Altered behavioral performance and live imaging of circuit-specific neural deficiencies in a zebrafish model for psychomotor retardation
Open accessAbstract
The mechanisms and treatment of psychomotor retardation, which includes motor and cognitive impairment, are indefinite. The Allan-Herndon-Dudley syndrome (AHDS) is an X-linked psychomotor retardation characterized by delayed development, severe intellectual disability, muscle hypotonia, and spastic paraplegia, in combination with disturbed thyroid hormone (TH) parameters. AHDS has been associated with mutations in the monocarboxylate transporter 8 (mct8/slc16a2) gene, which is a TH transporter. In order to determine the pathophysiological mechanisms of AHDS, MCT8 knockout mice were intensively studied. Although these mice faithfully replicated the abnormal serum TH levels, they failed to exhibit the neurological and behavioral symptoms of AHDS patients. Here, we generated an mct8 mutant (mct8-/-) zebrafish using zinc-finger nuclease (ZFN)-mediated targeted gene editing system. The elimination of MCT8 decreased the expression levels of TH receptors; however, it did not affect the expression of other TH-related genes. Similar to human patients, mct8-/- larvae exhibited neurological and behavioral deficiencies. High-throughput behavioral assays demonstrated that mct8-/- larvae exhibited reduced locomotor activity, altered response to external light and dark transitions and an increase in sleep time. These deficiencies in behavioral performance were associated with altered expression of myelin-related genes and neuron-specific deficiencies in circuit formation. Time-lapse imaging of single-axon arbors and synapses in live mct8-/- larvae revealed a reduction in filopodia dynamics and axon branching in sensory neurons and decreased synaptic density in motor neurons. These phenotypes enable assessment of the therapeutic potential of three TH analogs that can enter the cells in the absence of MCT8. The TH analogs restored the myelin and axon outgrowth deficiencies in mct8-/- larvae. These findings suggest a mechanism by which MCT8 regulates neural circuit assembly, ultimately mediating sensory and motor control of behavioral performance. We also propose that the administration of TH analogs early during embryo development can specifically reduce neurological damage in AHDS patients.
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These results align with reduced synaptophysin expression in post-mortem human brain and neuronal deficiencies in Mct8/Oatp1c1 dKO mouse and zebrafish Mct8 mutant.
“These results align with several findings in MCT8 deficiency, including reduced synaptophysin expression in the post-mortem human brain (López-Espíndola et al., 2014) and neuronal deficiencies observed in the Mct8/Oatp1c1 double KO (dKO) mouse (Mayerl et al., 2014) and zebrafish Mct8 mutant model (Zada et al., 2014).”
From Modeling Psychomotor Retardation using iPSCs from MCT8-Deficient Patients Indicates a Prominent Role for the Blood-Brain Barrier (2017) · cites this paper as Zada et al., 2014
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ZFN, TALEN, and CRISPR/Cas-based methods for genome engineering
Gaj, T., Gersbach, C. A., & Barbas, C. F. (2013) · Trends in Biotechnology · doi:10.1016/j.tibtech.2013.04.004 · Cited by 3,820 · see citing works
Physiological and Molecular Basis of Thyroid Hormone Action
Yen, P. M. (2001) · Physiological Reviews · doi:10.1152/physrev.2001.81.3.1097 · Cited by 2,011 · see citing works
A Transposon-Mediated Gene Trap Approach Identifies Developmentally Regulated Genes in Zebrafish
Kawakami, K., Takeda, H., Kawakami, N., Kobayashi, M., Matsuda, N., & Mishina, M. (2004) · Developmental Cell · doi:10.1016/j.devcel.2004.06.005 · Cited by 852 · see citing works
Dendritic Anomalies in Disorders Associated with Mental Retardation
Kaufmann, W. E. (2000) · Cerebral Cortex · doi:10.1093/cercor/10.10.981 · Cited by 733 · see citing works
The Role of Thyroid Hormones in Prenatal and Neonatal Neurological Development—Current Perspectives
Porterfield, S. P., & Hendrich, C. E. (1993) · Endocrine Reviews · doi:10.1210/edrv-14-1-94 · Cited by 724 · see citing works
Friesema, E. C., Grueters, A., Biebermann, H., Krude, H., von Moers, A., Reeser, M., Barrett, T. G., Mancilla, E. E., Svensson, J., Kester, M. H., et al. (2004) · The Lancet · doi:10.1016/s0140-6736(04)17226-7 · Cited by 709 · see citing works
Mechanisms of axon ensheathment and myelin growth
Sherman, D. L., & Brophy, P. J. (2005) · Nature Reviews Neuroscience · doi:10.1038/nrn1743 · Cited by 668 · see citing works
Developmental regulation of zebrafish MyoD in wild-type, no tail and spadetail embryos
Weinberg, E. S., Allende, M. L., Kelly, C., Abdelhamid, A., Murakami, T., Andermann, P., Doerre, O. G., Grunwald, D. J., & Riggleman, B. (1996) · Development · doi:10.1242/dev.122.1.271 · Cited by 650 · see citing works
Barrès, A., Lazar, M. A., & RAFF, M. C. (1994) · Development · doi:10.1242/dev.120.5.1097 · Cited by 593 · see citing works
Hypocretin/orexin overexpression induces an insomnia-like phenotype in zebrafish
Prober, D. A., Rihel, J., Onah, A. A., Sung, R.-J., & Schier, A. F. (2006) · The Journal of Neuroscience · doi:10.1523/jneurosci.4332-06.2006 · Cited by 508 · see citing works
Rapid Conduction and the Evolution of Giant Axons and Myelinated Fibers
Hartline, D. K., & Colman, D. R. (2007) · Current Biology · doi:10.1016/j.cub.2006.11.042 · Cited by 478 · cited 1× · see citing works
“Loss of myelin sheaths produces a wide variety of neurological symptoms, including the slow progression of action potential and deficient axon elaboration [79].”
BackgroundMelatonin promotes sleep-like state in zebrafish
Zhdanova, I. V., Wang, S. Y., Leclair, O. U., & Danilova, N. P. (2001) · Brain Research · doi:10.1016/s0006-8993(01)02444-1 · Cited by 418 · see citing works
Characterization of Sleep in Zebrafish and Insomnia in Hypocretin Receptor Mutants
Yokogawa, T., Marin, W., Faraco, J. H., Pézeron, G., Appelbaum, L., Zhang, J., Rosa, F., Mourrain, P., & Mignot, E. J.-M. (2007) · PLoS Biology · doi:10.1371/journal.pbio.0050277 · Cited by 402 · see citing works
Self-citationFailure of neuronal homeostasis results in common neuropsychiatric phenotypes
Ramocki, M. B., & Zoghbi, H. Y. (2008) · Nature · doi:10.1038/nature07457 · Cited by 399 · see citing works
Abnormal thyroid hormone metabolism in mice lacking the monocarboxylate transporter 8
Trajkovic, M., Visser, T. J., Mittag, J., Horn, S., Lukas, J., Darras, V. M., Raivich, G., Bauer, K., & Heuer, H. (2007) · Journal of Clinical Investigation · doi:10.1172/jci28253 · Cited by 353 · see citing works
Synapse Pathology in Psychiatric and Neurologic Disease
van Spronsen, M., & Hoogenraad, C. C. (2010) · Current Neurology and Neuroscience Reports · doi:10.1007/s11910-010-0104-8 · Cited by 345 · see citing works
Characterization of myelination in the developing zebrafish
Brösamle, C., & Halpern, M. E. (2002) · Glia · doi:10.1002/glia.10088 · Cited by 327 · see citing works
Pizzagalli, F., Hagenbuch, B., Stieger, B., Klenk, U., Folkers, G., & Meier, P. J. (2002) · Molecular Endocrinology · doi:10.1210/me.2001-0309 · Cited by 321 · see citing works
Roberts, L. M., Woodford, K., Zhou, M., Black, D. S., Haggerty, J. E., Tate, E. H., Grindstaff, K. K., Mengesha, W., Raman, C., & Zerangue, N. (2008) · Endocrinology · doi:10.1210/en.2008-0378 · Cited by 320 · see citing works
Dumitrescu, A. M., Liao, X.-H., Weiss, R. E., Millen, K., & Refetoff, S. (2006) · Endocrinology · doi:10.1210/en.2006-0390 · Cited by 318 · see citing works
olig2 is required for zebrafish primary motor neuron and oligodendrocyte development.
Park, H.-C., Mehta, A., Richardson, J., & Appel, B. (2002) · Developmental Biology · doi:10.1006/dbio.2002.0738 · Cited by 316 · see citing works
Zebrafish mutations affecting retinotectal axon pathfinding
Karlstrom, R. O., Trowe, T., Klostermann, S., Baier, H., Brand, M., Crawford, A. D., Grunewald, B., Haffter, P., Hoffmann, H., Meyer, S. U., et al. (1996) · Development · doi:10.1242/dev.123.1.427 · Cited by 302 · see citing works
Neural cell fate analysis in zebrafish using olig2 BAC transgenics.
Shin, J., Park, H.-C., Topczewska, J. M., Mawdsley, D., & Appel, B. (2003) · Methods in Cell Science · doi:10.1023/b:mics.0000006847.09037.3a · Cited by 295 · see citing works
Perspectives in the Study of Thyroid Hormone Action on Brain Development and Function
Bernal, J., Guadaño‐Ferraz, A., & Morte, B. (2003) · Thyroid · doi:10.1089/105072503770867174 · Cited by 287 · see citing works
Effective cellular uptake and efflux of thyroid hormone by human monocarboxylate transporter 10
Friesema, E. C. H., Jansen, J., Jachtenberg, J.-W., Visser, W. E., Kester, M. H. A., & Visser, T. J. (2008) · Molecular Endocrinology · doi:10.1210/me.2007-0112 · Cited by 285 · see citing works
Meyer, M. P., & Smith, S. J. (2006) · Journal of Neuroscience · doi:10.1523/jneurosci.0223-06.2006 · Cited by 283 · see citing works
Transporters MCT8 and OATP1C1 maintain murine brain thyroid hormone homeostasis
Mayerl, S., Müller, J., Bauer, R., Richert, S., Kassmann, C. M., Darras, V. M., Buder, K., Boelen, A., Visser, T. J., & Heuer, H. (2014) · Journal of Clinical Investigation · doi:10.1172/jci70324 · Cited by 281 · see citing works
A zebrafish model of tauopathy allows in vivo imaging of neuronal cell death and drug evaluation
Paquet, D., Bhat, R. V., Sydow, A., Mandelkow, E., von Berg, S., Hellberg, S., Fälting, J. M., Distel, M., Köster, R. W., Schmid, B., et al. (2009) · Journal of Clinical Investigation · doi:10.1172/jci37537 · Cited by 278 · see citing works
Heuer, H., Maier, M. K., Iden, S., Mittag, J., Friesema, E. C. H., Visser, T. J., & Bauer, K. (2005) · Endocrinology · doi:10.1210/en.2004-1179 · Cited by 269 · see citing works
Ito, K., Uchida, Y., Ohtsuki, S., Aizawa, S., Kawakami, H., Katsukura, Y., Kamiie, J., & Terasaki, T. (2011) · Journal of Pharmaceutical Sciences · doi:10.1002/jps.22487 · Cited by 231 · see citing works
From cells to circuits: development of the zebrafish spinal cord.
Lewis, K. E., & Eisen, J. (2003) · Progress in neurobiology · doi:10.1016/s0301-0082(03)00052-2 · Cited by 230 · see citing works
Protein Zero Gene Expression Is Regulated by the Glial Transcription Factor Sox10
Peirano, R. I., Goerich, D. E., Riethmacher, D., & Wegner, M. (2000) · Molecular and Cellular Biology · doi:10.1128/mcb.20.9.3198-3209.2000 · Cited by 223 · see citing works
A Behavioral Assay to Measure Responsiveness of Zebrafish to Changes in Light Intensities
Emran, F., Rihel, J., & Dowling, J. E. (2008) · Journal of Visualized Experiments · doi:10.3791/923 · Cited by 221 · see citing works
Russell, W., Harrison, R. F., Smith, N., Darzy, K., Shalet, S., Weetman, A. P., & Ross, R. J. (2008) · The Journal of Clinical Endocrinology & Metabolism · doi:10.1210/jc.2007-2674 · Cited by 219 · see citing works
Monitoring neural activity with bioluminescence during natural behavior
Naumann, E. A., Kampff, A. R., Prober, D. A., Schier, A. F., & Engert, F. (2010) · Nature Neuroscience · doi:10.1038/nn.2518 · Cited by 196 · see citing works
Sleep–wake regulation and hypocretin–melatonin interaction in zebrafish
Appelbaum, L., Wang, G. X., Maro, G. S., Mori, R., Tovin, A., Marin, W., Yokogawa, T., Kawakami, K., Smith, S. J., Gothilf, Y., et al. (2009) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.906637106 · Cited by 192 · see citing works
Self-citationPhenylthiourea disrupts thyroid function in developing zebrafish
Elsalini, O. A., & Rohr, K. B. (2003) · Development Genes and Evolution · doi:10.1007/s00427-002-0279-3 · Cited by 181 · see citing works
Circadian and Homeostatic Regulation of Structural Synaptic Plasticity in Hypocretin Neurons
Appelbaum, L., Wang, G. X., Yokogawa, T., Skariah, G. M., Smith, S. J., Mourrain, P., & Mignot, E. J.-M. (2010) · Neuron · doi:10.1016/j.neuron.2010.09.006 · Cited by 172 · see citing works
Self-citationHarsan, L. A., Steibel, J., Zaremba, A., Agin, A., Sapin, R., Poulet, P., Guignard, B., Parizel, N., Grucker, D., Boehm, N., et al. (2008) · Journal of Neuroscience · doi:10.1523/jneurosci.4453-08.2008 · Cited by 171 · see citing works
Lessons from morpholino-based screening in zebrafish
Bedell, V. M., Westcot, S. E., & Ekker, S. C. (2011) · Briefings in Functional Genomics · doi:10.1093/bfgp/elr021 · Cited by 168 · see citing works
The T3-induced gene KLF9 regulates oligodendrocyte differentiation and myelin regeneration
Dugas, J. C., Ibrahim, A., & Barres, B. A. (2012) · Molecular and Cellular Neuroscience · doi:10.1016/j.mcn.2012.03.007 · Cited by 167 · see citing works
Rodríguez‐Peña, A., Ibarrola, N., Iñiguez, M. A., Múñoz, A., & Bernal, J. (1993) · Journal of Clinical Investigation · doi:10.1172/jci116301 · Cited by 166 · see citing works
Sagasti, A., Guido, M. R., Raible, D. W., & Schier, A. F. (2005) · Current Biology · doi:10.1016/j.cub.2005.03.048 · Cited by 162 · see citing works
Calzà, L., Fernández, M., Giuliani, A., Aloe, L., & Giardino, L. (2002) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.052704499 · Cited by 156 · see citing works
Impact of Oatp1c1 deficiency on thyroid hormone metabolism and action in the mouse brain
Mayerl, S., Visser, T. J., Darras, V. M., Horn, S., & Heuer, H. (2012) · Endocrinology · doi:10.1210/en.2011-1633 · Cited by 138 · see citing works
Diiodothyropropionic acid (DITPA) in the treatment of MCT8 deficiency
Verge, C. F., Konrad, D., Cohen, M., Di Cosmo, C., Dumitrescu, A. M., Marcinkowski, T., Hameed, S., Hamilton, J., Weiss, R. E., & Refetoff, S. (2012) · The Journal of Clinical Endocrinology & Metabolism · doi:10.1210/jc.2012-2556 · Cited by 134 · cited 1× · see citing works
“Indeed, DITPA has been tested in adult MCT8-KO mouse and administered to several AHDS patients [49,50].”
MethodEmerging links between homeostatic synaptic plasticity and neurological disease
Wondolowski, J., & Dickman, D. K. (2013) · Frontiers in Cellular Neuroscience · doi:10.3389/fncel.2013.00223 · Cited by 132 · see citing works
Visualization of myelination in GFP‐transgenic zebrafish
Jung, S., Kim, S., Chung, A., Kim, H., So, J., Ryu, J.-H., Park, H., & Kim, C. (2009) · Developmental Dynamics · doi:10.1002/dvdy.22166 · Cited by 128 · see citing works
Sleep and its regulation in zebrafish
Zhdanova, I. V. (2011) · Reviews in the Neurosciences · doi:10.1515/rns.2011.005 · Cited by 125 · see citing works
Knipper, M., Bandtlow, C. E., Gestwa, L., Köpschall, I., Rohbock, K., Wiechers, B., Zenner, H.-P., & Zimmermann, U. (1998) · Development · doi:10.1242/dev.125.18.3709 · Cited by 123 · see citing works
Molecular cloning and characterization of thyroid hormone receptors in teleost fish
Marchand, O., Safi, R., Escrivá, H., Rompaey, E. V., Prunet, P., & Laudet, V. (2001) · Journal of Molecular Endocrinology · doi:10.1677/jme.0.0260051 · Cited by 119 · see citing works
Kinne, A., Kleinau, G., Hoefig, C. S., Grüters, A., Köhrle, J., Krause, G., & Schweizer, U. (2010) · Journal of Biological Chemistry · doi:10.1074/jbc.m110.129577 · Cited by 116 · see citing works
Genetic Ablation of Hypocretin Neurons Alters Behavioral State Transitions in Zebrafish
Elbaz, I., Yelin‐Bekerman, L., Nicenboim, J., Vatine, G. D., & Appelbaum, L. (2012) · Journal of Neuroscience · doi:10.1523/jneurosci.1284-12.2012 · Cited by 111 · see citing works
Self-citationDirect activation of the Mauthner cell by electric field pulses drives ultrarapid escape responses
Tabor, K. M., Bergeron, S. A., Horstick, E. J., Jordan, D. C., Lammi, V., Porkka‐Heiskanen, T., Haspel, G., & Burgess, H. A. (2014) · Journal of Neurophysiology · doi:10.1152/jn.00228.2014 · Cited by 111 · see citing works
Shine, H. D., Readhead, C., Popko, B., Hood, L., & Sidman, R. L. (1992) · Journal of Neurochemistry · doi:10.1111/j.1471-4159.1992.tb09316.x · Cited by 111 · see citing works
Cosmo, C. D., Liao, X.-H., Dumitrescu, A. M., Weiss, R. E., & Refetoff, S. (2009) · Endocrinology · doi:10.1210/en.2009-0209 · Cited by 109 · see citing works
X–linked paroxysmal dyskinesia and severe global retardation caused by defective MCT8 gene
Brockmann, K., Dumitrescu, A. M., Best, T. T., Hanefeld, F. A., & Refetoff, S. (2005) · Journal of Neurology · doi:10.1007/s00415-005-0713-3 · Cited by 108 · see citing works
In VivoImaging of Disease-Related Mitochondrial Dynamics in a Vertebrate Model System
Plucińska, G., Paquet, D., Hruscha, A., Godinho, L., Haass, C., Schmid, B., & Misgeld, T. (2012) · Journal of Neuroscience · doi:10.1523/jneurosci.1327-12.2012 · Cited by 103 · see citing works
Programmed Cell Death in Zebrafish Rohon Beard Neurons Is Influenced by TrkC1/NT-3 Signaling
Williams, J. A. E., Barrios, A., Gatchalian, C. L., Rubin, L. L., Wilson, S. W., & Holder, N. (2000) · Developmental Biology · doi:10.1006/dbio.2000.9860 · Cited by 99 · see citing works
Multigenic control of thyroid hormone functions in the nervous system
Nunez, J., Celi, F. S., Ng, L., & Forrest, D. (2008) · Molecular and Cellular Endocrinology · doi:10.1016/j.mce.2008.03.006 · Cited by 96 · see citing works
Oligodendrocyte development and myelination in GFP‐transgenic zebrafish
Yoshida, M., & Macklin, W. (2005) · Journal of Neuroscience Research · doi:10.1002/jnr.20516 · Cited by 93 · see citing works
X-Linked MCT8 Gene Mutations: Characterization of the Pediatric Neurologic Phenotype
Holden, K. R., Zuñiga, O. F., May, M. M., Su, H., Molinero, M. R., Rogers, R. C., & Schwartz, C. E. (2005) · Journal of Child Neurology · doi:10.1177/08830738050200101601 · Cited by 92 · see citing works
Namba, N., Etani, Y., Kitaoka, T., Nakamoto, Y., Nakacho, M., Bessho, K., Miyoshi, Y., Mushiake, S., Mohri, I., Arai, H., et al. (2007) · European Journal of Pediatrics · doi:10.1007/s00431-007-0589-6 · Cited by 89 · see citing works
Circadian clocks, rhythmic synaptic plasticity and the sleep-wake cycle in zebrafish
Elbaz, I., Foulkes, N. S., Gothilf, Y., & Appelbaum, L. (2013) · Frontiers in Neural Circuits · doi:10.3389/fncir.2013.00009 · Cited by 89 · see citing works
Self-citationTrajkovic-Arsic, M., Müller, J., Darras, V. M., Groba, C., Lee, S., Weih, D., Bauer, K., Visser, T. J., & Heuer, H. (2010) · Endocrinology · doi:10.1210/en.2010-0593 · Cited by 87 · see citing works
Horn, S., Kersseboom, S., Mayerl, S. E., Müller, J., Groba, C., Trajkovic‐Arsic, M., Ackermann, T., Visser, T. J., & Heuer, H. (2013) · Endocrinology · doi:10.1210/en.2012-1628 · Cited by 84 · see citing works
Schweitzer, J., Becker, T., Becker, C. G., & Schachner, M. (2003) · Glia · doi:10.1002/glia.10192 · Cited by 82 · see citing works
Arjona, F. J., de Vrieze, E., Visser, T. J., Flik, G., & Klaren, P. H. M. (2011) · Endocrinology · doi:10.1210/en.2011-1166 · Cited by 78 · see citing works
Temporal dynamics of myelination in the zebrafish spinal cord
Buckley, C. E., Marguerie, A., ALDERTON, W. K., & Franklin, R. J. M. (2010) · Glia · doi:10.1002/glia.20964 · Cited by 74 · see citing works
Zebrafish as a Model for Monocarboxyl Transporter 8-Deficiency
Vatine, G. D., Zada, D., Lerer‐Goldshtein, T., Tovin, A., Malkinson, G., Yaniv, K., & Appelbaum, L. (2012) · Journal of Biological Chemistry · doi:10.1074/jbc.m112.413831 · Cited by 72 · see citing works
Self-citationChanges in Thyroid Status During Perinatal Development of MCT8-Deficient Male Mice
Ferrara, A. M., Liao, X.-H., Gil-Ibáñez, P., Marcinkowski, T., Bernal, J., Weiss, R. E., Dumitrescu, A. M., & Refetoff, S. (2013) · Endocrinology · doi:10.1210/en.2012-2031 · Cited by 70 · see citing works
Zebrafish as a model to study peripheral thyroid hormone metabolism in vertebrate development
Heijlen, M., Houbrechts, A. M., & Darras, V. (2013) · General and Comparative Endocrinology · doi:10.1016/j.ygcen.2013.04.004 · Cited by 69 · see citing works
Campos‐Barros, Á., Musa, A., Flechner, A., Hessenius, C., Gaio, U. C., MEINHOLD, H., & Baumgartner, A. (1997) · Journal of Neurochemistry · doi:10.1046/j.1471-4159.1997.68020795.x · Cited by 65 · see citing works
Darras, V., Herck, S. L. J. V., Heijlen, M., & Groef, B. D. (2011) · Journal of Thyroid Research · doi:10.4061/2011/402320 · Cited by 65 · see citing works
Vincent, J., Legrand, C., Rabié, A., & Legrand, J. (1982) · PubMed · Cited by 65 · see citing works
Function of thyroid hormone transporters in the central nervous system
Schweizer, U., & Köhrle, J. (2012) · Biochimica et Biophysica Acta (BBA) - General Subjects · doi:10.1016/j.bbagen.2012.07.015 · Cited by 64 · see citing works
Cayrou, C., Denver, R. J., & Puymirat, J. (2002) · Endocrinology · doi:10.1210/endo.143.6.8856 · Cited by 64 · see citing works
Heuer, H., & Visser, T. J. (2013) · Biochimica et Biophysica Acta (BBA) - General Subjects · doi:10.1016/j.bbagen.2012.04.009 · Cited by 64 · see citing works
White matter abnormalities and dystonic motor disorder associated with mutations in the SLC16A2 gene
Gika, A. D., Siddiqui, A. U., Hulse, A., EDWARD, S., Fallon, P., McEntagart, M., Jan, W., Josifova, D. J., Lerman‐Sagie, T., Drummond, J., et al. (2009) · Developmental Medicine & Child Neurology · doi:10.1111/j.1469-8749.2009.03471.x · Cited by 63 · see citing works
Tonduti, D., Vanderver, A. L., Berardinelli, A. L., Schmidt, J., Collins, C. D., Novara, F., Genni, A. D., Mita, A., Triulzi, F. M., Brunstrom-Hernandez, J. E., et al. (2012) · Journal of Child Neurology · doi:10.1177/0883073812450944 · Cited by 61 · see citing works
Appelbaum, L., Skariah, G. M., Mourrain, P., & Mignot, E. J.-M. (2007) · Brain Research · doi:10.1016/j.brainres.2007.06.103 · Cited by 58 · see citing works
Self-citationKazakova, N., Li, H., Mora, A., Jessen, K., Mirsky, R., Richardson, W., & Smith, H. (2006) · Developmental Biology · doi:10.1016/j.ydbio.2006.03.020 · Cited by 58 · see citing works
Small molecule screening in zebrafish: swimming in potential drug therapies
Tamplin, O. J., White, R. M., Jing, L., Kaufman, C. K., Lacadie, S. A., Li, P., Taylor, A. M., & Zon, L. I. (2012) · Wiley Interdisciplinary Reviews Developmental Biology · doi:10.1002/wdev.37 · Cited by 56 · see citing works
Treichel, D., Becker, M.-B., & Gruß, P. (2001) · Mechanisms of Development · doi:10.1016/s0925-4773(00)00544-x · Cited by 53 · see citing works
Jeannin, E., Robyr, D., & Desvergne, B. (1998) · Journal of Biological Chemistry · doi:10.1074/jbc.273.37.24239 · Cited by 51 · see citing works
Triac regulation of transcription is T3 receptor isoform- and response element-specific
Messier, N. (2000) · Molecular and Cellular Endocrinology · doi:10.1016/s0303-7207(00)00266-5 · Cited by 50 · see citing works
Appelbaum, L., Toyama, R., Dawid, I. B., Klein, D. C., Baler, R., & Gothilf, Y. (2004) · Molecular Endocrinology · doi:10.1210/me.2003-0439 · Cited by 49 · see citing works
Self-citationSchebesta, M., & Serluca, F. C. (2009) · Developmental Dynamics · doi:10.1002/dvdy.21909 · Cited by 47 · see citing works
Tovin, A., Alon, S., Ben-Moshe, Z., Mracek, P., Vatine, G. D., Foulkes, N. S., Jacob‐Hirsch, J., Rechavi, G., Toyama, R., Coon, S. L., et al. (2012) · PLoS Genetics · doi:10.1371/journal.pgen.1003116 · Cited by 46 · see citing works
Self-citationCosmo, C. D., Liao, X.-H., Ye, H., Ferrara, A. M., Weiss, R. E., Refetoff, S., & Dumitrescu, A. M. (2013) · Endocrinology · doi:10.1210/en.2013-1150 · Cited by 45 · see citing works
1H Magnetic Resonance Spectroscopy in Monocarboxylate Transporter 8 Gene Deficiency
Sijens, P. E., Rödiger, L. A., Meiners, L. C., & Lunsing, R. J. (2008) · The Journal of Clinical Endocrinology & Metabolism · doi:10.1210/jc.2007-2441 · Cited by 45 · see citing works
Thyroid hormone transporters and deiodinases in the developing human hypothalamus
Friesema, E. C. H., Visser, T. J., Borgers, A. J., Kalsbeek, A., Swaab, D. F., Fliers, E., & Alkemade, A. (2012) · European Journal of Endocrinology · doi:10.1530/eje-12-0177 · Cited by 45 · see citing works
Mutations in MCT8 in patients with Allan-Herndon-Dudley-syndrome affecting its cellular distribution
Kersseboom, S., Kremers, G.-J., Friesema, E. C. H., Visser, W. E., Klootwijk, W., Peeters, R. P., & Visser, T. J. (2013) · Molecular Endocrinology · doi:10.1210/me.2012-1356 · Cited by 45 · see citing works
Filippi, A., Jainok, C., & Driever, W. (2012) · Developmental Biology · doi:10.1016/j.ydbio.2012.06.010 · Cited by 43 · see citing works
LAR Receptor Tyrosine Phosphatases and HSPGs Guide Peripheral Sensory Axons to the Skin
Wang, F., Wolfson, S. N., Gharib, A., & Sagasti, A. (2012) · Current Biology · doi:10.1016/j.cub.2012.01.040 · Cited by 42 · see citing works
Inherited defects of thyroid hormone-cell-membrane transport
Fu, J., Refetoff, S., & Dumitrescu, A. M. (2013) · Current Opinion in Endocrinology Diabetes and Obesity · doi:10.1097/01.med.0000432531.03233.ad · Cited by 36 · see citing works
Dendritic Anomalies in Disorders Associated With Mental Retardation
Moser, H. W. (1999) · Developmental Neuropsychology · doi:10.1207/s15326942dn1603_18 · Cited by 28 · see citing works
Boccone, L., Dessì, V., Meloni, A., & Loudianos, G. (2013) · European Journal of Medical Genetics · doi:10.1016/j.ejmg.2013.02.001 · Cited by 28 · see citing works
Thyroid hormone transport in developing brain
Bernal, J. (2011) · Current Opinion in Endocrinology Diabetes and Obesity · doi:10.1097/med.0b013e32834a78b3 · Cited by 24 · see citing works
Simpson, H. D., Kita, E. M., Scott, E. K., & Goodhill, G. J. (2012) · The Journal of Comparative Neurology · doi:10.1002/cne.23248 · Cited by 23 · see citing works
Influence of thyroid in nervous system growth.
Mussa, G. C., Mussa, F., Bretto, R., Zambelli, M. C., & SILVESTRO, L. (2001) · PubMed · Cited by 23 · see citing works
Rodrigues, T. B., Ceballos, A., Grijota-Martínez, C., Núñez, B., Refetoff, S., Cerdán, S., Morte, B., & Bernal, J. (2013) · PLoS ONE · doi:10.1371/journal.pone.0074621 · Cited by 21 · see citing works
Gne depletion during zebrafish development impairs skeletal muscle structure and function
Daya, A., Vatine, G. D., Becker‐Cohen, M., Tal-Goldberg, T., Friedmann, A., Gothilf, Y., Du, S., & Mitrani‐Rosenbaum, S. (2014) · Human Molecular Genetics · doi:10.1093/hmg/ddu045 · Cited by 20 · see citing works
Self-citationVerhoeven, F. A., van der Putten, H., HENNEMANN, G., Lamers, J. M. J., Visser, T. J., & Everts, M. E. (2002) · Journal of Endocrinology · doi:10.1677/joe.0.1730247 · Cited by 18 · see citing works
Imaging circuit formation in zebrafish
Nikolaou, N., & Meyer, M. P. (2011) · Developmental Neurobiology · doi:10.1002/dneu.20874 · Cited by 15 · see citing works
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Thyroid hormone transporters—functions and clinical implications
Bernal, J., Guadaño‐Ferraz, A., & Morte, B. (2015) · Nature Reviews Endocrinology · doi:10.1038/nrendo.2015.66 · Cited by 293 · see citing works
Vatine, G. D., Al-Ahmad, A., Barriga, B. K., Svendsen, S., Salim, A., Garcia, L., Garcia, V. J., Ho, R., Yucer, N., Qian, T., et al. (2017) · Cell Stem Cell · doi:10.1016/j.stem.2017.04.002 · Cited by 233 · see citing works
Self-citationGroeneweg, S., van Geest, F. S., Peeters, R. P., Heuer, H., & Visser, W. E. (2020) · Endocrine Reviews · doi:10.1210/endrev/bnz008 · Cited by 216 · see citing works
Zebrafish Models of Human Disease: Gaining Insight into Human Disease at ZFIN
Bradford, Y. M., Toro, S., Ramachandran, S., Ruzicka, L., Howe, D. G., Eagle, A. E., Kalita, P., Martin, R., Moxon, S. A. T., Schaper, K., et al. (2016) · ILAR Journal · doi:10.1093/ilar/ilw040 · Cited by 175 · see citing works
Shamay-Ramot, A., Khermesh, K., Porath, H. T., Barak, M., Pinto, Y., Wachtel, C., Zilberberg, A., Lerer‐Goldshtein, T., Efroni, S., Levanon, E. Y., et al. (2015) · PLoS Genetics · doi:10.1371/journal.pgen.1005702 · Cited by 83 · see citing works
Self-citationTriclosan affects axon formation in the neural development stages of zebrafish embryos (Danio rerio)
Kim, J., Oh, H., Ryu, B., Kim, U., Lee, J. M., Jung, C., Kim, C., & Park, J. (2018) · Environmental Pollution · doi:10.1016/j.envpol.2017.12.110 · Cited by 80 · see citing works
Sleep and Development in Genetically Tractable Model Organisms
Kayser, M. S., & Biron, D. (2016) · Genetics · doi:10.1534/genetics.116.189589 · Cited by 78 · see citing works
Comparative Analysis of Neurotoxicity of Six Phthalates in Zebrafish Embryos
Tran, C. M., Do, T., & Kim, K. (2021) · Toxics · doi:10.3390/toxics9010005 · Cited by 70 · see citing works
Thyroid Hormone Signaling in Oligodendrocytes: from Extracellular Transport to Intracellular Signal
Lee, J. Y., & Petratos, S. (2016) · Molecular Neurobiology · doi:10.1007/s12035-016-0013-1 · Cited by 70 · see citing works
Walter, K. M., Miller, G. W., Chen, X., Yaghoobi, B., Puschner, B., & Lein, P. J. (2018) · General and Comparative Endocrinology · doi:10.1016/j.ygcen.2018.11.007 · Cited by 66 · see citing works
Marelli, F., Carra, S., Agostini, M., Cotelli, F., Peeters, R. P., Chatterjee, K., & Persani, L. (2016) · Molecular and Cellular Endocrinology · doi:10.1016/j.mce.2016.01.020 · Cited by 65 · see citing works
Zada, D., Tovin, A., Lerer‐Goldshtein, T., & Appelbaum, L. (2016) · Disease Models & Mechanisms · doi:10.1242/dmm.027227 · Cited by 64 · see citing works
Self-citationHoubrechts, A. M., Delarue, J., Gabriëls, I. J., Sourbron, J., & Darras, V. (2016) · Endocrinology · doi:10.1210/en.2016-1077 · Cited by 61 · see citing works
Thyroid hormone regulation of neural stem cell fate: From development to ageing
Gothié, J. M., Vancamp, P., DEMENEIX, B. A., & Remaud, S. (2019) · Acta Physiologica · doi:10.1111/apha.13316 · Cited by 51 · see citing works
Bárez‐López, S., Grijota-Martínez, C., Ausó, E., Frutos, M. F.-D., Montero‐Pedrazuela, A., & Guadaño‐Ferraz, A. (2019) · Thyroid · doi:10.1089/thy.2019.0068 · Cited by 51 · see citing works
MCT8 Deficiency: The Road to Therapies for a Rare Disease
Grijota-Martínez, C., Bárez‐López, S., Gómez‐Andrés, D., & Guadaño‐Ferraz, A. (2020) · Frontiers in Neuroscience · doi:10.3389/fnins.2020.00380 · Cited by 50 · see citing works
Testing for thyroid hormone disruptors, a review of non-mammalian in vivo models
Couderq, S., Leemans, M., & Fini, J. (2020) · Molecular and Cellular Endocrinology · doi:10.1016/j.mce.2020.110779 · Cited by 47 · see citing works
Pagnin, M., Kondos‐Devcic, D., Chincarini, G., Cumberland, A. L., Richardson, S. J., & Tolcos, M. (2021) · Frontiers in Neuroendocrinology · doi:10.1016/j.yfrne.2021.100901 · Cited by 47 · see citing works
Modeling sleep and neuropsychiatric disorders in zebrafish
Levitas‐Djerbi, T., & Appelbaum, L. (2017) · Current Opinion in Neurobiology · doi:10.1016/j.conb.2017.02.017 · Cited by 46 · see citing works
Self-citationMonocarboxylate Transporter 8 Deficiency: Delayed or Permanent Hypomyelination?
Vancamp, P., DEMENEIX, B. A., & Remaud, S. (2020) · Frontiers in Endocrinology · doi:10.3389/fendo.2020.00283 · Cited by 43 · see citing works
Haigis, A.-C., Vergauwen, L., LaLone, C. A., Villeneuve, D. L., O’Brien, J. M., & Knapen, D. (2023) · Toxicological Sciences · doi:10.1093/toxsci/kfad063 · Cited by 40 · see citing works
How zebrafish research has helped in understanding thyroid diseases
Marelli, F., & Persani, L. (2017) · F1000Research · doi:10.12688/f1000research.12142.1 · Cited by 40 · see citing works
Lee, J. Y., Kim, M. J., Deliyanti, D., Azari, M. F., Rossello, F. J., Costin, A. J., Ramm, G., Stanley, E. G., Elefanty, A. G., Wilkinson‐Berka, J. L., et al. (2017) · EBioMedicine · doi:10.1016/j.ebiom.2017.10.016 · Cited by 38 · see citing works
Vancamp, P., Houbrechts, A. M., & Darras, V. (2018) · General and Comparative Endocrinology · doi:10.1016/j.ygcen.2018.09.011 · Cited by 40 · see citing works
Novel hypophysiotropic AgRP2 neurons and pineal cells revealed by BAC transgenesis in zebrafish
Shainer, I., Hazak, A., Hawkins, T. A., Wilson, S. W., Cone, R. D., & Gothilf, Y. (2017) · Scientific Reports · doi:10.1038/srep44777 · Cited by 37 · see citing works
Sleep loss and structural plasticity
Areal, C. C., Warby, S. C., & Mongrain, V. (2017) · Current Opinion in Neurobiology · doi:10.1016/j.conb.2016.12.010 · Cited by 35 · see citing works
MCT8 deficiency in Purkinje cells disrupts embryonic chicken cerebellar development
Delbaere, J., Vancamp, P., Herck, S. L. J. V., Bourgeois, N. M. A., Green, M. J., Wingate, R. J. T., & Darras, V. (2016) · Journal of Endocrinology · doi:10.1530/joe-16-0323 · Cited by 34 · see citing works
Lazcano, I., Pech-Pool, S. M., Olvera, A., García-Martínez, I., Palacios-Pérez, S., & Orozco, A. (2023) · General and Comparative Endocrinology · doi:10.1016/j.ygcen.2023.114225 · Cited by 34 · see citing works
Vancamp, P., & Darras, V. (2017) · General and Comparative Endocrinology · doi:10.1016/j.ygcen.2017.11.023 · Cited by 33 · see citing works
Understanding neurobehavioral genetics of zebrafish
Cheresiz, S. V., Volgin, A. D., Evsyukova, A. K., Bashirzade, A. A., Demin, K. A., de Abreu, M. S., Amstislavskaya, T. G., & Kalueff, A. V. (2020) · Journal of Neurogenetics · doi:10.1080/01677063.2019.1698565 · Cited by 30 · see citing works
Imaging Approaches to Investigate Pathophysiological Mechanisms of Brain Disease in Zebrafish
Turrini, L., Roschi, L., de Vito, G., Pavone, F. S., & Vanzi, F. (2023) · International Journal of Molecular Sciences · doi:10.3390/ijms24129833 · Cited by 30 · see citing works
Valcárcel-Hernández, V., López-Espíndola, D., Guillén-Yunta, M., García-Aldea, Á., Soler, I. L. D. T., Bárez‐López, S., & Guadaño‐Ferraz, A. (2021) · Neurobiology of Disease · doi:10.1016/j.nbd.2021.105567 · Cited by 28 · see citing works
Thyroid hormone deficiency during zebrafish development impairs central nervous system myelination
Farías-Serratos, B. M., Lazcano, I., Villalobos, P., Darras, V., & Orozco, A. (2021) · PLoS ONE · doi:10.1371/journal.pone.0256207 · Cited by 26 · see citing works
Vancamp, P., Deprez, M.-A., Remmerie, M., & Darras, V. (2017) · Journal of Neuroscience · doi:10.1523/jneurosci.1917-17.2017 · Cited by 26 · see citing works
Vancamp, P., Bourgeois, N. M. A., Houbrechts, A. M., & Darras, V. (2018) · Experimental Eye Research · doi:10.1016/j.exer.2018.09.018 · Cited by 26 · see citing works
Elbaz, I., Zada, D., Tovin, A., Braun, T., Lerer‐Goldshtein, T., Wang, G. X., Mourrain, P., & Appelbaum, L. (2016) · Molecular Neurobiology · doi:10.1007/s12035-016-0175-x · Cited by 25 · see citing works
Self-citationSong, J., Lu, Y., Cheng, X., Shi, C., Lou, Q., Jin, X., He, J., Zhai, G., & Yin, Z. (2021) · Cells · doi:10.3390/cells10081984 · Cited by 24 · see citing works
Hypothalamic leptin‐neurotensin‐hypocretin neuronal networks in zebrafish
Levitas‐Djerbi, T., Yelin‐Bekerman, L., Lerer‐Goldshtein, T., & Appelbaum, L. (2014) · The Journal of Comparative Neurology · doi:10.1002/cne.23716 · Cited by 24 · see citing works
Self-citationElbaz, I., Lerer‐Goldshtein, T., Okamoto, H., & Appelbaum, L. (2014) · The FASEB Journal · doi:10.1096/fj.14-258350 · Cited by 23 · see citing works
Self-citationZebrafish – An emerging model to explore thyroid hormone transporters and psychomotor retardation
Zada, D., Blitz, E., & Appelbaum, L. (2017) · Molecular and Cellular Endocrinology · doi:10.1016/j.mce.2017.03.004 · Cited by 23 · see citing works
Self-citationOligodendroglial Lineage Cells in Thyroid Hormone-Deprived Conditions
Kim, M. J., & Petratos, S. (2019) · Stem Cells International · doi:10.1155/2019/5496891 · Cited by 23 · see citing works
Admati, I., Wasserman-Bartov, T., Tovin, A., Rozenblat, R., Blitz, E., Zada, D., Lerer‐Goldshtein, T., & Appelbaum, L. (2019) · Thyroid · doi:10.1089/thy.2019.0320 · Cited by 23 · see citing works
Self-citationVatine, G. D., Shelest, O., Barriga, B. K., Ofan, R., Rabinski, T., Mattis, V. B., Heuer, H., & Svendsen, C. N. (2021) · Glia · doi:10.1002/glia.24014 · Cited by 23 · see citing works
Self-citationKeene, A. C., & Appelbaum, L. (2019) · Handbook of behavioral neuroscience · doi:10.1016/b978-0-12-813743-7.00024-4 · Cited by 22 · see citing works
Self-citationRapid habituation of a touch-induced escape response in Zebrafish (Danio rerio) Larvae
Roberts, A. C., Chornak, J., Alzagatiti, J. B., Ly, T., Bill, B., Trinkeller, J., Pearce, K. C., Choe, R. C., Campbell, C. S., Wong, D., et al. (2019) · PLoS ONE · doi:10.1371/journal.pone.0214374 · Cited by 21 · see citing works
Naef, V., Mero, S., Fichi, G., D’Amore, A., Ogi, A., Gemignani, F., Santorelli, F. M., & Marchese, M. (2019) · Frontiers in Neuroscience · doi:10.3389/fnins.2019.01311 · Cited by 20 · see citing works
Zebrafish as an emerging tool for drug discovery and development for thyroid diseases
Yadav, P., Sarode, L. P., Gaddam, R. R., Kumar, P., Bhatti, J. S., Khurana, A., & Navik, U. (2022) · Fish & Shellfish Immunology · doi:10.1016/j.fsi.2022.09.001 · Cited by 19 · see citing works
Robo2 regulates synaptic oxytocin content by affecting actin dynamics
Anbalagan, S., Blechman, J., Gliksberg, M., Gordon, L., Rotkopf, R., Dadosh, T., Shimoni, E., & Levkowitz, G. (2019) · eLife · doi:10.7554/elife.45650 · Cited by 19 · see citing works
The Toolbox for Conditional Zebrafish Cancer Models
Mayrhofer, M., & Mione, M. (2016) · Advances in experimental medicine and biology · doi:10.1007/978-3-319-30654-4_2 · Cited by 18 · see citing works
Chen, J., Salveridou, E., Liebmann, L., Sundaram, S. M., Doycheva, D., Markova, B., Hübner, C. A., Boelen, A., Visser, W. E., Heuer, H., et al. (2023) · International Journal of Molecular Sciences · doi:10.3390/ijms24043452 · Cited by 17 · see citing works
In Vitro Characterization of Human, Mouse, and Zebrafish MCT8 Orthologues
Groeneweg, S., Kersseboom, S., van den Berge, A., Dolcetta‐Capuzzo, A., van Geest, F. S., van Heerebeek, R. E. A., Arjona, F. J., Meima, M. E., Peeters, R. P., Visser, W. E., et al. (2019) · Thyroid · doi:10.1089/thy.2019.0009 · Cited by 17 · see citing works
Erratum: Thyroid hormone transporters—functions and clinical implications
Bernal, J., Guadaño‐Ferraz, A., & Morte, B. (2015) · Nature Reviews Endocrinology · doi:10.1038/nrendo.2015.113 · Cited by 17 · see citing works
Gölz, L., Blanc, M., Rinderknecht, M., Behnstedt, L., Coordes, S., Reger, L., Sire, S., Cousin, X., Braunbeck, T. A., & Baumann, L. (2024) · Environmental Toxicology and Chemistry · doi:10.1002/etc.5878 · Cited by 16 · see citing works
Functional Characterization of Xenopus Thyroid Hormone Transporters mct8 and oatp1c1
Mughal, B. B., Leemans, M., de Souza, E. C. L., Mével, S. L., Špirhanzlová, P., Visser, T. J., Fini, J., & DEMENEIX, B. A. (2017) · Endocrinology · doi:10.1210/en.2017-00108 · Cited by 15 · see citing works
Delbaere, J., Herck, S. L. J. V., Bourgeois, N. M. A., Vancamp, P., Yang, S., Wingate, R. J. T., & Darras, V. (2015) · The Cerebellum · doi:10.1007/s12311-015-0744-y · Cited by 15 · see citing works
Thyroid Hormone Hyposensitivity: From Genotype to Phenotype and Back
Rurale, G., Cicco, E. D., Dentice, M., Salvatore, D., Persani, L., Marelli, F., & Luongo, C. (2020) · Frontiers in Endocrinology · doi:10.3389/fendo.2019.00912 · Cited by 13 · see citing works
Induction of Short-Term Sensitization by an Aversive Chemical Stimulus in Zebrafish Larvae
Roberts, A. C., Alzagatiti, J. B., Ly, T., Chornak, J., Ma, Y., Razee, A., Zavradyan, G., Khan, U., Lewis, J., Natarajan, A., et al. (2020) · eNeuro · doi:10.1523/eneuro.0336-19.2020 · Cited by 13 · see citing works
Emamnejad, R., Dass, M., Mahlis, M., Bozkurt, S., Ye, S., Pagnin, M., Theotokis, P., Grigoriadis, N., & Petratos, S. (2022) · Frontiers in Pharmacology · doi:10.3389/fphar.2022.934971 · Cited by 13 · see citing works
Evaluating the association of biallelic OGDHL variants with significant phenotypic heterogeneity
Lin, S., Vona, B., Lau, T., Huang, K., Zaki, M. S., Aldeen, H. S., Karimiani, E. G., Rocca, C., Noureldeen, M. M., Saad, A. K., et al. (2023) · Genome Medicine · doi:10.1186/s13073-023-01258-4 · Cited by 12 · see citing works
Toxicity and teratogenicity in zebrafish Danio rerio embryos exposed to chromium
Sánchez-Olivares, M. A., Gaytán-Oyarzún, J. C., Gordillo-Martínez, A. J., García, F. P., & Cabrera-Cruz, R. B. E. (2021) · Latin American Journal of Aquatic Research · doi:10.3856/vol49-issue2-fulltext-2561 · Cited by 12 · see citing works
Valcárcel-Hernández, V., Guillén-Yunta, M., Bueno-Arribas, M., Montero‐Pedrazuela, A., Grijota-Martínez, C., Markossian, S., García-Aldea, Á., Flamant, F., Bárez‐López, S., & Guadaño‐Ferraz, A. (2022) · Neurobiology of Disease · doi:10.1016/j.nbd.2022.105896 · Cited by 12 · see citing works
NGLY1 Deficiency Zebrafish Model Manifests Abnormalities of the Nervous and Musculoskeletal Systems
Mesika, A., Nadav, G., Shochat, C., Kalfon, L., Jackson, K., Khalaileh, A., Karasik, D., & Falik‐Zaccai, T. C. (2022) · Frontiers in Cell and Developmental Biology · doi:10.3389/fcell.2022.902969 · Cited by 10 · see citing works
Curpăn, A.-Ș., Balmuș, I.-M., Dobrin, R., Ciobica, A. S., Chele, G. E., Gorgan, L., & Boloș, A. (2022) · Brain Sciences · doi:10.3390/brainsci12091147 · Cited by 9 · see citing works
Genetic and Neurological Deficiencies in the Visual System of mct8 Mutant Zebrafish
Rozenblat, R., Tovin, A., Zada, D., Lebenthal-Loinger, I., Lerer‐Goldshtein, T., & Appelbaum, L. (2022) · International Journal of Molecular Sciences · doi:10.3390/ijms23052464 · Cited by 9 · see citing works
Self-citationTsh Induces Agrp1 Neuron Proliferation in Oatp1c1-Deficient Zebrafish
Wasserman-Bartov, T., Admati, I., Lebenthal-Loinger, I., Sharabany, J., Lerer‐Goldshtein, T., & Appelbaum, L. (2022) · Journal of Neuroscience · doi:10.1523/jneurosci.0002-22.2022 · Cited by 9 · see citing works
Self-citationDiving deep: zebrafish models in motor neuron degeneration research
Garg, V., & Geurten, B. R. H. (2024) · Frontiers in Neuroscience · doi:10.3389/fnins.2024.1424025 · Cited by 8 · see citing works
Convergent thyroid-ATPase interactions regulate collective behavior in Danionella
Zada, D., Kadobianskyi, M., Judkewitz, B., & Lovett-Barron, M. (2025) · Cell Reports · doi:10.1016/j.celrep.2025.116730 · Cited by 8 · see citing works
Self-citationUsing Zebrafish to Assess Developmental Neurotoxicity
Padilla, S., MacPhail, R. C., & Legradi, J. (2017) · Elsevier eBooks · doi:10.1016/b978-0-12-804239-7.00016-0 · Cited by 8 · see citing works
Chincarini, G., Walker, D. W., Wong, F. Y., Richardson, S. J., Cumberland, A. L., & Tolcos, M. (2024) · Journal of Neurochemistry · doi:10.1111/jnc.16124 · Cited by 7 · see citing works
Silva, N., & Campinho, M. A. (2023) · Frontiers in Endocrinology · doi:10.3389/fendo.2023.1157685 · Cited by 6 · see citing works
Thyroid Hormones Regulate Goblet Cell Differentiation and Fgf19-Fgfr4 Signaling
Blitz, E., Matsuda, H., Günther, S., Morikawa, T., Kubota, Y., Zada, D., Lerer‐Goldshtein, T., Stainier, D. Y. R., & Appelbaum, L. (2021) · Endocrinology · doi:10.1210/endocr/bqab047 · Cited by 6 · see citing works
Self-citationNeurogranin-like immunoreactivity in the zebrafish brain during development
Alba‐González, A., Yáñez, J., Anadón, R., & Folgueira, M. (2022) · Brain Structure and Function · doi:10.1007/s00429-022-02550-6 · Cited by 5 · see citing works
Alba‐González, A., Folgueira, M., Castro, A., Anadón, R., & Yáñez, J. (2022) · The Journal of Comparative Neurology · doi:10.1002/cne.25297 · Cited by 5 · see citing works
Pez cebra (Danio rerio): modelo experimental en la evaluación de compuestos xenobióticos
Castillo-Salas, D. L., Gaytán-Oyarzún, J. C., Herrera, M. L., & Sánchez-Olivares, M. A. (2022) · PÄDI Boletín Científico de Ciencias Básicas e Ingenierías del ICBI · doi:10.29057/icbi.v10i19.8870 · Cited by 4 · see citing works
Vergauwen, L., Tait, S., Bajard, L., Buławska, N., Gutleb, A. C., Hessel, E. V. S., Hilscherová, K., Holbech, H., Kowalska, D., Novák, J., et al. (2026) · Environment International · doi:10.1016/j.envint.2026.110451 · Cited by 3 · see citing works
Role of TRs in Zebrafish Development
Marelli, F., & Persani, L. (2018) · Methods in molecular biology · doi:10.1007/978-1-4939-7902-8_22 · Cited by 3 · see citing works
Studying Human Genetic Variation in Zebrafish
Lepanto, P., Zolessi, F. R., & Badano, J. L. (2019) · Elsevier eBooks · doi:10.1016/b978-0-12-816573-7.00005-5 · Cited by 3 · see citing works
Mutagenesis and Transgenesis in Zebrafish
Poulain, F. E. (2017) · Springer eBooks · doi:10.1007/978-3-319-33774-6_1 · Cited by 3 · see citing works
Abe, T., Ichimura, M., & Kudo, H. (2022) · Fish Physiology and Biochemistry · doi:10.1007/s10695-022-01063-7 · Cited by 2 · see citing works
Identification of Iodotyrosines as Novel Substrates for the Thyroid Hormone Transporter MCT8
Groeneweg, S., Zevenbergen, C., de Souza, E. C. L., van Geest, F. S., Kloeckener‐Gruissem, B., Laczkó, E., Camargo, S. M. R., Meima, M. E., Peeters, R. P., & Visser, W. E. (2024) · Thyroid · doi:10.1089/thy.2023.0551 · Cited by 2 · see citing works
Sciatic nerve analysis in thyroid hormone transporters Mct8 and Oatp1c1 knockout mice
Mayerl, S. E., Alcaide, M. A., Bauer, R., & Heuer, H. (2025) · European Thyroid Journal · doi:10.1530/etj-24-0248 · Cited by 1 · see citing works
Alzagatiti, J. B., Salazar, L., Brown, H., Rosas, G., Jaber, L. A., Minaya, J. L., Scaramella, C., Roberts, A. C., & Glanzman, D. L. (2025) · Journal of Visualized Experiments · doi:10.3791/67301 · Cited by 1 · see citing works
The not-so-long history of zebrafish research in Israel
Blechman, J., Levkowitz, G., & Gothilf, Y. (2017) · The International Journal of Developmental Biology · doi:10.1387/ijdb.160346gl · Cited by 1 · see citing works
Combined deletion of Mct8 and Dio2 impairs SVZ neurogliogenesis and olfactory function in adult mice
Valcárcel-Hernández, V., Vancamp, P., Butruille, L., Remaud, S., & Guadaño‐Ferraz, A. (2024) · Neurobiology of Disease · doi:10.1016/j.nbd.2024.106572 · Cited by 1 · see citing works
Zebrafish come modello per lo studio di malattie della tiroide
Marelli, F., & Persani, L. (2021) · L Endocrinologo · doi:10.1007/s40619-021-00833-4 · Cited by 1 · see citing works
Using zebrafish to assess developmental neurotoxicity
Padilla, S., Hill, B. N., Legradi, J., & Klüver, N. (2022) · Elsevier eBooks · doi:10.1016/b978-0-323-89773-0.00013-8
Lin, S., Vona, B., Lau, T., Huang, K., Zaki, M. S., Aldeen, H. S., Karimiani, E. G., Rocca, C., Noureldeen, M. M., Saad, A. K., et al. (2023) · Figshare · doi:10.6084/m9.figshare.24670002
Cellular Transporters for Thyroid Hormones
Bernal, J. (2024) · BENTHAM SCIENCE PUBLISHERS eBooks · doi:10.2174/9789815274226124010010
Lin, S.-J., Vona, B., Lau, T., Huang, K., Zaki, M. S., Aldeen, H. S., Karimiani, E. G., Rocca, C., Noureldeen, M. M., Saad, A. K., et al. (2024) · Figshare · doi:10.6084/m9.figshare.26647047
Lin, S.-J., Vona, B., Lau, T., Huang, K., Zaki, M. S., Aldeen, H. S., Karimiani, E. G., Rocca, C., Noureldeen, M. M., Saad, A. K., et al. (2024) · Figshare · doi:10.6084/m9.figshare.26647053
Lin, S.-J., Vona, B., Lau, T., Huang, K., Zaki, M. S., Aldeen, H. S., Karimiani, E. G., Rocca, C., Noureldeen, M. M., Saad, A. K., et al. (2024) · Figshare · doi:10.6084/m9.figshare.24670323
Lin, S.-J., Vona, B., Lau, T., Huang, K., Zaki, M. S., Aldeen, H. S., Karimiani, E. G., Rocca, C., Noureldeen, M. M., Saad, A. K., et al. (2024) · Figshare · doi:10.6084/m9.figshare.26647050
Lin, S.-J., Vona, B., Lau, T., Huang, K., Zaki, M. S., Aldeen, H. S., Karimiani, E. G., Rocca, C., Noureldeen, M. M., Saad, A. K., et al. (2024) · Figshare · doi:10.6084/m9.figshare.26647044
Kulkarni, A. (2017) · theses.fr (ABES) · doi:10.70675/b28867a5zcc5dz4649z8912za9ace126d231
Alcaide-Martin, A., Budeus, B., Bauer, R., Boelen, A., Schwaninger, M., Führer, D., Heuer, H., & Mayerl, S. E. (2026) · Experimental Neurology · doi:10.1016/j.expneurol.2026.115962
- Authors
- Zada, D., Tovin, A., Lerer-Goldshtein, T., Vatine, G. D., & Appelbaum, L.
- Affiliations
- Bar-Ilan University (IL)
- Cedars-Sinai Medical Center (US)
- Corresponding author
- Appelbaum, Lior
- Venue
- PLoS Genetics
- Publisher
- Public Library of Science (PLoS)
- Published
- 2014-09-25
- Volume
- 10
- Issue
- 9
- Pages
- e1004615
- ISSN
- 1553-7390 (ISSN-L)
- 1553-7404
- PMC
- PMC4177677
- OpenAlex
- W1999728382
- Cited by
- 92
- References
- 108
- Citations by year
- 2026: 2 · 2025: 3 · 2024: 7 · 2023: 6 · 2022: 13 · 2021: 10 · 2020: 6 · 2019: 11 · 2018: 5 · 2017: 14 · 2016: 9 · 2015: 4 · 2014: 2
- FWCI
- 4.82
- Type
- journal article
- Language
- en
- Open access
- gold
- doi.org
- Locations
- PLoS Genetics (publishedVersion, cc-by)
- PubMed Central (publishedVersion, cc-by)
- PubMed (publishedVersion)
- DOAJ (DOAJ: Directory of Open Access Journals) (submittedVersion, cc-by-sa)
- Figshare (submittedVersion, cc-by)
- Funders
- E-Rare
- European Commission (274333)
- Bar-Ilan University
- Israel Science Foundation (366/11)
- Ministry of Health, State of Israel
- FP7 People: Marie-Curie Actions (FP7-PEOPLE-2010-RG)
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Cell Biology › Zebrafish Biomedical Research Applications
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetics and Neurodevelopmental Disorders
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › Developmental Biology and Gene Regulation
- Keywords
- Allan-Herndon-Dudley syndrome, MCT8, thyroid hormone transport, zebrafish mutants, behavioral deficits, locomotor activity, sleep, myelin genes, axon branching, sensory neurons, motor neurons, synaptic density, live imaging, thyroid hormone analogs, neural circuit assembly, intellectual disability
- MeSH terms
- Animals, Disease Models, Animal, Gene Expression Regulation, Hypothalamo-Hypophyseal System, Motor Activity, Muscle Hypotonia, Muscular Atrophy, Mutation, Myelin Sheath, Neurons, Phenotype, Pseudopodia, Thyroid Gland, Thyroid Hormones, Zebrafish, Zinc Fingers, Photoperiod, Gene Targeting, Gene Order, Monocarboxylic Acid Transporters, Animals, Genetically Modified, Thyroid Hormone Receptors alpha, Mental Retardation, X-Linked, X-Linked Intellectual Disability, Neurogranin, Kruppel-Like Transcription Factors, Gene Knockout Techniques
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