Abramsson, A., Kettunen, P., Banote, R.K., Lott, E., Li, M., Arner, A., and Zetterberg, H. (2013) The zebrafish amyloid precursor protein-b is required for motor neuron guidance and synapse formation. Developmental Biology. 381(2):377-88
|
Ahuja, G., Ivandi, I., Saltürk, M., Oka, Y., Nadler, W., and Korsching, S.I. (2013) Zebrafish crypt neurons project to a single, identified mediodorsal glomerulus. Scientific Reports. 3:2063
|
|
|
|
|
Appelbaum, L., Wang, G., Yokogawa, T., Skariah, G.M., Smith, S.J., Mourrain, P., and Mignot, E. (2010) Circadian and homeostatic regulation of structural synaptic plasticity in hypocretin neurons. Neuron. 68(1):89-98
|
|
|
Asante, E., Hummel, D., Gurung, S., Kassim, Y.M., Al-Shakarji, N., Palaniappan, K., Sittaramane, V., Chandrasekhar, A. (2021) Defective Neuronal Positioning Correlates With Aberrant Motor Circuit Function in Zebrafish. Frontiers in neural circuits. 15:690475
|
Bailey, E.C., Alrowaished, S.S., Kilroy, E.A., Crooks, E.S., Drinkert, D.M., Karunasiri, C.M., Belanger, J.J., Khalil, A., Kelley, J.B., Henry, C.A. (2019) NAD+ improves neuromuscular development in a zebrafish model of FKRP-associated dystroglycanopathy. Skeletal muscle. 9:21
|
Banerjee, S., Gordon, L., Donn, T.M., Berti, C., Moens, C.B., Burden, S.J., and Granato, M. (2011) A novel role for MuSK and non-canonical Wnt signaling during segmental neural crest cell migration. Development (Cambridge, England). 138(15):3287-3296
|
|
|
|
|
|
|
|
|
|
|
Beckers, A., Van Dyck, A., Bollaerts, I., Van Houcke, J., Lefevere, E., Andries, L., Agostinone, J., Van Hove, I., Di Polo, A., Lemmens, K., Moons, L. (2018) An Antagonistic Axon-Dendrite Interplay Enables Efficient Neuronal Repair in the Adult Zebrafish Central Nervous System. Molecular neurobiology. 56(5):3175-3192
|
Bento-Abreu, A., Jager, G., Swinnen, B., Rué, L., Hendrickx, S., Jones, A., Staats, K.A., Taes, I., Eykens, C., Nonneman, A., Nuyts, R., Timmers, M., Silva, L., Chariot, A., Nguyen, L., Ravits, J., Lemmens, R., Cabooter, D., Ludo, V.D.B., Van Damme, P., Al-Chalabi, A., Bystrom, A., Robberecht, W. (2018) Elongator subunit 3 (ELP3) modifies ALS through tRNA modification. Human molecular genetics. 27(7):1276-1289
|
Bhattarai, P., Gunasekaran, T.I., Belloy, M.E., Reyes-Dumeyer, D., Jülich, D., Tayran, H., Yilmaz, E., Flaherty, D., Turgutalp, B., Sukumar, G., Alba, C., McGrath, E.M., Hupalo, D.N., Bacikova, D., Le Guen, Y., Lantigua, R., Medrano, M., Rivera, D., Recio, P., Nuriel, T., Ertekin-Taner, N., Teich, A.F., Dickson, D.W., Holley, S., Greicius, M., Dalgard, C.L., Zody, M., Mayeux, R., Kizil, C., Vardarajan, B.N. (2024) Rare genetic variation in fibronectin 1 (FN1) protects against APOEε4 in Alzheimer's disease. Acta Neuropathologica. 147:70
|
Blokhuis, A.M., Koppers, M., Groen, E.J., van den Heuvel, D.M., Dini Modigliani, S., Anink, J.J., Fumoto, K., van Diggelen, F., Snelting, A., Sodaar, P., Verheijen, B.M., Demmers, J.A., Veldink, J.H., Aronica, E., Bozzoni, I., den Hertog, J., van den Berg, L.H., Pasterkamp, R.J. (2016) Comparative interactomics analysis of different ALS-associated proteins identifies converging molecular pathways. Acta Neuropathologica. 132(2):175-96
|
Boon, K.L., Xiao, S., McWhorter, M.L., Donn, T., Wolf-Saxon, E., Bohnsack, M.T., Moens, C.B., and Beattie, C.E. (2009) Zebrafish survival motor neuron Mutants Exhibit Presynaptic Neuromuscular Junction Defects. Human molecular genetics. 18(19):3615-3625
|
|
|
|
|
Bragato, C., Pistocchi, A., Bellipanni, G., Confalonieri, S., Balciunie, J., Monastra, F.M., Carra, S., Vitale, G., Mantecca, P., Cotelli, F., Gaudenzi, G. (2023) Zebrafish dnm1a gene plays a role in the formation of axons and synapses in the nervous tissue. Journal of neuroscience research. 101(8):1345-1359
|
|
|
Braubach, O.R., Miyasaka, N., Koide, T., Yoshihara, Y., Croll, R.P., and Fine, A. (2013) Experience-Dependent versus Experience-Independent Postembryonic Development of Distinct Groups of Zebrafish Olfactory Glomeruli. The Journal of neuroscience : the official journal of the Society for Neuroscience. 33(16):6905-6916
|
|
|
|
|
Brown, T.L., Horton, E.C., Craig, E.W., Goo, C.E.A., Black, E.C., Hewitt, M.N., Yee, N.G., Fan, E.T., Raible, D.W., Rasmussen, J.P. (2023) Dermal appendage-dependent patterning of zebrafish atoh1a+ Merkel cells. eLIFE. 12:
|
|
|
Bryant, D., Seda, M., Peskett, E., Maurer, C., Pomeranz, G., Ghosh, M., Hawkins, T.A., Cleak, J., Datta, S., Hariri, H., Eckert, K.M., Jafree, D.J., Walsh, C., Demetriou, C., Ishida, M., Alemán-Charlet, C., Vestito, L., Seselgyte, R., McDonald, J.G., Bitner-Glindzicz, M., Hemberger, M., Rihel, J., Teboul, L., Henne, W.M., Jenkins, D., Moore, G.E., Stanier, P. (2020) Diverse species-specific phenotypic consequences of loss of function sorting nexin 14 mutations. Scientific Reports. 10:13763
|
Burns, D.T., Donkervoort, S., Müller, J.S., Knierim, E., Bharucha-Goebel, D., Faqeih, E.A., Bell, S.K., AlFaifi, A.Y., Monies, D., Millan, F., Retterer, K., Dyack, S., MacKay, S., Morales-Gonzalez, S., Giunta, M., Munro, B., Hudson, G., Scavina, M., Baker, L., Massini, T.C., Lek, M., Hu, Y., Ezzo, D., AlKuraya, F.S., Kang, P.B., Griffin, H., Foley, A.R., Schuelke, M., Horvath, R., Bönnemann, C.G. (2018) Variants in EXOSC9 Disrupt the RNA Exosome and Result in Cerebellar Atrophy with Spinal Motor Neuronopathy. American journal of human genetics. 102:858-873
|
Butterfield, R.J., Stevenson, T.J., Xing, L., Newcomb, T.M., Nelson, B., Zeng, W., Li, X., Lu, H.M., Lu, H., Farwell Gonzalez, K.D., Wei, J.P., Chao, E.C., Prior, T.W., Snyder, P.J., Bonkowsky, J.L., Swoboda, K.J. (2014) Congenital lethal motor neuron disease with a novel defect in ribosome biogenesis. Neurology. 82:1322-30
|
|
|
Cairns, G., Burté, F., Price, R., O'Connor, E., Toms, M., Mishra, R., Moosajee, M., Pyle, A., Sayer, J.A., Yu-Wai-Man, P. (2021) A mutant wfs1 zebrafish model of Wolfram syndrome manifesting visual dysfunction and developmental delay. Scientific Reports. 11:20491
|
|
|
|
|
Casey, M.A., Hill, J.T., Hoshijima, K., Bryan, C.D., Gribble, S.L., Brown, J.T., Chien, C.B., Yost, H.J., Kwan, K.M. (2021) Shutdown corner, a large deletion mutant isolated from a haploid mutagenesis screen in zebrafish. G3 (Bethesda). 12(3):
|
Castro, A.F., Figueiredo, A.S., Loureiro, J.R., Azevedo, M.M., Sampaio, P., Valentim, A.M., Bessa, J., Silveira, I. (2026) Embryonic spinocerebellar ataxia type 37 AUUUC repeat RNA causes neurodevelopmental defects in zebrafish. Disease models & mechanisms. 19:dmm052636
|
Chaouch, A., Porcelli, V., Cox, D., Edvardson, S., Scarcia, P., De Grassi, A., Pierri, C.L., Cossins, J., Laval, S.H., Griffin, H., Müller, J.S., Evangelista, T., Töpf, A., Abicht, A., Huebner, A., von der Hagen, M., Bushby, K., Straub, V., Horvath, R., Elpeleg, O., Palace, J., Senderek, J., Beeson, D., Palmieri, L., Lochmüller, H. (2014) Mutations in the Mitochondrial Citrate Carrier SLC25A1 are Associated with Impaired Neuromuscular Transmission. Journal of neuromuscular diseases. 1:75-90
|
|
|
Chen, Z., Long, H., Guo, J., Wang, Y., He, K., Tao, C., Li, X., Jiang, K., Guo, S., Pi, Y. (2022) Autism-Risk Gene necab2 Regulates Psychomotor and Social Behavior as a Neuronal Modulator of mGluR1 Signaling. Frontiers in molecular neuroscience. 15:901682
|
Choueiri, C.M., Lau, J., O'Connor, E., DiBattista, A., Wong, B.Y., Spendiff, S., Horvath, R., Pena, I., MacKenzie, A., Lochmüller, H. (2024) Development of a riboflavin-responsive model of riboflavin transporter deficiency in zebrafish. Human molecular genetics. 34:265-276
|
Chung, A.Y., Kim, M.J., Kim, D., Bang, S., Hwang, S.W., Lim, C.S., Lee, S., Park, H.C., and Huh, T.L. (2010) Neuron-specific expression of atp6v0c2 in zebrafish CNS. Developmental Dynamics : an official publication of the American Association of Anatomists. 239(9):2501-2508
|
Cinzia, B., Flavia, B., Gary, I., Renato, M., Lorenzo, M. (2021) Therapeutic efficacy of 3,4-Diaminopyridine phosphate on neuromuscular junction in Pompe disease. Biomedicine & pharmacotherapy = Biomedecine & pharmacotherapie. 137:111357
|
Claes, F., Rudyak, S., Laird, A.S., Louros, N., Beerten, J., Debulpaep, M., Michiels, E., van der Kant, R., Van Durme, J., De Baets, G., Houben, B., Ramakers, M., Yuan, K., Gwee, S.S.L., Hernandez, S., Broersen, K., Oliveberg, M., Moahamed, B., Kirstein, J., Robberecht, W., Rousseau, F., Schymkowitz, J. (2020) Exposure of a cryptic Hsp70 binding site determines the cytotoxicity of the ALS-associated SOD1-mutant A4V. Protein engineering, design & selection : PEDS. 32(10):443-457
|
|
|
|
|
Craig, E.W., Black, E.C., Fernandes, S.Z., Ferdous, A.S., Goo, C.E.A., Sargent, S.M., Quitevis, E.J.A., Swearer, A.A., Yee, N.G., Shin, J., Solnica-Krezel, L., Rasmussen, J.P. (2025) Dendritic atoh1a+ cells serve as Merkel cell precursors during skin development and regeneration. Development (Cambridge, England). 152:dev204810
|
Crouzier, L., Richard, E.M., Diez, C., Alzaeem, H., Denus, M., Cubedo, N., Delaunay, T., Glendenning, E., Baxendale, S., Liévens, J.C., Whitfield, T.T., Maurice, T., Delprat, B. (2022) Morphological, behavioral and cellular analyses revealed different phenotypes in Wolfram syndrome wfs1a and wfs1b zebrafish mutant lines. Human molecular genetics. 31(16):2711-2727
|
D'Gama, P.P., Jeong, I., Nygård, A.M., Trinh, A.T., Yaksi, E., Jurisch-Yaksi, N. (2024) Ciliogenesis defects after neurulation impact brain development and neuronal activity in larval zebrafish. iScience. 27:110078
|
Da Costa, M.M., Allen, C.E., Higginbottom, A., Ramesh, T., Shaw, P.J., and McDermott, C.J. (2014) A new zebrafish model of SOD1 ALS produced by TILLING replicates key features of the disease and represents a tool for in vivo therapeutic screening. Disease models & mechanisms. 7(1):73-81
|
Daniele, L.L., Emran, F., Lobo, G.P., Gaivin, R.J., Perkins, B.D. (2016) Mutation of wrb, a Component of the Guided Entry of Tail-Anchored Protein Pathway, Disrupts Photoreceptor Synapse Structure and Function. Investigative ophthalmology & visual science. 57:2942-54
|
de Borsetti, N.H., Dean, B.J., Bain, E.J., Clanton, J.A., Taylor, R.W., and Gamse, J.T. (2011) Light and melatonin schedule neuronal differentiation in the habenular nuclei. Developmental Biology. 358(1):251-61
|
De Muynck, L., Herdewyn, S., Beel, S., Scheveneels, W., Van Den Bosch, L., Robberecht, W., and Van Damme, P. (2013) The neurotrophic properties of progranulin depend on the granulin E domain but do not require sortilin binding. Neurobiology of aging. 34(11):2541-7
|
De Pace, R., Maroofian, R., Paimboeuf, A., Zamani, M., Zaki, M.S., Sadeghian, S., Azizimalamiri, R., Galehdari, H., Zeighami, J., Williamson, C.D., Fleming, E., Zhou, D., Gannon, J.L., Thiffault, I., Roze, E., Suri, M., Zifarelli, G., Bauer, P., Houlden, H., Severino, M., Patten, S.A., Farrow, E., Bonifacino, J.S. (2023) Biallelic BORCS8 variants cause an infantile-onset neurodegenerative disorder with altered lysosome dynamics. Brain : a journal of neurology. 147(5):1751-1767
|
Demaria, S., Berke, A.P., Van Name, E., Heravian, A., Ferreira, T., and Ngai, J. (2013) Role of a ubiquitously expressed receptor in the vertebrate olfactory system. The Journal of neuroscience : the official journal of the Society for Neuroscience. 33(38):15235-15247
|
|
|
Dhakal, S., Rotem-Bamberger, S., Sejd, J.R., Sebbagh, M., Ronin, N., Frey, R.A., Beitsch, M., Batty, M., Taler, K., Blackerby, J.F., Inbal, A., Stenkamp, D.L. (2021) Selective Requirements for Vascular Endothelial Cells and Circulating Factors in the Regulation of Retinal Neurogenesis. Frontiers in cell and developmental biology. 9:628737
|
Dhakal, S., Stevens, C.B., Sebbagh, M., Weiss, O., Frey, R.A., Adamson, S., Shelden, E.A., Inbal, A., Stenkamp, D.L. (2015) Abnormal retinal development in cloche mutant zebrafish. Developmental Dynamics : an official publication of the American Association of Anatomists. 244(11):1439-55
|
|
|
|
|
|
|
|
|
|
|
Efthymiou, S., Leo, C.P., Deng, C., Lin, S.J., Maroofian, R., Lin, R., Karagoz, I., Zhang, K., Kaiyrzhanov, R., Scardamaglia, A., Owrang, D., Turchetti, V., Jahnke, F., Huang, K., Petree, C., Derrick, A.V., Rees, M.I., Alvi, J.R., Sultan, T., Li, C., Jacquemont, M.L., Tran-Mau-Them, F., Valenzuela-Palafoll, M., Sidlow, R., Yoon, G., Morrow, M.M., Carere, D.A., O'Connor, M., Fleischer, J., Gerkes, E.H., Phornphutkul, C., Isidor, B., Rivier-Ringenbach, C., Philippe, C., Kurul, S.H., Soydemir, D., Kara, B., Sunnetci-Akkoyunlu, D., Bothe, V., Platzer, K., Wieczorek, D., Koch-Hogrebe, M., Rahner, N., Thuresson, A.C., Matsson, H., Frykholm, C., Bozdoğan, S.T., Bisgin, A., Chatron, N., Lesca, G., Cabet, S., Tümer, Z., Hjortshøj, T.D., Rønde, G., Marquardt, T., Reunert, J., Afzal, E., Zamani, M., Azizimalamiri, R., Galehdari, H., Nourbakhsh, P., Chamanrou, N., Chung, S.K., Suri, M., Benke, P.J., Zaki, M.S., Gleeson, J.G., Calame, D.G., Pehlivan, D., Yilmaz, H.I., Gezdirici, A., Rad, A., Abumansour, I.S., Oprea, G., Bereketoğlu, M.B., Banneau, G., Julia, S., Zeighami, J., Ashoori, S., Shariati, G., Sedaghat, A., Sabri, A., Hamid, M., Parvas, S., Tajudin, T.A., Abdullah, U., Baig, S.M., Chung, W.K., Glazunova, O.O., Sabine, S., Cheema, H.A., Zifarelli, G., Bauer, P., Sidpra, J., Mankad, K., Vona, B., Fry, A.E., Varshney, G.K., Houlden, H., Fu, D. (2025) Biallelic pathogenic variants in TRMT1 disrupt tRNA modification and induce a neurodevelopmental disorder. American journal of human genetics. 112:1117-1138
|
|
|
|
|
|
|
|
|
|
|
|
|
Flannery, K.P., Mowla, S., Battula, N., Clark, L.R., Oliveira, C.D., Simhon, L.M., Liu, D., Venkatesan, C., Karas, B.F., Terez, K.R., Burbano, D., Manzini, M.C. (2026) Impact of maternal compensation on developmental phenotypes in a zebrafish model of severe congenital muscular dystrophy. PLoS Genetics. 22:e1011987
|
Fleisch, V.C., Leighton, P.L., Wang, H., Pillay, L.M., Ritzel, R.G., Bhinder, G., Roy, B., Tierney, K.B., Ali, D.W., Waskiewicz, A.J., and Allison, W.T. (2013) Targeted Mutation of the Gene Encoding Prion Protein in Zebrafish reveals a Conserved Role in Neuron Excitability. Neurobiology of disease. 55:11-25
|
Folgueira, M., Riva-Mendoza, S., Ferreño-Galmán, N., Castro, A., Bianco, I.H., Anadón, R., Yáñez, J. (2020) Anatomy and Connectivity of the Torus Longitudinalis of the Adult Zebrafish. Frontiers in neural circuits. 14:8
|
Fontenas, L., De Santis, F., Di Donato, V., Degerny, C., Chambraud, B., Del Bene, F., Tawk, M. (2016) Neuronal Ndrg4 Is Essential for Nodes of Ranvier Organization in Zebrafish. PLoS Genetics. 12:e1006459
|
|
|
Freifeld, L., Odstrcil, I., Förster, D., Ramirez, A., Gagnon, J.A., Randlett, O., Costa, E.K., Asano, S., Celiker, O.T., Gao, R., Martin-Alarcon, D.A., Reginato, P., Dick, C., Chen, L., Schoppik, D., Engert, F., Baier, H., Boyden, E.S. (2017) Expansion microscopy of zebrafish for neuroscience and developmental biology studies. Proceedings of the National Academy of Sciences of the United States of America. 114(50):E10799-E10808
|
Gangras, P., Gallagher, T.L., Parthun, M.A., Yi, Z., Patton, R.D., Tietz, K.T., Deans, N.C., Bundschuh, R., Amacher, S.L., Singh, G. (2020) Zebrafish rbm8a and magoh mutants reveal EJC developmental functions and new 3'UTR intron-containing NMD targets. PLoS Genetics. 16:e1008830
|
Garcia-Reyero, N., Escalon, B.L., Prats, E., Stanley, J.K., Thienpont, B., Melby, N.L., Barón, E., Eljarrat, E., Barceló, D., Mestres, J., Babin, P.J., Perkins, E.J., and Raldúa, D. (2014) Effects of BDE-209 contaminated sediments on zebrafish development and potential implications to human health. Environment International. 63:216-223
|
|
|
|
|
Ghosh, S., Singh, J., Damseh, N.S., Severino, M., De Pace, R., Golding, A.E., Jarnik, M., Thakran, P., Faivre, L., Heitz, J., Denommé-Pichon, A.S., Vitobello, A., AlAbdi, L., Abdulwahab, F., Sumayli, S., Alqahtani, M., Cheema, H.A., Javed, I., Kim, J., Lochmüller, H., Mor-Shaked, H., Neil, J.E., Mochida, G.H., Zifarelli, G., Bauer, P., Barkhordari, E., Ghayoor Karimiani, E., Houlden, H., Abu-Libdeh, B., Edvardson, S., Elpeleg, O., Maroofian, R., Patten, S.A., Bonifacino, J.S. (2025) EIPR1 variants cause a neurodevelopmental disorder with endolysosomal and dense core vesicle defects. Brain : a journal of neurology. 149:1568-1585
|
|
|
Gibbs, E.M., Clarke, N.F., Rose, K., Oates, E.C., Webster, R., Feldman, E.L., and Dowling, J.J. (2013) Neuromuscular junction abnormalities in DNM2-related centronuclear myopathy. Journal of molecular medicine (Berlin, Germany). 91(6):727-37
|
Giunta, M., Edvardson, S., Xu, Y., Schuelke, M., Gomez-Duran, A., Boczonadi, V., Elpeleg, O., Müller, J.S., Horvath, R. (2016) Altered RNA metabolism due to a homozygous RBM7 mutation in a patient with spinal motor neuropathy. Human molecular genetics. 25(14):2985-2996
|
Goldshtein, H., Muhire, A., Petel Légaré, V., Pushett, A., Rotkopf, R., Shefner, J.M., Peterson, R.T., Armstrong, G.A.B., Russek-Blum, N. (2020) Efficacy of Ciprofloxacin/Celecoxib combination in zebrafish models of amyotrophic lateral sclerosis. Annals of clinical and translational neurology. 7(10):1883-1897
|
Goode, C., Voeun, M., Ncube, D., Eisen, J., Washbourne, P., Tallafuss, A. (2020) Late onset of Synaptotagmin 2a expression at synapses relevant to social behavior. The Journal of comparative neurology. 529(9):2176-2188
|
Hao, L.T., Duy, P.Q., Jontes, J.D., Wolman, M., Granato, M., and Beattie, C.E. (2013) Temporal requirement for SMN in motoneuron development. Human molecular genetics. 22(13):2612-25
|
|
|
Harris, J.M., Wang, A.Y., Boulanger-Weill, J., Santoriello, C., Foianini, S., Lichtman, J.W., Zon, L.I., Arlotta, P. (2020) Long-Range Optogenetic Control of Axon Guidance Overcomes Developmental Boundaries and Defects. Developmental Cell. 53:577-588.e7
|
Helferich, A.M., Brockmann, S.J., Reinders, J., Deshpande, D., Holzmann, K., Brenner, D., Andersen, P.M., Petri, S., Thal, D.R., Michaelis, J., Otto, M., Just, S., Ludolph, A.C., Danzer, K.M., Freischmidt, A., Weishaupt, J.H. (2018) Dysregulation of a novel miR-1825/TBCB/TUBA4A pathway in sporadic and familial ALS. Cellular and molecular life sciences : CMLS. 75(23):4301-4319
|
|
|
Hoffman, E.J., Turner, K.J., Fernandez, J.M., Cifuentes, D., Ghosh, M., Ijaz, S., Jain, R.A., Kubo, F., Bill, B.R., Baier, H., Granato, M., Barresi, M.J., Wilson, S.W., Rihel, J., State, M.W., Giraldez, A.J. (2016) Estrogens Suppress a Behavioral Phenotype in Zebrafish Mutants of the Autism Risk Gene, CNTNAP2. Neuron. 89(4):725-33
|
Hogan, A.L., Don, E.K., Rayner, S.L., Lee, A., Laird, A.S., Watchon, M., Winnick, C., Tarr, I.S., Morsch, M., Fifita, J.A., Gwee, S., Formella, I., Hortle, E., Yuan, K., Molloy, M.P., Williams, K.L., Nicholson, G.A., Chung, R.S., Blair, I.P., Cole, N.J. (2017) Expression of ALS/FTD-linked mutant CCNF in zebrafish leads to increased cell death in the spinal cord and an aberrant motor phenotype. Human molecular genetics. 26(14):2616-2626
|
Hogan, A.L., Kane, M., Chiu, P., Richter, G., Maurel, C., Wu, S., Scherer, N.M., Don, E.K., Lee, A., Blair, I.P., Chung, R.S., Morsch, M. (2025) Human TDP-43 overexpression in zebrafish motor neurons triggers MND-like phenotypes through gain-of-function mechanism. Acta neuropathologica communications. 14:63
|
Hong, Y.B., Kang, J., Kim, J.H., Lee, J., Kwak, G., Hyun, Y.S., Nam, S.H., Hong, H.D., Choi, Y.R., Jung, S.C., Koo, H., Lee, J.E., Choi, B.O., Chung, K.W. (2016) DGAT2 Mutation in a Family with Autosomal Dominant Early-Onset Axonal Charcot-Marie-Tooth Disease. Human Mutation. 37(5):473-80
|
Horstick, E.J., Linsley, J.W., Dowling, J.J., Hauser, M.A., McDonald, K.K., Ashley-Koch, A., Saint-Amant, L., Satish, A., Cui, W.W., Zhou, W., Sprague, S.M., Stamm, D.S., Powell, C.M., Speer, M.C., Franzini-Armstrong, C., Hirata, H., and Kuwada, J.Y. (2013) Stac3 is a component of the excitation-contraction coupling machinery and mutated in Native American myopathy. Nature communications. 4:1952
|
Hotz, A.L., Jamali, A., Rieser, N.N., Niklaus, S., Aydin, E., Myren-Svelstad, S., Lalla, L., Jurisch-Yaksi, N., Yaksi, E., Neuhauss, S.C.F. (2021) Loss of glutamate transporter eaat2a leads to aberrant neuronal excitability, recurrent epileptic seizures, and basal hypoactivity. Glia. 70(1):196-214
|
|
|
Hunter, P.R., Nikolaou, N., Odermatt, B., Williams, P.R., Drescher, U., and Meyer, M.P. (2011) Localization of cadm2a and cadm3 proteins during development of the zebrafish nervous system. The Journal of comparative neurology. 519(11):2252-70
|
Häberlein, F., Mingardo, E., Merten, N., Schulze Köhling, N.K., Reinoß, P., Simon, K., Japp, A., Nagarajan, B., Schrage, R., Pegurier, C., Gillard, M., Monk, K.R., Odermatt, B., Kostenis, E., Gomeza, J. (2022) Humanized zebrafish as a tractable tool for in vivo evaluation of pro-myelinating drugs. Cell chemical biology. 29(10):1541-1555.e7
|
Hüsken, U., Stickney, H.L., Gestri, G., Bianco, I.H., Faro, A., Young, R.M., Roussigne, M., Hawkins, T.A., Beretta, C.A., Brinkmann, I., Paolini, A., Jacinto, R., Albadri, S., Dreosti, E., Tsalavouta, M., Schwarz, Q., Cavodeassi, F., Barth, A.K., Wen, L., Zhang, B., Blader, P., Yaksi, E., Poggi, L., Zigman, M., Lin, S., Wilson, S.W., Carl, M. (2014) Tcf7l2 Is Required for Left-Right Asymmetric Differentiation of Habenular Neurons. Current biology : CB. 24(19):2217-27
|
|
|
Jardine, S., Anderson, S., Babcock, S., Leung, G., Pan, J., Dhingani, N., Warner, N., Guo, C., Siddiqui, I., Kotlarz, D., Dowling, J.J., Melnyk, R., Snapper, S.B., Klein, C., Thiagarajah, J.R., Muise, A.M. (2019) Drug Screen Identifies Leflunomide for Treatment of Inflammatory Bowel Diseases Caused by TTC7A Deficiency. Gastroenterology. 158(4):1000-1015
|
Jaroszynska, N., Salzinger, A., Tsarouchas, T.M., Becker, C.G., Becker, T., Lyons, D.A., MacDonald, R.B., Keatinge, M. (2024) C9ORF72 deficiency results in neurodegeneration in the zebrafish retina. The Journal of neuroscience : the official journal of the Society for Neuroscience. 44(25):
|
|
|
Jeong, H.S., Kim, H.J., Kim, D.H., Chung, K.W., Choi, B.O., Lee, J.E. (2022) Therapeutic Potential of CKD-504, a Novel Selective Histone Deacetylase 6 Inhibitor, in a Zebrafish Model of Neuromuscular Junction Disorders. Molecules and cells. 45(4):231-242
|
Jia, S., Muto, A., Orisme, W., Henson, H.E., Parupalli, C., Ju, B., Baier, H., and Taylor, M.R. (2014) Zebrafish Cacna1fa is required for cone photoreceptor function and synaptic ribbon formation. Human molecular genetics. 23(11):2981-94
|
Julien, D.P., Chan, A.W., Barrios, J., Mathiaparanam, J., Douglass, A., Wolman, M.A., Sagasti, A. (2018) Zebrafish expression reporters and mutants reveal that the IgSF cell adhesion molecule Dscamb is required for feeding and survival. Journal of neurogenetics. 32(4):336-352
|
Kabashi, E., Bercier, V., Lissouba, A., Liao, M., Brustein, E., Rouleau, G.A, and Drapeau, P. (2011) FUS and TARDBP but Not SOD1 Interact in Genetic Models of Amyotrophic Lateral Sclerosis. PLoS Genetics. 7(8):e1002214
|
Kabashi, E., El Oussini, H., Bercier, V., Gros-Louis, F., Valdmanis, P.N., McDearmid, J., Mejier, I.A., Dion, P.A., Dupre, N., Hollinger, D., Sinniger, J., Dirrig-Grosch, S., Camu, W., Meininger, V., Loeffler, J.P., René, F., Drapeau, P., Rouleau, G.A., and Dupuis, L. (2013) Investigating the contribution of VAPB/ALS8 loss of function in amyotrophic lateral sclerosis. Human molecular genetics. 22(12):2350-60
|
Kabashi, E., Lin, L., Tradewell, M.L., Dion, P.A., Bercier, V., Bourgouin, P., Rochefort, D., Bel Hadj, S., Durham, H.D., Vande Velde, C., Rouleau, G.A., and Drapeau, P. (2010) Gain and loss of function of ALS-related mutations of TARDBP (TDP-43) cause motor deficits in vivo. Human molecular genetics. 19(4):671-683
|
Kaniganti, T., Deogade, A., Maduskar, A., Mukherjee, A., Guru, A., Subhedar, N., Ghose, A. (2021) Sensitivity of Olfactory Sensory Neurons to food cues is tuned to nutritional states by Neuropeptide Y signalling. Journal of neurochemistry. 159(6):1028-1044
|
Karas, B.F., Terez, K.R., Mowla, S., Battula, N., Flannery, K.P., Gural, B.M., Aboussleman, G., Mubin, N., Manzini, M.C. (2024) Removal of pomt1 in zebrafish leads to loss of α-dystroglycan glycosylation and dystroglycanopathy phenotypes. Human molecular genetics. 33(8):709-723
|
Kikel-Coury, N.L., Brandt, J.P., Correia, I.A., O'Dea, M.R., DeSantis, D.F., Sterling, F., Vaughan, K., Ozcebe, G., Zorlutuna, P., Smith, C.J. (2021) Identification of astroglia-like cardiac nexus glia that are critical regulators of cardiac development and function. PLoS Biology. 19:e3001444
|
Kilroy, E.A., Ignacz, A.C., Brann, K.L., Schaffer, C.E., Varney, D., Alrowaished, S.S., Silknitter, K.J., Miner, J.N., Almaghasilah, A., Spellen, T.L., Lewis, A.D., Tilbury, K., King, B.L., Kelley, J.B., Henry, C.A. (2022) Beneficial impacts of neuromuscular electrical stimulation on muscle structure and function in the zebrafish model of Duchenne muscular dystrophy. eLIFE. 11:
|
Kim, M.J., Liu, I.H., Song, Y., Lee, J.A., Halfter, W., Balice-Gordon, R.J., Linney, E., and Cole, G.J. (2007) Agrin is Required for Posterior Development and Motor Axon Outgrowth and Branching in Embryonic Zebrafish. Glycobiology. 17(2):231-247
|
Kim, S., Chung, A.Y., Na, J.E., Lee, S.J., Jeong, S.H., Kim, E., Sun, W., Rhyu, I.J., Park, H.C. (2019) Myelin degeneration induced by mutant superoxide dismutase 1 accumulation promotes amyotrophic lateral sclerosis. Glia. 67(10):1910-1921
|
|
|
Koh, A., Tao, S., Jing, G.Y., Chaganty, V., See, K., Purushothaman, K., Orbán, L., Mathuru, A.S., Wohland, T., Winkler, C. (2020) A Neurexin2aa deficiency results in axon pathfinding defects and increased anxiety in zebrafish. Human molecular genetics. 29(23):3765-3780
|
Koide, T., Miyasaka, N., Morimoto, K., Asakawa, K., Urasaki, A., Kawakami, K., and Yoshihara, Y. (2009) Olfactory neural circuitry for attraction to amino acids revealed by transposon-mediated gene trap approach in zebrafish. Proceedings of the National Academy of Sciences of the United States of America. 106(24):9884-9889
|
Korac, J., Schaeffer, V., Kovacevic, I., Clement, A.M., Jungblut, B., Behl, C., Terzic, J., and Dikic, I. (2013) Ubiquitin-independent function of optineurin in autophagic clearance of protein aggregates. Journal of Cell Science. 126(2):580-592
|
Kroehne, V., Freudenreich, D., Hans, S., Kaslin, J., and Brand, M. (2011) Regeneration of the adult zebrafish brain from neurogenic radial glia-type progenitors. Development (Cambridge, England). 138(22):4831-4841
|
|
|
|
|
König, D., Dagenais, P., Senk, A., Djonov, V., Aegerter, C.M., Jaźwińska, A. (2019) Distribution and Restoration of Serotonin-Immunoreactive Paraneuronal Cells During Caudal Fin Regeneration in Zebrafish. Frontiers in molecular neuroscience. 12:227
|
|
|
Laird, A.S., Van Hoecke, A., De Muynck, L., Timmers, M., Van Den Bosch, L., Van Damme, P., and Robberecht, W. (2010) Progranulin is Neurotrophic In Vivo and Protects against a Mutant TDP-43 Induced Axonopathy. PLoS One. 5(10):pii: e13368
|
Lambton, J., Asano, S., Huang, Y., Suomi, F., Eguchi, T., Petree, C., Huang, K., Prigent, M., Imam, A., McCorvie, T.J., Warren, D., Hobson, E., McCullagh, H., Misceo, D., Bjerre, A., Smeland, M.F., Klingenberg, C., Frengen, E., Naik, S., Ryan, G., Sudarsanam, A., Foster, K., Vasudevan, P., Samanta, R., Rahman, F., Maqbool, S., Udani, V., Efthymiou, S., Houlden, H., McFarland, R., Collier, J.J., Maroofian, R., Yue, W.W., Varshney, G.K., Klionsky, D.J., Legouis, R., McWilliams, T.G., Mizushima, N., Oláhová, M., Alston, C.L., Taylor, R.W. (2026) Bi-allelic ATG12 variants impair autophagy and cause a neurodevelopmental disorder. American journal of human genetics. :
|
Lancman, J.J., Zvenigorodsky, N., Gates, K.P., Zhang, D., Solomon, K., Humphrey, R.K., Kuo, T., Setiawan, L., Verkade, H., Chi, Y.I., Jhala, U.S., Wright, C.V., Stainier, D.Y., and Dong, P.D. (2013) Specification of hepatopancreas progenitors in zebrafish by hnf1ba and wnt2bb. Development (Cambridge, England). 140(13):2669-2679
|
|
|
Lemmens, K., Bollaerts, I., Bhumika, S., De Groef, L., Van Houcke, J., Darras, V.M., Van Hove, I., Moons, L. (2016) Matrix metalloproteinases as promising regulators of axonal regrowth in the injured adult zebrafish retinotectal system. The Journal of comparative neurology. 524(7):1472-93
|
|
|
|
|
Lin, C.Y., Zhang, P.H., Chen, Y.J., Wu, C.L., Tsai, H.J. (2018) Conditional Overexpression of rtn4al in Muscle of Adult Zebrafish Displays Defects Similar to Human Amyotrophic Lateral Sclerosis. Marine biotechnology (New York, N.Y.). 21(1):52-64
|
Lin, S.Y., Vollrath, M.A., Mangosing, S., Shen, J., Cardenas, E., Corey, D.P. (2016) The zebrafish pinball wizard gene encodes WRB, a tail-anchored-protein receptor essential for inner-ear hair cells and retinal photoreceptors. The Journal of physiology. 594(4):895-914
|
|
|
Low, S.E., Woods, I.G., Lachance, M., Ryan, J., Schier, A.F., and Saint-Amant, L. (2012) Touch responsiveness in zebrafish requires voltage-gated calcium channel 2.1b. Journal of neurophysiology. 108(1):148-159
|
Lu, X., Song, Y., Wang, J., Cai, Y., Peng, S., Lin, J., Lai, B., Sun, J., Liu, T., Chen, G., Xing, L. (2024) Developmental dopaminergic signaling modulates neural circuit formation and contributes to autism spectrum disorder (ASD)-related phenotypes. The American journal of pathology. 194(6):1062-1077
|
|
|
|
|
|
|
|
|
Martin, N.R., Patel, R., Kossack, M.E., Tian, L., Camarillo, M.A., Cintrón-Rivera, L.G., Gawdzik, J.C., Yue, M.S., Nwagugo, F.O., Elemans, L.M.H., Plavicki, J.S. (2022) Proper modulation of AHR signaling is necessary for establishing neural connectivity and oligodendrocyte precursor cell development in the embryonic zebrafish brain. Frontiers in molecular neuroscience. 15:1032302
|
Masek, M., Etard, C., Hofmann, C., Hülsmeier, A.J., Zang, J., Takamiya, M., Gesemann, M., Neuhauss, S.C.F., Hornemann, T., Strähle, U., Bachmann-Gagescu, R. (2022) Loss of the Bardet-Biedl protein Bbs1 alters photoreceptor outer segment protein and lipid composition. Nature communications. 13:1282
|
McCann, T., Sundaramurthi, H., Walsh, C., Virdi, S., Alvarez, Y., Sapetto-Rebow, B., Collery, R.F., Carter, S.P., Moran, A., Mulholland, R., O'Connor, J.J., Taylor, M.R., Rauch, N., Starostik, M.R., English, M.A., Swaroop, A., Geisler, R., Reynolds, A.L., Kennedy, B.N. (2024) Emc1 is essential for vision and zebrafish photoreceptor outer segment morphogenesis. FASEB journal : official publication of the Federation of American Societies for Experimental Biology. 38:e70086
|
McGinn, T.E., Mitchell, D.M., Meighan, P.C., Partington, N., Leoni, D.C., Jenkins, C.E., Varnum, M.D., Stenkamp, D.L. (2017) Restoration of Dendritic Complexity, Functional Connectivity, and Diversity of Regenerated Retinal Bipolar Neurons in Adult Zebrafish. The Journal of neuroscience : the official journal of the Society for Neuroscience. 38(1):120-136
|
McGown, A., McDearmid, J.R., Panagiotaki, N., Tong, H., Al Mashhadi, S., Redhead, N., Lyon, A.N., Beattie, C.E., Shaw, P.J., and Ramesh, T.M. (2013) Early interneuron dysfunction in ALS: Insights from a mutant sod1 zebrafish model. Annals of neurology. 73(2):246-258
|
McMacken, G., Cox, D., Roos, A., Müller, J., Whittaker, R., Lochmüller, H. (2018) The Beta-Adrenergic Agonist Salbutamol Modulates Neuromuscular Junction Formation in Zebrafish Models of Human Myasthenic Syndromes. Human molecular genetics. 27(9):1556-1564
|
|
|
Medrano, M.P., Pisera Fuster, A., Sanchis, P., Paez, N., Bernabeu, R.O., Faillace, M.P. (2018) Characterization of proliferative, glial and angiogenic responses after a CoCl2 -induced injury of photoreceptor cells in the adult zebrafish retina.. The European journal of neuroscience. 48(9):3019-3042
|
|
|
|
|
|
|
|
|
Miller, A.H., Howe, H.B., Krause, B.M., Friedle, S.A., Banks, M.I., Perkins, B.D., Wolman, M.A. (2018) Pregnancy associated plasma protein-aa (Pappaa) regulates photoreceptor synaptic development to mediate visually guided behavior. The Journal of neuroscience : the official journal of the Society for Neuroscience. 38(22):5220-5236
|
Miyasaka, N., Arganda-Carreras, I., Wakisaka, N., Masuda, M., Sümbül, U., Seung, H.S., Yoshihara, Y. (2014) Olfactory projectome in the zebrafish forebrain revealed by genetic single-neuron labelling. Nature communications. 5:3639
|
Miyasaka, N., Morimoto, K., Tsubokawa, T., Higashijima, S., Okamoto, H., and Yoshihara, Y. (2009) From the olfactory bulb to higher brain centers: genetic visualization of secondary olfactory pathways in zebrafish. The Journal of neuroscience : the official journal of the Society for Neuroscience. 29(15):4756-4767
|
Montgomery, J.E., Wahlstrom-Helgren, S., Wiggin, T.D., Corwin, B.M., Lillesaar, C., Masino, M.A. (2018) Intraspinal serotonergic signaling suppresses locomotor activity in larval zebrafish. Developmental Neurobiology.
|
Mott, M., Luna, V.M., Park, J.Y., Downes, G.B., Epley, K., Ono, F. (2017) Expressing acetylcholine receptors after innervation suppresses spontaneous vesicle release and causes muscle fatigue. Scientific Reports. 7:1674
|
Müller, J.S., Jepson, C.D., Laval, S.H., Bushby, K., Straub, V., and Lochmüller, H. (2010) Dok-7 promotes slow muscle integrity as well as neuromuscular junction formation in a zebrafish model of congenital myasthenic syndromes. Human molecular genetics. 19(9):1726-1740
|
Nakano, Y., Fujita, M., Ogino, K., Saint-Amant, L., Kinoshita, T., Oda, Y., and Hirata, H. (2010) Biogenesis of GPI-anchored proteins is essential for surface expression of sodium channels in zebrafish Rohon-Beard neurons to respond to mechanosensory stimulation. Development (Cambridge, England). 137(10):1689-1698
|
|
|
|
|
Niklaus, S., Glasauer, S.M.K., Kovermann, P., Farshori, K.F., Cadetti, L., Früh, S., Rieser, N.N., Gesemann, M., Zang, J., Fahlke, C., Neuhauss, S.C.F. (2024) Glutamate transporters are involved in direct inhibitory synaptic transmission in the vertebrate retina. Open Biology. 14:240140
|
Nikolaou, N., Gordon, P.M., Hamid, F., Taylor, R., Lloyd-Jones, J., Makeyev, E.V., Houart, C. (2022) Cytoplasmic pool of U1 spliceosome protein SNRNP70 shapes the axonal transcriptome and regulates motor connectivity. Current biology : CB. 32(23):5099-5115.e8
|
Noble, A.R., Masek, M., Hofmann, C., Cuoco, A., Rusterholz, T.D.S., Özkoc, H., Greter, N.R., Phelps, I.G., Vladimirov, N., Kollmorgen, S., Stoeckli, E., Bachmann-Gagescu, R. (2024) Shared and unique consequences of Joubert Syndrome gene dysfunction on the zebrafish central nervous system. Biology Open. 13(11):
|
Nozawa, K., Lin, Y., Kubodera, R., Shimizu, Y., Tanaka, H., Ohshima, T. (2017) Zebrafish Mecp2 is required for proper axonal elongation of motor neurons and synapse formation. Developmental Neurobiology. 77(9):1101-1113
|
O'Connor, E., Cairns, G., Spendiff, S., Burns, D., Hettwer, S., Mäder, A., Müller, J., Horvath, R., Slater, C., Roos, A., Lochmüller, H. (2019) Modulation of Agrin and RhoA Pathways Ameliorates Movement Defects and Synapse Morphology in MYO9A-Depleted Zebrafish. Cells. 8(8)
|
O'Connor, E., Phan, V., Cordts, I., Cairns, G., Hettwer, S., Cox, D., Lochmüller, H., Roos, A. (2018) MYO9A deficiency in motor neurons is associated with reduced neuromuscular agrin secretion. Human molecular genetics. 27(8):1434-1446
|
O'Connor, E., Töpf, A., Müller, J.S., Cox, D., Evangelista, T., Colomer, J., Abicht, A., Senderek, J., Hasselmann, O., Yaramis, A., Laval, S.H., Lochmüller, H. (2016) Identification of mutations in the MYO9A gene in patients with congenital myasthenic syndrome. Brain : a journal of neurology. 139(Pt 8):2143-53
|
Ogino, K., Ramsden, S.L., Keib, N., Schwarz, G., Harvey, R.J., and Hirata, H. (2011) Duplicated Gephyrin Genes Showing Distinct Tissue Distribution and Alternative Splicing Patterns Mediate Molybdenum Cofactor Biosynthesis, Glycine Receptor Clustering, and Escape Behavior in Zebrafish. The Journal of biological chemistry. 286(1):806-817
|
|
|
|
|
Oprişoreanu, A.M., Smith, H.L., Arya, S., Webster, R., Zhong, Z., Wehner, D., Cardozo, M.J., Becker, T., Talbot, K., Becker, C.G. (2019) Interaction of Axonal Chondrolectin with Collagen XIXa1 Is Necessary for Precise Neuromuscular Junction Formation. Cell Reports. 29:1082-1098.e10
|
|
|
|
|
Park, J.Y., Mott, M., Williams, T., Ikeda, H., Wen, H., Linhoff, M., Ono, F. (2014) A Single Mutation in the Acetylcholine Receptor δ-Subunit Causes Distinct Effects in Two Types of Neuromuscular Synapses. The Journal of neuroscience : the official journal of the Society for Neuroscience. 34:10211-10218
|
Paul, S., Pant, R., Sharma, P., Walia, K., Gupta, S., Aseem, A., Bajwa, K.K., George, R., Varma, Y., Bhatia, T., Ramachandran, R., Tuli, A., Sharma, M. (2025) The neuropathy-linked protein TECPR2 is a Rab5 effector that regulates cargo recycling from early endosomes. Nature communications. 16:10537
|
|
|
|
|
Phan, V., Cox, D., Cipriani, S., Spendiff, S., Buchkremer, S., O'Connor, E., Horvath, R., Goebel, H.H., Hathazi, D., Lochmüller, H., Straka, T., Rudolf, R., Weis, J., Roos, A. (2018) SIL1 deficiency causes degenerative changes of peripheral nerves and neuromuscular junctions in fish, mice and human. Neurobiology of disease. 124:218-229
|
Phillips, J.B., Blanco-Sanchez, B., Lentz, J.J., Tallafuss, A., Khanobdee, K., Sampath, S., Jacobs, Z.G., Han, P.F., Mishra, M., Williams, D.S., Keats, B.J., Washbourne, P., and Westerfield, M. (2011) Harmonin (Ush1c) is required in zebrafish Muller glial cells for photoreceptor synaptic development and function. Disease models & mechanisms. 4(6):786-800
|
Pistocchi, A., Gaudenzi, G., Foglia, E., Monteverde, S., Moreno-Fortuny, A., Pianca, A., Cossu, G., Cotelli, F., and Messina, G. (2013) Conserved and divergent functions of Nfix in skeletal muscle development during vertebrate evolution. Development (Cambridge, England). 140(7):1528-1536
|
Plavicki, J.S., Baker, T.R., Burns, F.R., Xiong, K.M., Gooding, A.J., Hofsteen, P., Peterson, R.E., Heideman, W. (2014) Construction and characterization of a sox9b transgenic reporter line.. The International journal of developmental biology. 58(9):693-9
|
Powell, G.T., Faro, A., Zhao, Y., Stickney, H., Novellasdemunt, L., Henriques, P., Gestri, G., Redhouse White, E., Ren, J., Lu, W., Young, R.M., Hawkins, T.A., Cavodeassi, F., Schwarz, Q., Dreosti, E., Raible, D.W., Li, V.S.W., Wright, G.J., Jones, E.Y., Wilson, S.W. (2024) Cachd1 interacts with Wnt receptors and regulates neuronal asymmetry in the zebrafish brain. Science (New York, N.Y.). 384:573579573-579
|
|
|
Quelle-Regaldie, A., Folgueira, M., Yáñez, J., Sobrido-Cameán, D., Alba-González, A., Barreiro-Iglesias, A., Sobrido, M.J., Sánchez, L. (2022) A nop56 Zebrafish Loss-of-Function Model Exhibits a Severe Neurodegenerative Phenotype. Biomedicines. 10(8)
|
Ramanoudjame, L., Rocancourt, C., Lainé, J., Klein, A., Joassard, L., Gartioux, C., Fleury, M., Lyphout, L., Kabashi, E., Ciura, S., Cousin, X., Allamand, V. (2015) Two novel COLVI long chains in zebrafish that are essential for muscle development. Human molecular genetics. 24(23):6624-39
|
Ramesh, T., Lyon, A.N., Pineda, R.H., Wang, C., Janssen, P.M., Canan, B.D., Burghes, A.H., and Beattie, C.E. (2010) A genetic model of amyotrophic lateral sclerosis in zebrafish displays phenotypic hallmarks of motoneuron disease. Disease models & mechanisms. 3(9-10):652-662
|
Randlett, O., MacDonald, R.B., Yoshimatsu, T., Almeida, A.D., Suzuki, S.C., Wong, R.O., Harris, W.A. (2013) Cellular requirements for building a retinal neuropil. Cell Reports. 3:282-90
|
Rauschenberger, V., Wardenburg, N.V., Schaefer, N., Ogino, K., Hirata, H., Lillesaar, C., Kluck, C.J., Meinck, H.M., Borrmann, M., Weishaupt, A., Doppler, K., Wickel, J., Geis, C., Sommer, C., Villmann, C. (2020) GlyR autoantibodies impair receptor function and induce motor dysfunction. Annals of neurology. 88(3):544-561
|
Rayamajhi, D., Ege, M., Ukhanov, K., Ringers, C., Zhang, Y., Jung, I., D'Gama, P.P., Li, S.S., Cosacak, M.I., Kizil, C., Park, H.C., Yaksi, E., Martens, J.R., Brody, S.L., Jurisch-Yaksi, N., Roy, S. (2024) The forkhead transcription factor Foxj1 controls vertebrate olfactory cilia biogenesis and sensory neuron differentiation. PLoS Biology. 22:e3002468
|
|
|
Reinoß, P., Ciglieri, E., Minére, M., Bremser, S., Klein, A., Löhr, H., Fuller, P.M., Büschges, A., Kloppenburg, P., Fenselau, H., Hammerschmidt, M. (2020) Hypothalamic Pomc Neurons Innervate the Spinal Cord and Modulate the Excitability of Premotor Circuits. Current biology : CB. 30(23):4579-4593.e7
|
Ricatti, M.J., Alfie, L.D., Lavoie, E.G., Sévigny, J., Schwarzbaum, P.J., and Faillace, M.P. (2009) Immunocytochemical localization of NTPDases1 and 2 in the neural retina of mouse and zebrafish. Synapse (New York, N.Y.). 63(4):291-307
|
|
|
|
|
Rothenaigner, I., Krecsmarik, M., Hayes, J.A., Bahn, B., Lepier, A., Fortin, G., Götz, M., Jagasia, R., and Bally-Cuif, L. (2011) Clonal analysis by distinct viral vectors identifies bona fide neural stem cells in the adult zebrafish telencephalon and characterizes their division properties and fate. Development (Cambridge, England). 138(8):1459-1469
|
Rouleau, M., Saxena, V., Rodrigue, A., Paquet, E.R., Gagnon, A., Hendzel, M.J., Masson, J.Y., Ekker, M., and Poirier, G.G. (2011) A Key Role for Poly(ADP-Ribose) Polymerase 3 in Ectodermal Specification and Neural Crest Development. PLoS One. 6(1):e15834
|
|
|
Sakowski, S.A., Lunn, J.S., Busta, A.S., Oh, S.S., Zamora-Berridi, G., Palmer, M., Rosenberg, A.A., Philip, S.G., Dowling, J.J., and Feldman, E.L. (2012) Neuromuscular effects of G93A-SOD1 expression in zebrafish. Molecular neurodegeneration. 7(1):44
|
Sakowski, S.A., Lunn, J.S., Busta, A.S., Palmer, M., Dowling, J.J., and Feldman, E.L. (2012) A novel approach to study motor neurons from zebrafish embryos and larvae in culture. Journal of Neuroscience Methods. 205(2):277-282
|
Salam, S., Tacconelli, S., Smith, B.N., Mitchell, J.C., Glennon, E., Nikolaou, N., Houart, C., Vance, C. (2021) Identification of a novel interaction of FUS and syntaphilin may explain synaptic and mitochondrial abnormalities caused by ALS mutations. Scientific Reports. 11:13613
|
Santhanam, A., Shihabeddin, E., Atkinson, J.A., Nguyen, D., Lin, Y.P., O'Brien, J. (2020) A Zebrafish Model of Retinitis Pigmentosa Shows Continuous Degeneration and Regeneration of Rod Photoreceptors. Cells. 9(10):
|
Schaffer, A.E., Eggens, V.R., Caglayan, A.O., Reuter, M.S., Scott, E., Coufal, N.G., Silhavy, J.L., Xue, Y., Kayserili, H., Yasuno, K., Rosti, R.O., Abdellateef, M., Caglar, C., Kasher, P.R., Cazemier, J.L., Weterman, M.A., Cantagrel, V., Cai, N., Zweier, C., Altunoglu, U., Satkin, N.B., Aktar, F., Tuysuz, B., Yalcinkaya, C., Caksen, H., Bilguvar, K., Fu, X.D., Trotta, C.R., Gabriel, S., Reis, A., Gunel, M., Baas, F., Gleeson, J.G. (2014) CLP1 Founder Mutation Links tRNA Splicing and Maturation to Cerebellar Development and Neurodegeneration. Cell. 157:651-63
|
Schlueter, P.J., Royer, T., Farah, M.H., Laser, B., Chan, S.J., Steiner, D.F., and Duan, C. (2006) Gene duplication and functional divergence of the zebrafish insulin-like growth factor 1 receptors. FASEB journal : official publication of the Federation of American Societies for Experimental Biology. 20(8):1230-1232
|
Senderek, J., Müller, J.S., Dusl, M., Strom, T.M., Guergueltcheva, V., Diepolder, I., Laval, S.H., Maxwell, S., Cossins, J., Krause, S., Muelas, N., Vilchez, J.J., Colomer, J., Mallebrera, C.J., Nascimento, A., Nafissi, S., Kariminejad, A., Nilipour, Y., Bozorgmehr, B., Najmabadi, H., Rodolico, C., Sieb, J.P., Steinlein, O.K., Schlotter, B., Schoser, B., Kirschner, J., Herrmann, R., Voit, T., Oldfors, A., Lindbergh, C., Urtizberea, A., von der Hagen, M., Hübner, A., Palace, J., Bushby, K., Straub, V., Beeson, D., Abicht, A., and Lochmüller, H. (2011) Hexosamine biosynthetic pathway mutations cause neuromuscular transmission defect. American journal of human genetics. 88(2):162-172
|
Serrano, R.J., Lee, C., Douek, A.M., Kaslin, J., Bryson-Richardson, R.J., Sztal, T.E. (2021) Novel pre-clinical model for CDKL5 Deficiency Disorder. Disease models & mechanisms. 15(3):
|
|
|
Shin, K.C., Hasan, W., Ali, G., Abdelrahman, D., Abuarja, T., Stanton, L.W., Da'as, S.I., Park, Y. (2025) Seizure-like behavior and hyperactivity in napb knockout zebrafish as a model for autism and epilepsy. Scientific Reports. 15:14579
|
|
|
|
|
Solheim, N., Pinho, B.R., Oliveira, N.A.S., Lima, L.P., Børve, A., Oliveira, J.M.A., Fladmark, K.E. (2026) Early motor deficits, sleep dysfunction and reduction in dopaminergic neurons in a PARK7-/- zebrafish larval model of Parkinson's disease. Scientific Reports. 16:9525
|
Solin, S.L., Wang, Y., Mauldin, J., Schultz, L.E., Lincow, D.E., Brodskiy, P.A., Jones, C.A., Syrkin-Nikolau, J., Linn, J.M., Essner, J.J., Hostetter, J.M., Whitley, E.M., Cameron, J.D., Chou, H.H., Severin, A.J., Sakaguchi, D.S., McGrail, M. (2014) Molecular and Cellular Characterization of a Zebrafish Optic Pathway Tumor Line Implicates Glia-Derived Progenitors in Tumorigenesis. PLoS One. 9:e114888
|
Son, W., Jeong, H.S., Nam, D.E., Lee, A.J., Nam, S.H., Lee, J.E., Choi, B.O., Chung, K.W. (2023) Peripheral Neuropathy and Decreased Locomotion of a RAB40B Mutation in Human and Model Animals. Experimental Neurobiology. 32:410422410-422
|
Song, Y., Selak, M.A., Watson, C.T., Coutts, C., Scherer, P.C., Panzer, J.A., Gibbs, S., Scott, M.O., Willer, G., Gregg, R.G., Ali, D.W., Bennett, M.J., and Balice-Gordon, R.J. (2009) Mechanisms Underlying Metabolic and Neural Defects in Zebrafish and Human Multiple Acyl-CoA Dehydrogenase Deficiency (MADD). PLoS One. 4(12):e8329
|
Song, Y., Willer, J.R., Scherer, P.C., Panzer, J.A., Kugath, A., Skordalakes, E., Gregg, R.G., Willer, G.B., and Balice-Gordon, R.J. (2010) Neural and Synaptic Defects in slytherin, a Zebrafish Model for Human Congenital Disorders of Glycosylation. PLoS One. 5(10):e13743
|
|
|
|
|
Sun, J., Pan, C.Q., Chew, T.W., Liang, F., Burmeister, M., Low, B.C. (2015) BNIP-H Recruits the Cholinergic Machinery to Neurite Terminals to Promote Acetylcholine Signaling and Neuritogenesis. Developmental Cell. 34(5):555-68
|
Swinnen, B., Bento-Abreu, A., Gendron, T.F., Boeynaems, S., Bogaert, E., Nuyts, R., Timmers, M., Scheveneels, W., Hersmus, N., Wang, J., Mizielinska, S., Isaacs, A.M., Petrucelli, L., Lemmens, R., Van Damme, P., Van Den Bosch, L., Robberecht, W. (2018) A zebrafish model for C9orf72 ALS reveals RNA toxicity as a pathogenic mechanism. Acta Neuropathologica. 135(3):427-443
|
|
|
Takesono, A., Schirrmacher, P., Scott, A., Green, J.M., Lee, O., Winter, M.J., Kudoh, T., Tyler, C.R. (2022) Estrogens regulate early embryonic development of the olfactory sensory system via estrogen-responsive glia. Development (Cambridge, England). 149(1):
|
Taylor, R., Hamid, F., Fielding, T., Gordon, P.M., Maloney, M., Makeyev, E.V., Houart, C. (2022) Prematurely terminated intron-retaining mRNAs invade axons in SFPQ null-driven neurodegeneration and are a hallmark of ALS. Nature communications. 13:6994
|
Tayran, H., Yilmaz, E., Bhattarai, P., Min, Y., Wang, X., Ma, Y., Wang, N., Jeong, I., Nelson, N., Kassara, N., Cosacak, M.I., Dogru, R.M., Reyes-Dumeyer, D., Stenersen, J.M., Reddy, J.S., Qiao, M., Flaherty, D., Gunasekaran, T.I., Yang, Z., Jurisch-Yaksi, N., Teich, A.F., Kanekiyo, T., Tosto, G., Vardarajan, B.N., İş, Ö., Ertekin-Taner, N., Mayeux, R., Kizil, C. (2024) ABCA7-dependent induction of neuropeptide Y is required for synaptic resilience in Alzheimer's disease through BDNF/NGFR signaling. Cell genomics. 4(9):100642
|
Turner, K.J., Hawkins, T.A., Henriques, P.M., Valdivia, L.E., Bianco, I.H., Wilson, S.W., Folgueira, M. (2022) A Structural Atlas of the Developing Zebrafish Telencephalon Based on Spatially-Restricted Transgene Expression. Frontiers in Neuroanatomy. 16:840924
|
Turner, K.J., Hawkins, T.A., Yáñez, J., Anadón, R., Wilson, S.W., Folgueira, M. (2016) Afferent Connectivity of the Zebrafish Habenulae. Frontiers in neural circuits. 10:30
|
Turner, K.J., Hoyle, J., Valdivia, L.E., Cerveny, K.L., Hart, W., Mangoli, M., Geisler, R., Rees, M., Houart, C., Poole, R.J., Wilson, S.W., Gestri, G. (2019) Abrogation of Stem Loop Binding Protein (Slbp) function leads to a failure of cells to transition from proliferation to differentiation, retinal coloboma and midline axon guidance deficits. PLoS One. 14:e0211073
|
Umeda, K., Shoji, W., Sakai, S., Muto, A., Kawakami, K., Ishizuka, T., and Yawo, H. (2013) Targeted expression of a chimeric channelrhodopsin in zebrafish under regulation of Gal4-UAS system. Neuroscience research. 75(1):69-75
|
Vaccaro, A., Patten, S.A., Ciura, S., Maios, C., Therrien, M., Drapeau, P., Kabashi, E., and Parker, J.A. (2012) Methylene Blue Protects against TDP-43 and FUS Neuronal Toxicity in C. elegans and D. rerio. PLoS One. 7(7):e42117
|
Van Houcke, J., Geeraerts, E., Vanhunsel, S., Beckers, A., Noterdaeme, L., Christiaens, M., Bollaerts, I., De Groef, L., Moons, L. (2018) Extensive growth is followed by neurodegenerative pathology in the continuously expanding adult zebrafish retina. Biogerontology. 20(1):109-125
|
Van Schoor, E., Strubbe, D., Braems, E., Weishaupt, J., Ludolph, A.C., Van Damme, P., Thal, D.R., Bercier, V., Van Den Bosch, L. (2024) TUBA4A downregulation as observed in ALS post-mortem motor cortex causes ALS-related abnormalities in zebrafish. Frontiers in Cellular Neuroscience. 18:1340240
|
Vorhees, N.W., Groenwold, S.L., Williams, M.T., Putt, L.S., Sanchez-Gama, N., Stalions, G.A., Taylor, G.M., Van Dort, H.E., Calvo-Ochoa, E. (2025) Olfactory Dysfunction in a Novel Model of Prodromal Parkinson's Disease in Adult Zebrafish. International Journal of Molecular Sciences. 26:
|
Wakisaka, N., Miyasaka, N., Koide, T., Masuda, M., Hiraki-Kajiyama, T., Yoshihara, Y. (2017) An Adenosine Receptor for Olfaction in Fish. Current biology : CB. 27(10):1437-1447.e4
|
|
|
Wen, H., Linhoff, M.W., McGinley, M.J., Li, G.L., Corson, G.M., Mandel, G., and Brehm, P. (2010) Distinct roles for two synaptotagmin isoforms in synchronous and asynchronous transmitter release at zebrafish neuromuscular junction. Proceedings of the National Academy of Sciences of the United States of America. 107(31):13906-13911
|
Weston, L.J., Cook, Z.T., Stackhouse, T.L., Sal, M.K., Schultz, B., Tobias, Z.J.C., Osterberg, V.R., Brockway, N.L., Pizano, S., Glover, G., Weissman, T.A., Unni, V.K. (2021) In vivo aggregation of presynaptic alpha-synuclein is not influenced by its phosphorylation at serine-129. Neurobiology of disease. 152:105291
|
Woehlbier, U., Colombo, A., Saaranen, M.J., Pérez, V., Ojeda, J., Bustos, F.J., Andreu, C.I., Torres, M., Valenzuela, V., Medinas, D.B., Rozas, P., Vidal, R.L., Lopez-Gonzalez, R., Salameh, J., Fernandez-Collemann, S., Muñoz, N., Matus, S., Armisen, R., Sagredo, A., Palma, K., Irrazabal, T., Almeida, S., Gonzalez-Perez, P., Campero, M., Gao, F.B., Henny, P., van Zundert, B., Ruddock, L.W., Concha, M.L., Henriquez, J.P., Brown, R.H., Hetz, C. (2016) ALS-linked protein disulfide isomerase variants cause motor dysfunction. The EMBO journal. 35(8):845-65
|
Xing, L., Hoshijima, K., Grunwald, D.J., Fujimoto, E., Quist, T.S., Sneddon, J., Chien, C.B., Stevenson, T.J., and Bonkowsky, J.L. (2012) Zebrafish foxP2 Zinc Finger Nuclease Mutant Has Normal Axon Pathfinding. PLoS One. 7(8):e43968
|
|
|
Yabuki, Y., Koide, T., Miyasaka, N., Wakisaka, N., Masuda, M., Ohkura, M., Nakai, J., Tsuge, K., Tsuchiya, S., Sugimoto, Y., Yoshihara, Y. (2016) Olfactory receptor for prostaglandin F2α mediates male fish courtship behavior. Nature Neuroscience. 19(7):897-904
|
|
|
Yu, Y.M., Cristofanilli, M., Valiveti, A., Ma, L., Yoo, M., Morellini, F., and Schachner, M. (2011) The extracellular matrix glycoprotein tenascin-C promotes locomotor recovery after spinal cord injury in adult zebrafish. Neuroscience. 183:238-50
|
|
|
|
|
|
|
|
|
Zelina, P., de Ruiter, A.A., Kolsteeg, C., van Ginneken, I., Vos, H.R., Supiot, L.F., Burgering, B.M.T., Meye, F.J., Veldink, J.H., van den Berg, L.H., Pasterkamp, R.J. (2024) ALS-associated C21ORF2 variant disrupts DNA damage repair, mitochondrial metabolism, neuronal excitability and NEK1 levels in human motor neurons. Acta neuropathologica communications. 12:144
|
|
|
