Published in Development on June 27, 2007
Linking human diseases to animal models using ontology-based phenotype annotation. PLoS Biol (2009) 5.17
In vivo protein trapping produces a functional expression codex of the vertebrate proteome. Nat Methods (2011) 2.85
Loss of myotubularin function results in T-tubule disorganization in zebrafish and human myotubular myopathy. PLoS Genet (2009) 2.23
Congenital myopathies: an update. Curr Neurol Neurosci Rep (2012) 1.66
Stac3 is a component of the excitation-contraction coupling machinery and mutated in Native American myopathy. Nat Commun (2013) 1.35
Selenoprotein N is required for ryanodine receptor calcium release channel activity in human and zebrafish muscle. Proc Natl Acad Sci U S A (2008) 1.34
The zebrafish dag1 mutant: a novel genetic model for dystroglycanopathies. Hum Mol Genet (2011) 1.30
Rbfox-regulated alternative splicing is critical for zebrafish cardiac and skeletal muscle functions. Dev Biol (2011) 1.16
Oxidative stress and successful antioxidant treatment in models of RYR1-related myopathy. Brain (2012) 1.14
Proper restoration of excitation-contraction coupling in the dihydropyridine receptor beta1-null zebrafish relaxed is an exclusive function of the beta1a subunit. J Biol Chem (2008) 1.14
Single channel properties of heterotetrameric mutant RyR1 ion channels linked to core myopathies. J Biol Chem (2008) 1.01
Non-Ca2+-conducting Ca2+ channels in fish skeletal muscle excitation-contraction coupling. Proc Natl Acad Sci U S A (2010) 0.99
Swimming into prominence: the zebrafish as a valuable tool for studying human myopathies and muscular dystrophies. FEBS J (2013) 0.97
Structure-activity relationship of non-coplanar polychlorinated biphenyls toward skeletal muscle ryanodine receptors in rainbow trout (Oncorhynchus mykiss). Aquat Toxicol (2013) 0.96
Multi-minicore disease and atypical periodic paralysis associated with novel mutations in the skeletal muscle ryanodine receptor (RYR1) gene. Neuromuscul Disord (2010) 0.95
Analysis of skeletal muscle defects in larval zebrafish by birefringence and touch-evoke escape response assays. J Vis Exp (2013) 0.92
Genotype-phenotype correlations in recessive RYR1-related myopathies. Orphanet J Rare Dis (2013) 0.90
Defective glycinergic synaptic transmission in zebrafish motility mutants. Front Mol Neurosci (2010) 0.88
Neuromuscular effects of G93A-SOD1 expression in zebrafish. Mol Neurodegener (2012) 0.88
Pbx and Prdm1a transcription factors differentially regulate subsets of the fast skeletal muscle program in zebrafish. Biol Open (2013) 0.86
Triadopathies: an emerging class of skeletal muscle diseases. Neurotherapeutics (2014) 0.86
α-Actinin-2 deficiency results in sarcomeric defects in zebrafish that cannot be rescued by α-actinin-3 revealing functional differences between sarcomeric isoforms. FASEB J (2012) 0.86
Pathophysiological concepts in the congenital myopathies: blurring the boundaries, sharpening the focus. Brain (2014) 0.86
Ryanodine receptors, a family of intracellular calcium ion channels, are expressed throughout early vertebrate development. BMC Res Notes (2011) 0.84
Touch responsiveness in zebrafish requires voltage-gated calcium channel 2.1b. J Neurophysiol (2012) 0.83
Transcriptional response of zebrafish embryos exposed to neurotoxic compounds reveals a muscle activity dependent hspb11 expression. PLoS One (2011) 0.82
Force measurement during contraction to assess muscle function in zebrafish larvae. J Vis Exp (2013) 0.82
Na(v)1.6a is required for normal activation of motor circuits normally excited by tactile stimulation. Dev Neurobiol (2010) 0.81
Duplicated gephyrin genes showing distinct tissue distribution and alternative splicing patterns mediate molybdenum cofactor biosynthesis, glycine receptor clustering, and escape behavior in zebrafish. J Biol Chem (2010) 0.80
Connexin 39.9 protein is necessary for coordinated activation of slow-twitch muscle and normal behavior in zebrafish. J Biol Chem (2011) 0.80
The role of Sox6 in zebrafish muscle fiber type specification. Skelet Muscle (2015) 0.79
Triclosan impairs swimming behavior and alters expression of excitation-contraction coupling proteins in fathead minnow (Pimephales promelas). Environ Sci Technol (2013) 0.79
Zebrafish needle EMG: a new tool for high-throughput drug screens. J Neurophysiol (2015) 0.78
Recent advances using zebrafish animal models for muscle disease drug discovery. Expert Opin Drug Discov (2014) 0.77
Impaired embryonic motility in dusp27 mutants reveals a developmental defect in myofibril structure. Dis Model Mech (2013) 0.77
The signaling protein CD38 is essential for early embryonic development. J Biol Chem (2012) 0.76
Defects of the Glycinergic Synapse in Zebrafish. Front Mol Neurosci (2016) 0.75
Candidate exome capture identifies mutation of SDCCAG8 as the cause of a retinal-renal ciliopathy. Nat Genet (2010) 3.99
Nephronophthisis-associated ciliopathies. J Am Soc Nephrol (2007) 3.40
A systematic approach to mapping recessive disease genes in individuals from outbred populations. PLoS Genet (2009) 3.06
Roles of bHLH genes in neural stem cell differentiation. Exp Cell Res (2005) 2.73
Exome capture reveals ZNF423 and CEP164 mutations, linking renal ciliopathies to DNA damage response signaling. Cell (2012) 2.54
Mutations in mitochondrial carrier family gene SLC25A38 cause nonsyndromic autosomal recessive congenital sideroblastic anemia. Nat Genet (2009) 2.37
Loss of myotubularin function results in T-tubule disorganization in zebrafish and human myotubular myopathy. PLoS Genet (2009) 2.23
COQ6 mutations in human patients produce nephrotic syndrome with sensorineural deafness. J Clin Invest (2011) 2.21
Mutation analysis in nephronophthisis using a combined approach of homozygosity mapping, CEL I endonuclease cleavage, and direct sequencing. Hum Mutat (2008) 1.91
Mutation analysis of 18 nephronophthisis associated ciliopathy disease genes using a DNA pooling and next generation sequencing strategy. J Med Genet (2010) 1.76
ADCK4 mutations promote steroid-resistant nephrotic syndrome through CoQ10 biosynthesis disruption. J Clin Invest (2013) 1.74
KANK deficiency leads to podocyte dysfunction and nephrotic syndrome. J Clin Invest (2015) 1.69
Assaying autophagic activity in transgenic GFP-Lc3 and GFP-Gabarap zebrafish embryos. Autophagy (2009) 1.57
The heat-inducible zebrafish hsp70 gene is expressed during normal lens development under non-stress conditions. Mech Dev (2002) 1.57
Mutation of the Mg2+ transporter SLC41A1 results in a nephronophthisis-like phenotype. J Am Soc Nephrol (2013) 1.52
ARHGDIA mutations cause nephrotic syndrome via defective RHO GTPase signaling. J Clin Invest (2013) 1.52
Persistent and high levels of Hes1 expression regulate boundary formation in the developing central nervous system. Development (2006) 1.48
Development of the locomotor network in zebrafish. Prog Neurobiol (2002) 1.47
Stac3 is a component of the excitation-contraction coupling machinery and mutated in Native American myopathy. Nat Commun (2013) 1.35
Zebrafish bandoneon mutants display behavioral defects due to a mutation in the glycine receptor beta-subunit. Proc Natl Acad Sci U S A (2005) 1.28
ZMYND10 is mutated in primary ciliary dyskinesia and interacts with LRRC6. Am J Hum Genet (2013) 1.24
Developmental toxicology of cadmium in living embryos of a stable transgenic zebrafish line. Environ Health Perspect (2002) 1.23
accordion, a zebrafish behavioral mutant, has a muscle relaxation defect due to a mutation in the ATPase Ca2+ pump SERCA1. Development (2004) 1.23
Semaphorin3a1 regulates angioblast migration and vascular development in zebrafish embryos. Development (2003) 1.20
The retinitis pigmentosa protein RP2 interacts with polycystin 2 and regulates cilia-mediated vertebrate development. Hum Mol Genet (2010) 1.20
Individuals with mutations in XPNPEP3, which encodes a mitochondrial protein, develop a nephronophthisis-like nephropathy. J Clin Invest (2010) 1.20
Chemokine signaling regulates sensory cell migration in zebrafish. Dev Biol (2004) 1.20
Oxidative stress and successful antioxidant treatment in models of RYR1-related myopathy. Brain (2012) 1.14
Steps during the development of the zebrafish locomotor network. J Physiol Paris (2003) 1.14
Hes repressors are essential regulators of hematopoietic stem cell development downstream of Notch signaling. J Exp Med (2012) 1.13
A quantitative analysis of the kinetics of Gal4 activator and effector gene expression in the zebrafish. Mech Dev (2002) 1.09
Glutamate drives the touch response through a rostral loop in the spinal cord of zebrafish embryos. Dev Neurobiol (2009) 1.04
Identification of nonvisual photomotor response cells in the vertebrate hindbrain. J Neurosci (2013) 1.04
Chemokine signaling guides axons within the retina in zebrafish. J Neurosci (2005) 1.03
Involvement of Islet-2 in the Slit signaling for axonal branching and defasciculation of the sensory neurons in embryonic zebrafish. Mech Dev (2004) 1.03
Genetic modifiers of otocephalic phenotypes in Otx2 heterozygous mutant mice. Development (2002) 1.02
The zebrafish shocked gene encodes a glycine transporter and is essential for the function of early neural circuits in the CNS. J Neurosci (2005) 1.02
bHLH transcription factor Her5 links patterning to regional inhibition of neurogenesis at the midbrain-hindbrain boundary. Development (2003) 0.98
Non-sense mutations in the dihydropyridine receptor beta1 gene, CACNB1, paralyze zebrafish relaxed mutants. Cell Calcium (2005) 0.98
TRPM7 is required within zebrafish sensory neurons for the activation of touch-evoked escape behaviors. J Neurosci (2011) 0.95
shocked Gene is required for the function of a premotor network in the zebrafish CNS. J Neurophysiol (2004) 0.93
Semaphorin3D guides retinal axons along the dorsoventral axis of the tectum. J Neurosci (2004) 0.93
Transmembrane sema4E guides branchiomotor axons to their targets in zebrafish. J Neurosci (2003) 0.92
Biomineralization and size control of stable calcium phosphate core-protein shell nanoparticles: potential for vaccine applications. Bioconjug Chem (2012) 0.92
Roles of the bHLH gene Hes1 in retinal morphogenesis. Brain Res (2004) 0.91
Sema3a1 guides spinal motor axons in a cell- and stage-specific manner in zebrafish. Development (2006) 0.90
Identification and expression of voltage-gated calcium channel beta subunits in Zebrafish. Dev Dyn (2008) 0.90
A mutation in the RNF170 gene causes autosomal dominant sensory ataxia. Brain (2010) 0.90
The zebrafish ennui behavioral mutation disrupts acetylcholine receptor localization and motor axon stability. Dev Neurobiol (2008) 0.89
Caspy, a zebrafish caspase, activated by ASC oligomerization is required for pharyngeal arch development. J Biol Chem (2002) 0.89
Acetylcholine receptor clustering is required for the accumulation and maintenance of scaffolding proteins. Curr Biol (2008) 0.89
Defective glycinergic synaptic transmission in zebrafish motility mutants. Front Mol Neurosci (2010) 0.88
Integrity of N- and C-termini is important for E. coli Hsp31 chaperone activity. Protein Sci (2009) 0.87
touché Is required for touch-evoked generator potentials within vertebrate sensory neurons. J Neurosci (2010) 0.86
Myo1c is an unconventional myosin required for zebrafish glomerular development. Kidney Int (2013) 0.86
Biogenesis of GPI-anchored proteins is essential for surface expression of sodium channels in zebrafish Rohon-Beard neurons to respond to mechanosensory stimulation. Development (2010) 0.85
Touch responsiveness in zebrafish requires voltage-gated calcium channel 2.1b. J Neurophysiol (2012) 0.83
The neuronal growth and regeneration associated Cntn1 (F3/F11/Contactin) gene is duplicated in fish: expression during development and retinal axon regeneration. Mol Cell Neurosci (2005) 0.83
Na(v)1.6a is required for normal activation of motor circuits normally excited by tactile stimulation. Dev Neurobiol (2010) 0.81
Cellular, genetic and molecular mechanisms of axonal guidance in the zebrafish. Results Probl Cell Differ (2002) 0.80
The cfy mutation disrupts cell divisions in a stage-dependent manner in zebrafish embryos. Dev Biol (2004) 0.80
Connexin 39.9 protein is necessary for coordinated activation of slow-twitch muscle and normal behavior in zebrafish. J Biol Chem (2011) 0.80
Wnt5a is necessary for normal kidney development in zebrafish and mice. Nephron Exp Nephrol (2014) 0.80
Retroviral vectors for gene delivery to neural precursor cells. Int Rev Neurobiol (2003) 0.79
Comparison of intrabursal transfer of spermatozoa, a new method for artificial insemination in mice, with intraoviductal transfer of spermatozoa. J Assist Reprod Genet (2002) 0.78