A potassium channel mutation in weaver mice implicates membrane excitability in granule cell differentiation.
paper
Cited
Public
Unavailable
- Authors
- Patil, N; Cox, D R; Bhat, D; Faham, M; Myers, R M; Peterson, A S
- Year
- 1995
- Journal
- Nature genetics
- PMID
- 7550338
- DOI
- 10.1038/ng1095-126
Early events in neuronal differentiation are generally considered to be regulated by factors independent of alterations in membrane permeability. Weaver mice harbour a mutation that blocks neuronal differentiation just after cessation of cell division, prior to cell migration and synaptogenesis. Cerebellar granule cells in homozygous weaver mice fail to differentiate, either because intrinsic cues are absent or because the granule cells are unable to respond to those cues. We now report that weaver mice have a missense mutation in a gene encoding a G-protein coupled inward rectifier potassium channel. The mutation alters the putative ion-permeable, pore-forming domain of the protein, suggesting that granule cell differentiation is regulated by changes in membrane permeability.
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| Dominant-negative mutants identify a role for GIRK channels in D3 dopamine receptor-mediated regulation of spontaneous secretory activity. | Kuzhikandathil EV et al. | β | 2000 | β |
| Evidence of elevated intracellular calcium levels in weaver homozygote mice. | Harkins AB et al. | β | 2000 | β |
| Gain of function mutants: ion channels and G protein-coupled receptors. | Lester HA et al. | β | 2000 | β |
| G-protein mediated gating of inward-rectifier K+ channels. | Mark MD et al. | β | 2000 | β |
| Impaired inhibition of epileptiform activity by baclofen, but not by adenosine in the weaver hippocampus. | Jarolimek W et al. | β | 2000 | β |
| Inhibition by various antipsychotic drugs of the G-protein-activated inwardly rectifying K(+) (GIRK) channels expressed in xenopus oocytes. | Kobayashi T et al. | β | 2000 | β |
| Insights from mouse models into the molecular basis of neurodegeneration. | Heintz N et al. | β | 2000 | β |
| Involvement of GIRK2 in postnatal development of the weaver cerebellum. | Liesi P et al. | β | 2000 | β |
| Involvement of G-protein-activated inwardly rectifying K (GIRK) channels in opioid-induced analgesia. | Ikeda K et al. | β | 2000 | β |
| Is there a role for potassium channel openers in neuronal ion channel disorders? | Lawson K | β | 2000 | β |
| Lineage, development and morphogenesis of cerebellar interneurons. | Schilling K | β | 2000 | β |
| Modulation of GABA(A) receptor subunit mRNA levels in olivocerebellar neurons of purkinje cell degeneration and weaver mutant mice. | Rotter A et al. | β | 2000 | β |
| Mutation analysis of the inwardly rectifying K(+) channels KCNJ6 (GIRK2) and KCNJ3 (GIRK1) in juvenile myoclonic epilepsy. | Hallmann K et al. | β | 2000 | β |
| Paradoxical increase of tyrosine hydroxylase-immunoreactive retinopetal fibers in the weaver mouse. | Simon A et al. | β | 2000 | β |
| The anticonvulsant retigabine potently suppresses epileptiform discharges in the low Ca ++ and low Mg++ model in the hippocampal slice preparation. | Dost R et al. | β | 2000 | β |
| The weaver gene continues to target late-generated dopaminergic neurons in midbrain areas at P90. | MartΓ J et al. | β | 2000 | β |
| The weaver mutation reverses the function of dopamine and GABA in mouse dopaminergic neurons. | Guatteo E et al. | β | 2000 | β |
| Ultrastructural analysis of catecholaminergic innervation in weaver and normal mouse cerebellar cortices. | Abbott LC et al. | β | 2000 | β |
| [3H]CNQX and NMDA-sensitive [3H]glutamate binding sites and AMPA receptor subunit RNA transcripts in the striatum of normal and weaver mutant mice and effects of ventral mesencephalic grafts. | Mitsacos A et al. | β | 1999 | β |
| Abortive synaptogenesis as a factor in the inner hair cell degeneration in the Bronx Waltzer (bv) mutant mouse. | Sobkowicz HM et al. | β | 1999 | β |
| A cell cycle alteration precedes apoptosis of granule cell precursors in the weaver mouse cerebellum. | Migheli A et al. | β | 1999 | β |
| Alterations in serotonin receptors in the neostriatum of weaver mutant mice. | Dewar KM | β | 1999 | β |
| Altered development of dopaminergic cells in the retina of weaver mice. | Savy C et al. | β | 1999 | β |
| Alternative sulfonylurea receptor expression defines metabolic sensitivity of K-ATP channels in dopaminergic midbrain neurons. | Liss B et al. | β | 1999 | β |
| Benign familial neonatal epilepsy with mutations in two potassium channel genes. | Leppert M et al. | β | 1999 | β |
| Cell cycle events in neurons. Proliferation or death? | Zhu X et al. | β | 1999 | β |
| Channelopathies of inwardly rectifying potassium channels. | Abraham MR et al. | β | 1999 | β |
| Characterization of G-protein-gated K+ channels composed of Kir3.2 subunits in dopaminergic neurons of the substantia nigra. | Inanobe A et al. | β | 1999 | β |
| Characterization of the K+ channel opening effect of the anticonvulsant retigabine in PC12 cells. | Rundfeldt C | β | 1999 | β |
| Cloning of a new mouse two-P domain channel subunit and a human homologue with a unique pore structure. | Salinas M et al. | β | 1999 | β |
| Developmental expression of voltage-gated potassium channel beta subunits. | Downen M et al. | β | 1999 | β |
| Distribution of dopamine, its metabolites, and D1 and D2 receptors in heterozygous and homozygous weaver mutant mice. | Reader TA et al. | β | 1999 | β |
| Down-regulated expression of glutamate transporter GLAST in Purkinje cell-associated astrocytes of reeler and weaver mutant cerebella. | Fukaya M et al. | β | 1999 | β |
| Dysfunction of delayed rectifier potassium channels in an inherited cardiac arrhythmia. | Sanguinetti MC | β | 1999 | β |
| Effects of Conus peptides on the behavior of mice. | Olivera BM et al. | β | 1999 | β |
| Ethanol opens G-protein-activated inwardly rectifying K+ channels. | Kobayashi T et al. | β | 1999 | β |
| Etiology of Parkinson's disease. | Stoessl AJ | β | 1999 | β |
| GIRK4 confers appropriate processing and cell surface localization to G-protein-gated potassium channels. | Kennedy ME et al. | β | 1999 | β |
| G protein gated potassium channels. | Sui JL et al. | β | 1999 | β |
| Ion channels and the genetic contribution to epilepsy. | Ryan SG | β | 1999 | β |
| Ion channels as physiological effectors for growth factor receptor and Ras/ERK signaling pathways. | Rane SG | β | 1999 | β |
| Isolation and analysis of chromosome 21 genes potentially involved in Down syndrome. | Gosset P et al. | β | 1999 | β |
| Localization and age-dependent expression of the inward rectifier K+ channel subunit Kir 5.1 in a mammalian reproductive system. | Salvatore L et al. | β | 1999 | β |
| Migration defects of cdk5(-/-) neurons in the developing cerebellum is cell autonomous. | Ohshima T et al. | β | 1999 | β |
| Molecular biology of adenosine triphosphate-sensitive potassium channels. | Aguilar-Bryan L et al. | β | 1999 | β |
| Molecular cloning and characterization of a novel splicing variant of the Kir3.2 subunit predominantly expressed in mouse testis. | Inanobe A et al. | β | 1999 | β |
| Neuronal migration is retarded in mice lacking the tissue plasminogen activator gene. | Seeds NW et al. | β | 1999 | β |
| Neurons exhibiting dopamine D2 receptor immunoreactivity in the substantia nigra of the mutant weaver mouse. | Xu SG et al. | β | 1999 | β |
| Partial restoration of striatal GABAA receptor balance by functional mesencephalic dopaminergic grafts in mice with hereditary parkinsonism. | Stasi K et al. | β | 1999 | β |
| Potassium channels: basic aspects, functional roles, and medical significance. | Yost CS | β | 1999 | β |
| Recent advances in the genetics of epilepsy: insights from human and animal studies. | Prasad AN et al. | β | 1999 | β |
| Rescue of cerebellar granule cells from death in weaver NR1 double mutants. | Jensen P et al. | β | 1999 | β |
| Retigabine strongly reduces repetitive firing in rat entorhinal cortex. | Hetka R et al. | β | 1999 | β |
| Seizure disorders in mutant mice: relevance to human epilepsies. | Puranam RS et al. | β | 1999 | β |
| Single gene defects in mice: the role of voltage-dependent calcium channels in absence models. | Burgess DL et al. | β | 1999 | β |
| The impact of genomics on mammalian neurobiology. | Hochgeschwender U et al. | β | 1999 | β |
| The inwardly rectifying K(+) channel subunit GIRK1 rescues the GIRK2 weaver phenotype. | Hou P et al. | β | 1999 | β |
| The weaver GIRK2 mutation leads to decreased levels of serum thyroid hormone: characterization of the effect on midbrain dopaminergic neuron survival. | Blum M et al. | β | 1999 | β |
| The weaver mouse gain-of-function phenotype of dopaminergic midbrain neurons is determined by coactivation of wvGirk2 and K-ATP channels. | Liss B et al. | β | 1999 | β |
| Tottering mouse motor dysfunction is abolished on the Purkinje cell degeneration (pcd) mutant background. | Campbell DB et al. | β | 1999 | β |
| Vermectomy enhances parvalbumin expression and improves motor performance in weaver mutant mice: an animal model for cerebellar ataxia. | GrΓΌsser-Cornehls U et al. | β | 1999 | β |
| Voltage-gated ion channels and hereditary disease. | Lehmann-Horn F et al. | β | 1999 | β |
| Weaver cerebellar granule neurons show altered expression of NMDA receptor subunits both in vivo and in vitro. | Liesi P et al. | β | 1999 | β |
| 10 years of Genomics, chromosome 21, and Down syndrome. | Antonarakis SE | β | 1998 | β |
| A novel potassium channel gene, KCNQ2, is mutated in an inherited epilepsy of newborns. | Singh NA et al. | β | 1998 | β |
| Apoptotic proteins Reaper and Grim induce stable inactivation in voltage-gated K+ channels. | Avdonin V et al. | β | 1998 | β |
| A second mammalian antizyme: conservation of programmed ribosomal frameshifting. | Ivanov IP et al. | β | 1998 | β |
| Ataxia, arrhythmia and ion-channel gene defects. | Doyle JL et al. | β | 1998 | β |
| ATP-sensitive potassium channels: structures, functions, and pathophysiology. | Inagaki N et al. | β | 1998 | β |
| Cell death during development of testis and cerebellum in the mutant mouse weaver. | Harrison SM et al. | β | 1998 | β |
| Characterization of murine Girk2 transcript isoforms: structure and differential expression. | Wei J et al. | β | 1998 | β |
| Current status of genetic discoveries in migraine: familial hemiplegic migraine and beyond. | Gardner K et al. | β | 1998 | β |
| Defective potassium currents in ataxia telangiectasia fibroblasts. | Rhodes N et al. | β | 1998 | β |
| Endomorphins fully activate a cloned human mu opioid receptor. | Gong J et al. | β | 1998 | β |
| Epilepsy genetics: an abundance of riches for biologists. | McNamara JO et al. | β | 1998 | β |
| Expression of Kv1.1, a Shaker-like potassium channel, is temporally regulated in embryonic neurons and glia. | Hallows JL et al. | β | 1998 | β |
| Expression of neurotrophins and neurotrophin receptors in the cerebellum of mutant weaver and lurcher mice. | WΓΌllner U et al. | β | 1998 | β |
| Expression of two inward rectifier potassium channels is essential for differentiation of primitive human hematopoietic progenitor cells. | Shirihai O et al. | β | 1998 | β |
| Expression pattern and neurotrophic role of the c-fms proto-oncogene M-CSF receptor in rodent Purkinje cells. | Murase S et al. | β | 1998 | β |
| Genetic aspects of Parkinson's disease. | Bandmann O et al. | β | 1998 | β |
| Girk2 expression in the ventral midbrain, cerebellum, and olfactory bulb and its relationship to the murine mutation weaver. | Schein JC et al. | β | 1998 | β |
| Higher seizure susceptibility and enhanced tyrosine phosphorylation of N-methyl-D-aspartate receptor subunit 2B in fyn transgenic mice. | Kojima N et al. | β | 1998 | β |
| Ionotropic glutamate receptors in the retina: moving from molecules to circuits. | Lo W et al. | β | 1998 | β |
| New directions for neuronal migration. | Pearlman AL et al. | β | 1998 | β |
| Orchestration of neuronal migration by activity of ion channels, neurotransmitter receptors, and intracellular Ca2+ fluctuations. | Komuro H et al. | β | 1998 | β |
| Pore mutation in a G-protein-gated inwardly rectifying K+ channel subunit causes loss of K+-dependent inhibition in weaver hippocampus. | Jarolimek W et al. | β | 1998 | β |
| Potassium ion channels and human disease: phenotypes to drug targets? | Curran ME | β | 1998 | β |
| Quantitative analysis of cerebellar lobulation in normal and agranular rats. | Doughty ML et al. | β | 1998 | β |
| The nematode degenerin UNC-105 forms ion channels that are activated by degeneration- or hypercontraction-causing mutations. | GarcΓa-AΓ±overos J et al. | β | 1998 | β |
| The weaver mutation causes a loss of inward rectifier current regulation in premigratory granule cells of the mouse cerebellum. | Rossi P et al. | β | 1998 | β |
| Transcriptional map of the 2.5-Mb CBR-ERG region of chromosome 21 involved in Down syndrome. | Dahmane N et al. | β | 1998 | β |
| Using knockout and transgenic mice to study neurophysiology and behavior. | Picciotto MR et al. | β | 1998 | β |
| Abnormalities of pancreatic islets by targeted expression of a dominant-negative KATP channel. | Miki T et al. | β | 1997 | β |
| Activation of heteromeric G protein-gated inward rectifier K+ channels overexpressed by adenovirus gene transfer inhibits the excitability of hippocampal neurons. | Ehrengruber MU et al. | β | 1997 | β |
| Altered intracellular localization of the glutamate receptor channel delta 2 subunit in weaver and reeler Purkinje cells. | Takayama C et al. | β | 1997 | β |
| A new inward rectifier potassium channel gene (KCNJ15) localized on chromosome 21 in the Down syndrome chromosome region 1 (DCR1). | Gosset P et al. | β | 1997 | β |
| An extracellular proteolytic cascade promotes neuronal degeneration in the mouse hippocampus. | Tsirka SE et al. | β | 1997 | β |
| An immunocytochemical study on the distribution of two G-protein-gated inward rectifier potassium channels (GIRK2 and GIRK4) in the adult rat brain. | Murer G et al. | β | 1997 | β |
| Atrophy and loss of dopaminergic mesencephalic neurons in heterozygous weaver mice. | Verina T et al. | β | 1997 | β |
| BAPTA-AM and ethanol protect cerebellar granule neurons from the destructive effect of the weaver gene. | Liesi P et al. | β | 1997 | β |
| Behavioral defects in C. elegans egl-36 mutants result from potassium channels shifted in voltage-dependence of activation. | Johnstone DB et al. | β | 1997 | β |
| Binding of the G protein betagamma subunit to multiple regions of G protein-gated inward-rectifying K+ channels. | Huang CL et al. | β | 1997 | β |
| BNaC1 and BNaC2 constitute a new family of human neuronal sodium channels related to degenerins and epithelial sodium channels. | GarcΓa-AΓ±overos J et al. | β | 1997 | β |
| Calcium channels prove to be a real headache. | Miller RJ | β | 1997 | β |
| Cardiac potassium channels in health and disease. | Brown AM | β | 1997 | β |
| Cloned potassium channels from eukaryotes and prokaryotes. | Jan LY et al. | β | 1997 | β |
| Cosmid contig and transcriptional map of three regions of human chromosome 21q22: identification of 37 novel transcripts by direct selection. | Guimera J et al. | β | 1997 | β |
| Defective gamma-aminobutyric acid type B receptor-activated inwardly rectifying K+ currents in cerebellar granule cells isolated from weaver and Girk2 null mutant mice. | Slesinger PA et al. | β | 1997 | β |
| Developmental expression of the GIRK family of inward rectifying potassium channels: implications for abnormalities in the weaver mutant mouse. | Chen SC et al. | β | 1997 | β |
| Diverse cell death pathways result from a single missense mutation in weaver mouse. | Migheli A et al. | β | 1997 | β |
| Expression of Tiam-1 in the developing brain suggests a role for the Tiam-1-Rac signaling pathway in cell migration and neurite outgrowth. | Ehler E et al. | β | 1997 | β |
| Failed cell migration and death of purkinje cells and deep nuclear neurons in the weaver cerebellum. | Maricich SM et al. | β | 1997 | β |
| Functional screening of 2 Mb of human chromosome 21q22.2 in transgenic mice implicates minibrain in learning defects associated with Down syndrome. | Smith DJ et al. | β | 1997 | β |
| Gene identification in 1.6-Mb region of the Down syndrome region on chromosome 21. | Ohira M et al. | β | 1997 | β |
| Genes and parkinsonism. | Wood N | β | 1997 | β |
| Genetic and physical maps of the stargazer locus on mouse chromosome 15. | Letts VA et al. | β | 1997 | β |
| G protein beta gamma subunits. | Clapham DE et al. | β | 1997 | β |
| High-resolution recombinational map of mouse chromosome 16. | Reeves RH et al. | β | 1997 | β |
| Identification and preclinical testing of novel antiepileptic compounds. | Meldrum BS | β | 1997 | β |
| Identification of caveolin and caveolin-related proteins in the brain. | Cameron PL et al. | β | 1997 | β |
| Identification of regions that regulate the expression and activity of G protein-gated inward rectifier K+ channels in Xenopus oocytes. | Stevens EB et al. | β | 1997 | β |
| Increased excitability and inward rectification in layer V cortical pyramidal neurons in the epileptic mutant mouse Stargazer. | Di Pasquale E et al. | β | 1997 | β |
| Influence of flupirtine on a G-protein coupled inwardly rectifying potassium current in hippocampal neurones. | Jakob R et al. | β | 1997 | β |
| Ion channel mutations in mouse models of inherited neurological disease. | Meisler MH et al. | β | 1997 | β |
| Mapping of a second locus for familial hemiplegic migraine to 1q21-q23 and evidence of further heterogeneity. | Ducros A et al. | β | 1997 | β |
| meander tail acts intrinsic to granule cell precursors to disrupt cerebellar development: analysis of meander tail chimeric mice. | Hamre KM et al. | β | 1997 | β |
| Mutations in the Cacnl1a4 calcium channel gene are associated with seizures, cerebellar degeneration, and ataxia in tottering and leaner mutant mice. | Doyle J et al. | β | 1997 | β |
| Neuregulin and erbB receptors play a critical role in neuronal migration. | Rio C et al. | β | 1997 | β |
| Neurodegeneration in Lurcher mice caused by mutation in delta2 glutamate receptor gene. | Zuo J et al. | β | 1997 | β |
| Neuropathology of degenerative cell death in Caenorhabditis elegans. | Hall DH et al. | β | 1997 | β |
| New modulatory alpha subunits for mammalian Shab K+ channels. | Salinas M et al. | β | 1997 | β |
| Normal cerebellar development but susceptibility to seizures in mice lacking G protein-coupled, inwardly rectifying K+ channel GIRK2. | Signorini S et al. | β | 1997 | β |
| Ontogeny of gene expression of Kir channel subunits in the rat. | Karschin C et al. | β | 1997 | β |
| Partial structure, chromosome localization, and expression of the mouse Girk4 gene. | Wickman K et al. | β | 1997 | β |
| Pharmacology of neurotrophic factors. | Hefti F | β | 1997 | β |
| Potassium channelopathies. | Sanguinetti MC et al. | β | 1997 | β |
| Properties of KvLQT1 K+ channel mutations in Romano-Ward and Jervell and Lange-Nielsen inherited cardiac arrhythmias. | Chouabe C et al. | β | 1997 | β |
| Removal of tissue plasminogen activator does not protect against neuronal degeneration in the cerebellum of the weaver mouse. | Mecenas PE et al. | β | 1997 | β |
| Sodium/hydrogen exchanger gene defect in slow-wave epilepsy mutant mice. | Cox GA et al. | β | 1997 | β |
| Spinal muscular atrophy gene wobbler of the mouse: evidence from chimeric spinal cord and testis for cell-autonomous function. | Augustin M et al. | β | 1997 | β |
| The contribution of the mouse to advances in human genetics. | Fisher EM | β | 1997 | β |
| The murine Dyrk protein maps to chromosome 16, localizes to the nucleus, and can form multimers. | Song WJ et al. | β | 1997 | β |
| Voltage-gated and inwardly rectifying potassium channels. | Jan LY et al. | β | 1997 | β |
| Vulnerability of midbrain dopaminergic neurons in calbindin-D28k-deficient mice: lack of evidence for a neuroprotective role of endogenous calbindin in MPTP-treated and weaver mice. | Airaksinen MS et al. | β | 1997 | β |
| Weaver mutant mouse cerebellar granule cells respond normally to chronic depolarization. | Bjerregaard A et al. | β | 1997 | β |
| A high-density molecular genetic map around the weaver locus. | Millonig JH et al. | β | 1996 | β |
| A missense mutation in the sodium channel Scn8a is responsible for cerebellar ataxia in the mouse mutant jolting. | Kohrman DC et al. | β | 1996 | β |
| A regenerative link in the ionic fluxes through the weaver potassium channel underlies the pathophysiology of the mutation. | Silverman SK et al. | β | 1996 | β |
| Cerebellar granule cell differentiation in mutant and X-irradiated rodents revealed by the neural adhesion molecule TAG-1. | Bailly Y et al. | β | 1996 | β |
| Conditional ablation of cerebellar astrocytes in postnatal transgenic mice. | Delaney CL et al. | β | 1996 | β |
| Expression pattern of integrin beta 1 subunit in Purkinje cells of rat and cerebellar mutant mice. | Murase S et al. | β | 1996 | β |
| Functional analysis of the weaver mutant GIRK2 K+ channel and rescue of weaver granule cells. | Kofuji P et al. | β | 1996 | β |
| Functional effects of the mouse weaver mutation on G protein-gated inwardly rectifying K+ channels. | Slesinger PA et al. | β | 1996 | β |
| Heteromeric channel formation and Ca(2+)-free media reduce the toxic effect of the weaver Kir 3.2 allele. | Tucker SJ et al. | β | 1996 | β |
| Heteromultimerization of G-protein-gated inwardly rectifying K+ channel proteins GIRK1 and GIRK2 and their altered expression in weaver brain. | Liao YJ et al. | β | 1996 | β |
| Identification of the weaver mouse mutation: the end of the beginning. | Hess EJ | β | 1996 | β |
| IRK(1-3) and GIRK(1-4) inwardly rectifying K+ channel mRNAs are differentially expressed in the adult rat brain. | Karschin C et al. | β | 1996 | β |
| Localization and interaction of epitope-tagged GIRK1 and CIR inward rectifier K+ channel subunits. | Kennedy ME et al. | β | 1996 | β |
| Neuron death in the substantia nigra of weaver mouse occurs late in development and is not apoptotic. | Oo TF et al. | β | 1996 | β |
| Nonselective and G betagamma-insensitive weaver K+ channels. | Navarro B et al. | β | 1996 | β |
| Pathophysiology of ion channel mutations. | Keating MT et al. | β | 1996 | β |
| Phenotypic effects of the weaver gene are evident in the embryonic cerebellum but not in the ventral midbrain. | Bayer SA et al. | β | 1996 | β |
| Targeting epilepsy genes. | Noebels JL | β | 1996 | β |
| The human homologue of the weaver mouse gene in familial and sporadic Parkinson's disease. | Bandmann O et al. | β | 1996 | β |
| The weaver mouse: a most cantankerous rodent. | Herrup K | β | 1996 | β |
| The weaver mutation changes the ion selectivity of the affected inwardly rectifying potassium channel GIRK2. | Tong Y et al. | β | 1996 | β |
| The weaver mutation of GIRK2 results in a loss of inwardly rectifying K+ current in cerebellar granule cells. | Surmeier DJ et al. | β | 1996 | β |
| Weaver granule neurons are rescued by calcium channel antagonists and antibodies against a neurite outgrowth domain of the B2 chain of laminin. | Liesi P et al. | β | 1996 | β |
| The usual suspects: GABA and glutamate may regulate proliferation in the neocortex. | LaMantia AS | β | 1995 | β |
| Tune into the weaver channel. | Goldowitz D et al. | β | 1995 | β |