular Atrophy / Q000235:genetics* / Q000378:metabolism / Q000473:pathology; D027361:Organic Anion Transporters / Q000235:genetics* / Q000378:metabolism; D011485:Protein Binding; D020033:Protein Isoforms / Q000172:deficiency / Q000235:genetics; D021381:Protein Transport; D015398:Signal Transduction; D013963:Thyroid Hormones / Q000235:genetics* / Q000378:metabolism; D015027:Zebrafish / Q000235:genetics* / Q000254:growth & development / Q000378:metabolism
chemical_list: D002352:Carrier Proteins; D027501:Monocarboxylic Acid Transporters; D027361:Organic Anion Transporters; D020033:Protein Isoforms; C570017:Slc16a2 protein, zebrafish; D013963:Thyroid Hormones
keywords: MCT8; Mental retardation; OATP1C1; Thyroid hormone analogues; Thyroid transporters; Zebrafish