Published in Hum Mol Genet on October 29, 2008
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Dampened Hedgehog signaling but normal Wnt signaling in zebrafish without cilia. Development (2009) 1.93
Mutations in axonemal dynein assembly factor DNAAF3 cause primary ciliary dyskinesia. Nat Genet (2012) 1.78
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Human retinopathy-associated ciliary protein retinitis pigmentosa GTPase regulator mediates cilia-dependent vertebrate development. Hum Mol Genet (2010) 1.11
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The planar cell polarity gene Vangl2 is required for mammalian kidney-branching morphogenesis and glomerular maturation. Hum Mol Genet (2010) 1.10
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Expanding the molecular basis and phenotypic spectrum of X-linked Joubert syndrome associated with OFD1 mutations. Eur J Hum Genet (2012) 1.02
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Developmental localization of nephrin in zebrafish and medaka pronephric glomerulus. J Histochem Cytochem (2013) 0.88
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Cauli: a mouse strain with an Ift140 mutation that results in a skeletal ciliopathy modelling Jeune syndrome. PLoS Genet (2013) 0.86
Wnt and planar cell polarity signaling in cystic renal disease. Organogenesis (2013) 0.86
Lethal giant larvae 2 regulates development of the ciliated organ Kupffer's vesicle. Development (2013) 0.85
Modelling a ciliopathy: Ahi1 knockdown in model systems reveals an essential role in brain, retinal, and renal development. Cell Mol Life Sci (2011) 0.84
The Meckel syndrome protein meckelin (TMEM67) is a key regulator of cilia function but is not required for tissue planar polarity. Hum Mol Genet (2013) 0.83
Functional consequences of copy number variants in miscarriage. Mol Cytogenet (2015) 0.82
Structural disorganization of pronephric glomerulus in zebrafish mpp5a/nagie oko mutant. Dev Dyn (2012) 0.82
Cell polarity and cystic kidney disease. Pediatr Nephrol (2012) 0.82
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Polarity proteins are required for left-right axis orientation and twin-twin instruction. Genesis (2011) 0.80
Orphan G-protein coupled receptor 22 (Gpr22) regulates cilia length and structure in the zebrafish Kupffer's vesicle. PLoS One (2014) 0.79
Regional selection acting on the OFD1 gene family. PLoS One (2011) 0.79
Using the avian mutant talpid2 as a disease model for understanding the oral-facial phenotypes of oral-facial-digital syndrome. Dis Model Mech (2015) 0.79
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Cilia in autophagy and cancer. Cilia (2016) 0.78
Abnormal glycosylation in Joubert syndrome type 10. Cilia (2017) 0.77
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Mechanisms of otoconia and otolith development. Dev Dyn (2014) 0.77
Function Over Form: Modeling Groups of Inherited Neurological Conditions in Zebrafish. Front Mol Neurosci (2016) 0.76
Canonical and noncanonical intraflagellar transport regulates craniofacial skeletal development. Proc Natl Acad Sci U S A (2016) 0.76
nlz1 is required for cilia formation in zebrafish embryogenesis. Dev Biol (2015) 0.76
From zebrafish heart jogging genes to mouse and human orthologs: using Gene Ontology to investigate mammalian heart development. F1000Res (2013) 0.76
Expression and knockdown of zebrafish folliculin suggests requirement for embryonic brain morphogenesis. BMC Dev Biol (2016) 0.76
Novel roles for the radial spoke head protein 9 in neural and neurosensory cilia. Sci Rep (2016) 0.75
Oral-facial-digital syndrome type 1 in males: Congenital heart defects are included in its phenotypic spectrum. Am J Med Genet A (2017) 0.75
Basal body positioning and anchoring in the multiciliated cell Paramecium tetraurelia: roles of OFD1 and VFL3. Cilia (2017) 0.75
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Polycystins 1 and 2 mediate mechanosensation in the primary cilium of kidney cells. Nat Genet (2003) 13.33
High-resolution in situ hybridization to whole-mount zebrafish embryos. Nat Protoc (2008) 10.25
The ciliopathies: an emerging class of human genetic disorders. Annu Rev Genomics Hum Genet (2006) 9.56
Vertebrate Smoothened functions at the primary cilium. Nature (2005) 9.24
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Mutations in INVS encoding inversin cause nephronophthisis type 2, linking renal cystic disease to the function of primary cilia and left-right axis determination. Nat Genet (2003) 5.24
Kif3a constrains beta-catenin-dependent Wnt signalling through dual ciliary and non-ciliary mechanisms. Nat Cell Biol (2007) 5.09
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Two populations of node monocilia initiate left-right asymmetry in the mouse. Cell (2003) 4.73
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Shaping the vertebrate body plan by polarized embryonic cell movements. Science (2002) 4.35
Defective planar cell polarity in polycystic kidney disease. Nat Genet (2005) 4.31
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Disruption of the basal body compromises proteasomal function and perturbs intracellular Wnt response. Nat Genet (2007) 4.24
Xwnt11 is a target of Xenopus Brachyury: regulation of gastrulation movements via Dishevelled, but not through the canonical Wnt pathway. Development (2000) 4.06
Ciliogenesis defects in embryos lacking inturned or fuzzy function are associated with failure of planar cell polarity and Hedgehog signaling. Nat Genet (2006) 3.97
A genetic screen in zebrafish identifies cilia genes as a principal cause of cystic kidney. Development (2004) 3.97
Ciliary proteins link basal body polarization to planar cell polarity regulation. Nat Genet (2007) 3.79
Zebrafish trilobite identifies new roles for Strabismus in gastrulation and neuronal movements. Nat Cell Biol (2002) 3.49
Dysfunction of axonemal dynein heavy chain Mdnah5 inhibits ependymal flow and reveals a novel mechanism for hydrocephalus formation. Hum Mol Genet (2004) 2.63
The zebrafish nodal-related gene southpaw is required for visceral and diencephalic left-right asymmetry. Development (2003) 2.56
The roles of cilia in developmental disorders and disease. Development (2006) 2.52
Polaris, a protein disrupted in orpk mutant mice, is required for assembly of renal cilium. Am J Physiol Renal Physiol (2002) 2.37
Use of real-time quantitative PCR to validate the results of cDNA array and differential display PCR technologies. Methods (2001) 2.30
Intraflagellar transport genes are essential for differentiation and survival of vertebrate sensory neurons. Neuron (2004) 2.27
The zebrafish slow-muscle-omitted gene product is required for Hedgehog signal transduction and the development of slow muscle identity. Development (2000) 2.25
Oral-facial-digital type I protein is required for primary cilia formation and left-right axis specification. Nat Genet (2005) 2.24
Spatial regulation of a zebrafish patched homologue reflects the roles of sonic hedgehog and protein kinase A in neural tube and somite patterning. Development (1996) 2.18
Initial formation of zebrafish brain ventricles occurs independently of circulation and requires the nagie oko and snakehead/atp1a1a.1 gene products. Development (2005) 2.11
Identification of the gene for oral-facial-digital type I syndrome. Am J Hum Genet (2001) 2.07
Mutations in Hydin impair ciliary motility in mice. J Cell Biol (2008) 2.06
The Wilms tumor genes wt1a and wt1b control different steps during formation of the zebrafish pronephros. Dev Biol (2007) 1.91
Cystic kidney gene seahorse regulates cilia-mediated processes and Wnt pathways. Dev Cell (2008) 1.87
Regulation of primary cilia formation and left-right patterning in zebrafish by a noncanonical Wnt signaling mediator, duboraya. Nat Genet (2006) 1.76
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Regulation of midline development by antagonism of lefty and nodal signaling. Development (1999) 1.67
Asymmetric nodal signaling in the zebrafish diencephalon positions the pineal organ. Development (2000) 1.62
Wnt signaling in polycystic kidney disease. J Am Soc Nephrol (2007) 1.58
A novel X-linked recessive mental retardation syndrome comprising macrocephaly and ciliary dysfunction is allelic to oral-facial-digital type I syndrome. Hum Genet (2006) 1.51
Polycystin-2 immunolocalization and function in zebrafish. J Am Soc Nephrol (2006) 1.50
OFD1 is a centrosomal/basal body protein expressed during mesenchymal-epithelial transition in human nephrogenesis. J Am Soc Nephrol (2004) 1.43
Clinical, molecular, and genotype-phenotype correlation studies from 25 cases of oral-facial-digital syndrome type 1: a French and Belgian collaborative study. J Med Genet (2006) 1.42
Van Gogh-like2 (Strabismus) and its role in planar cell polarity and convergent extension in vertebrates. Trends Genet (2004) 1.36
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Making a zebrafish kidney: a tale of two tubes. Trends Cell Biol (2003) 1.29
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New functions for a vertebrate Rho guanine nucleotide exchange factor in ciliated epithelia. Development (2007) 1.26
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The mouse homeobox gene Noto regulates node morphogenesis, notochordal ciliogenesis, and left right patterning. Proc Natl Acad Sci U S A (2007) 1.17
Diversin regulates heart formation and gastrulation movements in development. Proc Natl Acad Sci U S A (2006) 1.16
Mutational spectrum of the oral-facial-digital type I syndrome: a study on a large collection of patients. Hum Mutat (2008) 1.13
Parallel early development of zebrafish interrenal glands and pronephros: differential control by wt1 and ff1b. Development (2003) 1.13
The oral-facial-digital syndrome type 1 (OFD1), a cause of polycystic kidney disease and associated malformations, maps to Xp22.2-Xp22.3. Hum Mol Genet (1997) 1.10
The zebrafish floating head mutant demonstrates podocytes play an important role in directing glomerular differentiation. Dev Biol (2000) 1.09
Elucidation of megalin/LRP2-dependent endocytic transport processes in the larval zebrafish pronephros. J Cell Sci (2006) 1.08
OFD1, the gene mutated in oral-facial-digital syndrome type 1, is expressed in the metanephros and in human embryonic renal mesenchymal cells. J Am Soc Nephrol (2003) 1.08
Analysis of Kupffer's vesicle in zebrafish embryos using a cave automated virtual environment. Dev Dyn (2007) 1.07
Proneural gene requirement for hair cell differentiation in the zebrafish lateral line. Dev Biol (2006) 1.03
Altered pH(i) regulation and Na(+)/HCO3(-) transporter activity in choroid plexus of cilia-defective Tg737(orpk) mutant mouse. Am J Physiol Cell Physiol (2006) 0.98
Csrp1 regulates dynamic cell movements of the mesendoderm and cardiac mesoderm through interactions with Dishevelled and Diversin. Proc Natl Acad Sci U S A (2007) 0.91
Characterization of the OFD1/Ofd1 genes on the human and mouse sex chromosomes and exclusion of Ofd1 for the Xpl mouse mutant. Genomics (2003) 0.90
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FRT-seq: amplification-free, strand-specific transcriptome sequencing. Nat Methods (2010) 3.55
DNA detection using recombination proteins. PLoS Biol (2006) 3.53
A systematic genome-wide analysis of zebrafish protein-coding gene function. Nature (2013) 3.52
The deubiquitinase USP9X suppresses pancreatic ductal adenocarcinoma. Nature (2012) 3.23
MicroRNAs show a wide diversity of expression profiles in the developing and mature central nervous system. Genome Biol (2007) 3.05
Compound inheritance of a low-frequency regulatory SNP and a rare null mutation in exon-junction complex subunit RBM8A causes TAR syndrome. Nat Genet (2012) 2.80
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Prickle 1 regulates cell movements during gastrulation and neuronal migration in zebrafish. Development (2003) 2.26
Mutations in radial spoke head protein genes RSPH9 and RSPH4A cause primary ciliary dyskinesia with central-microtubular-pair abnormalities. Am J Hum Genet (2009) 2.07
Podocyte-specific expression of angiopoietin-2 causes proteinuria and apoptosis of glomerular endothelia. J Am Soc Nephrol (2007) 2.06
Laterotopic representation of left-right information onto the dorso-ventral axis of a zebrafish midbrain target nucleus. Curr Biol (2005) 2.03
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An integrated functional genomics approach identifies the regulatory network directed by brachyury (T) in chordoma. J Pathol (2012) 1.95
Mutations in ISPD cause Walker-Warburg syndrome and defective glycosylation of α-dystroglycan. Nat Genet (2012) 1.90
Incorporating RNA-seq data into the zebrafish Ensembl genebuild. Genome Res (2012) 1.89
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Disruption of ROBO2 is associated with urinary tract anomalies and confers risk of vesicoureteral reflux. Am J Hum Genet (2007) 1.85
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Fraser syndrome and mouse blebbed phenotype caused by mutations in FRAS1/Fras1 encoding a putative extracellular matrix protein. Nat Genet (2003) 1.71
Multiple mutations in mouse Chd7 provide models for CHARGE syndrome. Hum Mol Genet (2005) 1.71
Angiopoietin-1 therapy enhances fibrosis and inflammation following folic acid-induced acute renal injury. Kidney Int (2008) 1.70
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Removal of dystroglycan causes severe muscular dystrophy in zebrafish embryos. Development (2002) 1.66
Centriolar satellites are assembly points for proteins implicated in human ciliopathies, including oral-facial-digital syndrome 1. J Cell Sci (2011) 1.64
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Early stages of zebrafish eye formation require the coordinated activity of Wnt11, Fz5, and the Wnt/beta-catenin pathway. Neuron (2005) 1.62
The genetic heterogeneity and mutational burden of engineered melanomas in zebrafish models. Genome Biol (2013) 1.54
Distinct roles for Fgf, Wnt and retinoic acid in posteriorizing the neural ectoderm. Development (2002) 1.54
The ATPase-dependent chaperoning activity of Hsp90a regulates thick filament formation and integration during skeletal muscle myofibrillogenesis. Development (2008) 1.52
Identification of a new gene mutated in Fraser syndrome and mouse myelencephalic blebs. Nat Genet (2005) 1.51
Morphogenesis underlying the development of the everted teleost telencephalon. Neural Dev (2012) 1.51
Mutations in GDP-mannose pyrophosphorylase B cause congenital and limb-girdle muscular dystrophies associated with hypoglycosylation of α-dystroglycan. Am J Hum Genet (2013) 1.50
Retinoic acid signalling in the zebrafish embryo is necessary during pre-segmentation stages to pattern the anterior-posterior axis of the CNS and to induce a pectoral fin bud. Development (2002) 1.50
Functional genomics in zebrafish permits rapid characterization of novel platelet membrane proteins. Blood (2008) 1.45
Eph/Ephrin signaling regulates the mesenchymal-to-epithelial transition of the paraxial mesoderm during somite morphogenesis. Curr Biol (2003) 1.45
Slb/Wnt11 controls hypoblast cell migration and morphogenesis at the onset of zebrafish gastrulation. Development (2003) 1.45
Hedgehog signalling maintains the optic stalk-retinal interface through the regulation of Vax gene activity. Development (2003) 1.43
OFD1 is a centrosomal/basal body protein expressed during mesenchymal-epithelial transition in human nephrogenesis. J Am Soc Nephrol (2004) 1.43
Teashirt 3 is necessary for ureteral smooth muscle differentiation downstream of SHH and BMP4. Development (2008) 1.42
Lefty antagonism of Squint is essential for normal gastrulation. Curr Biol (2002) 1.39
Primary vesicoureteric reflux as a predictor of renal damage in children hospitalized with urinary tract infection: a systematic review and meta-analysis. J Am Soc Nephrol (2003) 1.37
Missense mutations in β-1,3-N-acetylglucosaminyltransferase 1 (B3GNT1) cause Walker-Warburg syndrome. Hum Mol Genet (2013) 1.36
Genetic screens for mutations affecting development of Xenopus tropicalis. PLoS Genet (2006) 1.33
Essential and overlapping roles for laminin alpha chains in notochord and blood vessel formation. Dev Biol (2006) 1.32
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HNF1B mutations associate with hypomagnesemia and renal magnesium wasting. J Am Soc Nephrol (2009) 1.29
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Protein restriction in pregnancy is associated with increased apoptosis of mesenchymal cells at the start of rat metanephrogenesis. Kidney Int (2002) 1.26
Comparative transcriptome analyses indicate molecular homology of zebrafish swimbladder and mammalian lung. PLoS One (2011) 1.25
Mutations in HPSE2 cause urofacial syndrome. Am J Hum Genet (2010) 1.24
Wnt/Axin1/beta-catenin signaling regulates asymmetric nodal activation, elaboration, and concordance of CNS asymmetries. Neuron (2007) 1.23
Brain asymmetry is encoded at the level of axon terminal morphology. Neural Dev (2008) 1.21
A genetic map of Xenopus tropicalis. Dev Biol (2011) 1.20
Solitary functioning kidney and diverse genital tract malformations associated with hepatocyte nuclear factor-1beta mutations. Kidney Int (2002) 1.18
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Combinatorial Fgf and Bmp signalling patterns the gastrula ectoderm into prospective neural and epidermal domains. Development (2004) 1.17
Six3 functions in anterior neural plate specification by promoting cell proliferation and inhibiting Bmp4 expression. Development (2005) 1.17
A family of acid-sensing ion channels from the zebrafish: widespread expression in the central nervous system suggests a conserved role in neuronal communication. J Biol Chem (2004) 1.16
Differential requirements for COPI transport during vertebrate early development. Dev Cell (2004) 1.16
De novo Uroplakin IIIa heterozygous mutations cause human renal adysplasia leading to severe kidney failure. J Am Soc Nephrol (2005) 1.15
High-throughput target-selected gene inactivation in zebrafish. Methods Cell Biol (2011) 1.15
Flamingo regulates epiboly and convergence/extension movements through cell cohesive and signalling functions during zebrafish gastrulation. Development (2008) 1.13
Nodal signalling imposes left-right asymmetry upon neurogenesis in the habenular nuclei. Development (2009) 1.10
The planar cell polarity gene Vangl2 is required for mammalian kidney-branching morphogenesis and glomerular maturation. Hum Mol Genet (2010) 1.10
Mutations in c10orf11, a melanocyte-differentiation gene, cause autosomal-recessive albinism. Am J Hum Genet (2013) 1.10
Lef1-dependent Wnt/β-catenin signalling drives the proliferative engine that maintains tissue homeostasis during lateral line development. Development (2011) 1.09
Zebrafish Fukutin family proteins link the unfolded protein response with dystroglycanopathies. Hum Mol Genet (2011) 1.09
The emerging use of zebrafish to model metabolic disease. Dis Model Mech (2013) 1.09
Heparan sulfate 6-o-sulfotransferase is essential for muscle development in zebrafish. J Biol Chem (2003) 1.08
OFD1, the gene mutated in oral-facial-digital syndrome type 1, is expressed in the metanephros and in human embryonic renal mesenchymal cells. J Am Soc Nephrol (2003) 1.08
A paradoxical teratogenic mechanism for retinoic acid. Proc Natl Acad Sci U S A (2012) 1.08
Monorail/Foxa2 regulates floorplate differentiation and specification of oligodendrocytes, serotonergic raphé neurones and cranial motoneurones. Development (2005) 1.07
An Fgf8-dependent bistable cell migratory event establishes CNS asymmetry. Neuron (2009) 1.06
Genetic and genomic prospects for Xenopus tropicalis research. Semin Cell Dev Biol (2006) 1.06
Ash1a and Neurogenin1 function downstream of Floating head to regulate epiphysial neurogenesis. Development (2003) 1.06
Dynamic coupling of pattern formation and morphogenesis in the developing vertebrate retina. PLoS Biol (2009) 1.05
Correction of G551D-CFTR transport defect in epithelial monolayers by genistein but not by CPX or MPB-07. Br J Pharmacol (2002) 1.05
Zebrafish notochordal basement membrane: signaling and structure. Curr Top Dev Biol (2005) 1.05
Conserved and divergent patterns of Reelin expression in the zebrafish central nervous system. J Comp Neurol (2002) 1.04
Organ-specific requirements for Hdac1 in liver and pancreas formation. Dev Biol (2008) 1.04
Maternal diet programs embryonic kidney gene expression. Physiol Genomics (2005) 1.03
Neurogenin1 is a determinant of zebrafish basal forebrain dopaminergic neurons and is regulated by the conserved zinc finger protein Tof/Fezl. Proc Natl Acad Sci U S A (2006) 1.02
Multi-allelic phenotyping--a systematic approach for the simultaneous analysis of multiple induced mutations. Methods (2013) 1.01
The zebrafish dystrophic mutant softy maintains muscle fibre viability despite basement membrane rupture and muscle detachment. Development (2009) 1.01
Distinct and cooperative roles for Nodal and Hedgehog signals during hypothalamic development. Development (2002) 1.01
Breaking symmetry: the zebrafish as a model for understanding left-right asymmetry in the developing brain. Dev Neurobiol (2012) 1.00
The future of model organisms in human disease research. Nat Rev Genet (2011) 1.00
Hedgehog signaling patterns the outgrowth of unpaired skeletal appendages in zebrafish. BMC Dev Biol (2007) 0.99
Fras1, a basement membrane-associated protein mutated in Fraser syndrome, mediates both the initiation of the mammalian kidney and the integrity of renal glomeruli. Hum Mol Genet (2008) 0.99