Affinage

DNM1

Dynamin-1 · UniProt Q05193

Round 2 corrected
Length
864 aa
Mass
97.4 kDa
Annotated
2026-04-28
97 papers in source corpus 31 papers cited in narrative 31 extracted findings

Mechanistic narrative

Synthesis pass · prose summary of the discoveries below

DNM1 encodes dynamin-1, a large mechanochemical GTPase that self-assembles into helical oligomers around membrane necks and uses GTP hydrolysis to generate constriction and longitudinal tension, driving membrane fission in both clathrin-mediated synaptic vesicle endocytosis and mitochondrial division (PMID:2144893, PMID:16648839, PMID:21170049). In neurons, dephosphorylation-dependent assembly with amphiphysin, synaptojanin, and endophilins couples dynamin-1 to the presynaptic vesicle recycling machinery, while in mitochondria the Insert B domain mediates recruitment via Fis1–Mdv1 adaptors, and completion of fission requires cooperation with the inner-membrane factor Atg44 (PMID:9694653, PMID:23148233, PMID:38818923). De novo dominant-negative mutations clustering in the GTPase and middle domains cause epileptic encephalopathy by impairing oligomerization-dependent vesicle scission at synapses, as demonstrated by knock-in mouse models exhibiting seizures, disrupted neurotransmission, and pharmacological or gene-therapy rescue (PMID:27066543, PMID:37648685, PMID:39127888).

Mechanistic history

Synthesis pass · year-by-year structured walk · 11 steps
  1. 1990 High

    Molecular cloning of dynamin-1 established it as a novel GTPase and microtubule-associated mechanochemical enzyme, founding a new GTPase superfamily distinct from small G proteins.

    Evidence cDNA cloning and sequence analysis from rat brain

    PMID:2144893

    Open questions at the time
    • GTPase activity was inferred from sequence; no catalytic rate measurement
    • cellular substrate (membrane vs. microtubule) was unresolved
  2. 1993 High

    GTPase domain mutations blocking receptor-mediated endocytosis at the coated-pit stage demonstrated that dynamin-1 GTPase activity is essential for clathrin-coated vesicle formation, resolving its primary cellular function.

    Evidence Transient expression of GTP-binding domain mutants; endocytosis intermediate assays in mammalian cells

    PMID:8101525

    Open questions at the time
    • Whether dynamin acts catalytically or purely mechanically was unclear
    • the fission step itself had not been directly visualized
  3. 1997 High

    Identification of endophilin SH3 domain interactions with dynamin-1's proline-rich domain placed dynamin-1 within a presynaptic protein network dedicated to synaptic vesicle recycling.

    Evidence Yeast two-hybrid; co-immunoprecipitation from brain extracts

    PMID:9238017

    Open questions at the time
    • Functional consequence of endophilin binding on fission was not tested
    • stoichiometry and order of partner recruitment were unknown
  4. 1998 High

    Demonstrating that dephosphorylation triggers dynamin-1 assembly with amphiphysin, synaptojanin, clathrin, and AP-2 revealed a phosphorylation cycle as the master switch governing endocytic machinery assembly at nerve terminals.

    Evidence In vitro dephosphorylation of rat brain extract; co-immunoprecipitation and complex assembly assays

    PMID:9694653

    Open questions at the time
    • Identity of the kinase and phosphatase controlling this cycle in vivo were not fully defined
    • whether phosphorylation also regulates mitochondrial roles was unknown
  5. 2001 High

    Two parallel advances established that (1) GTP hydrolysis, not just GTP binding or oligomerization, is the catalytic requirement for fission, and (2) the yeast ortholog Dnm1 has a separate role in mitochondrial division via the adaptor Mdv1/Net2 at outer membrane constriction sites.

    Evidence GED mutagenesis with endocytosis assays (mammalian cells); yeast two-hybrid, gene disruption, and immunoelectron microscopy (yeast mitochondria)

    PMID:11179417 PMID:11242086

    Open questions at the time
    • Physical mechanism by which hydrolysis generates force was unknown
    • how Mdv1 scaffolds Dnm1 assembly was unresolved
  6. 2006 High

    Real-time visualization of dynamin-coated lipid tubules showed that GTP hydrolysis drives twisting and longitudinal tension leading to tubule breakage, establishing the mechanoenzyme fission mechanism; concurrently, biochemical studies defined the dimer as the minimum cytoplasmic assembly unit and showed that higher-order multimerization is required to form functional fission complexes via Mdv1–Fis1.

    Evidence Lipid tubule twisting/bead rotation assays (reconstituted); in vivo fluorescence and biochemical oligomerization assays (yeast)

    PMID:16601120 PMID:16648839

    Open questions at the time
    • Inner membrane scission mechanism was unaddressed
    • whether constriction magnitude differs between endocytic and mitochondrial contexts was unclear
  7. 2010 High

    Cryo-EM structure of GTP-bound Dnm1 revealed a unique helical assembly that constricts liposomes to a greater extent than mammalian dynamin before dissociating, providing a structural basis for the mitochondrial fission mechanism.

    Evidence Cryo-EM 3D reconstruction; liposome constriction assay with yeast Dnm1

    PMID:21170049

    Open questions at the time
    • Atomic-resolution structure was lacking
    • how constriction is coupled to inner-membrane remodeling was unknown
  8. 2012 High

    Mapping of a conserved motif within the Insert B domain as the Mdv1-binding determinant, validated by suppressor mutations in Mdv1's β-propeller, defined the molecular interface governing Dnm1 recruitment to mitochondria.

    Evidence Site-directed mutagenesis; suppressor genetics; co-immunoprecipitation; fluorescence microscopy (yeast)

    PMID:23148233

    Open questions at the time
    • Structural detail of the Insert B–β-propeller interface was not resolved
    • whether mammalian adaptors use the same interface was untested
  9. 2015 High

    Functional characterization of epileptic encephalopathy mutations (A177P, K206N, G359A) showed dominant-negative impairment of vesicle scission and oligomerization, linking DNM1 loss-of-function directly to human disease pathogenesis.

    Evidence Transferrin endocytosis assays; electron microscopy of transfected cells; Western blotting

    PMID:27066543

    Open questions at the time
    • In vivo neuronal consequences in animal models were not yet shown
    • genotype–phenotype correlations across variant spectrum were incomplete
  10. 2023 High

    A heterozygous R237W knock-in mouse recapitulated human epileptic encephalopathy with disrupted GTPase activity, dysfunctional endocytosis, and seizures, and pharmacological acceleration of endocytosis rescued these phenotypes from cellular to in vivo scales, establishing proof-of-concept for therapeutic intervention.

    Evidence Knock-in mouse; electrophysiology; neuronal endocytosis assays; BMS-204352 pharmacological rescue

    PMID:37648685

    Open questions at the time
    • Long-term efficacy and safety of pharmacological approach were not assessed
    • whether drug rescues mitochondrial fission defects was untested
  11. 2024 High

    Two breakthroughs: (1) Atg44 was identified as the inner-membrane partner required for Dnm1 to complete mitochondrial fission, resolving the long-standing question of how outer and inner membranes are coordinately severed; (2) AAV9 knockdown-replace gene therapy rescued lethal seizures and corrected transcriptomic and synaptic transmission abnormalities in dominant-negative DNM1 encephalopathy mice.

    Evidence CLEM and genetic deletion (yeast); AAV9 RNAi-replace with RNA-seq and electrophysiology (mouse)

    PMID:38818923 PMID:39127888

    Open questions at the time
    • Molecular mechanism by which Atg44 acts on inner membrane is unknown
    • gene therapy durability and human translation remain to be evaluated

Open questions

Synthesis pass · forward-looking unresolved questions
  • Key unresolved questions include the atomic-resolution structure of assembled Dnm1 during active membrane scission, the identity of mammalian mitochondrial adaptors functionally equivalent to yeast Mdv1, and the mechanism by which phosphorylation integrates endocytic and mitochondrial fission activities of dynamin-1 in neurons.
  • No high-resolution cryo-EM structure of Dnm1 actively constricting a membrane
  • mammalian Mdv1 equivalent not identified
  • phosphorylation-dependent regulation of mitochondrial fission role unexplored

Mechanism profile

Synthesis pass · controlled-vocabulary classification · explore literature graph →
Molecular activity
GO:0003924 GTPase activity 6 GO:0008092 cytoskeletal protein binding 2 GO:0140657 ATP-dependent activity 2
Localization
GO:0005739 mitochondrion 10 GO:0005886 plasma membrane 4 GO:0031410 cytoplasmic vesicle 3 GO:0005829 cytosol 1
Pathway
R-HSA-1852241 Organelle biogenesis and maintenance 10 R-HSA-112316 Neuronal System 6 R-HSA-1643685 Disease 6 R-HSA-5653656 Vesicle-mediated transport 6
Partners
AMPHATG44FIS1MDA1MDV1PFN1SH3GL2SYNJ1
Complex memberships
Dnm1–Mdv1–Fis1 mitochondrial fission complexDynamin-1 oligomeric ring/helix

Evidence

Reading pass · 31 per-paper findings extracted from the source corpus
Year Finding Method Journal Conf PMIDs
1990 Dynamin (DNM1) was molecularly cloned as a microtubule-associated mechanochemical enzyme and identified as a GTPase; the predicted amino acid sequence revealed homology with Mx antiviral proteins and the yeast vacuolar protein sorting gene VPS1, establishing a new family of GTPases. cDNA cloning and sequence analysis Nature High 2144893
1993 Mutations in the GTP-binding domain of human dynamin 1 block receptor-mediated endocytosis at a stage after coat assembly initiation but before ligand sequestration into deeply invaginated coated pits, demonstrating that a functional dynamin GTPase is required for clathrin-coated vesicle formation. Transient expression of GTP-binding domain mutants in mammalian cells; endocytosis intermediate assays The Journal of cell biology High 8101525
1995 Yeast Dnm1p (DNM1 ortholog) participates in endosomal trafficking; dnm1 disruption delays receptor-mediated endocytosis after initial internalization but before delivery to the vacuole, indicating Dnm1p acts at a novel endosomal sorting step. Gene disruption; receptor half-life assay; ligand internalization and vacuolar delivery assays The Journal of cell biology High 7622557
1997 The SH3p4/SH3p8/SH3p13 protein family (endophilins) binds to the proline-rich domain of dynamin I via SH3 domain interactions; pools of dynamin I were co-precipitated from brain extracts with antibodies recognizing these proteins, placing dynamin I in a nerve terminal complex involved in synaptic vesicle recycling. Yeast two-hybrid screen; co-immunoprecipitation from brain extracts; immunofluorescence co-localization Proceedings of the National Academy of Sciences of the United States of America High 9238017
1998 Dephosphorylation of dynamin 1 promotes its assembly into complexes with synaptojanin 1 and amphiphysin (also containing clathrin and AP-2); phosphorylation of dynamin 1 inhibits its binding to amphiphysin, demonstrating that phosphorylation/dephosphorylation cycles regulate the assembly of the clathrin-based endocytic machinery at nerve terminals. In vitro dephosphorylation of rat brain extract; co-immunoprecipitation; complex assembly assays Science High 9694653
1998 Profilin I and II associate with dynamin I in mouse brain extracts and co-localize with dynamin I and synapsin in axonal and dendritic processes, linking actin assembly regulation to the endocytic/synaptic vesicle recycling machinery. Affinity pull-down; mass spectrometry; co-immunoprecipitation; immunofluorescence co-localization The EMBO journal Medium 9463375
2000 Dynamin I was identified as a component of NMDA receptor multiprotein complexes (NRC) purified from mouse brain, placing it in the postsynaptic density signaling network alongside kinases, phosphatases, and cytoskeletal proteins. Proteomic characterization by mass spectrometry and immunoblotting of NMDAR complexes isolated from mouse brain Nature neuroscience Medium 10862698
2001 GTP hydrolysis by dynamin and the associated conformational change are required for endocytosis; oligomerization and GTP binding alone are insufficient. Point mutants in the GTPase effector domain (GED) that retain GTP binding but impair hydrolysis block endocytosis in vivo. Site-directed mutagenesis of GED and GTPase domains; endocytosis assays in cells Nature High 11242086
2001 Net2p/Mdv1 is a Dnm1p-interacting protein required for mitochondrial division in yeast; Net2p co-localizes with Dnm1 at constriction sites on mitochondrial tubules, identifying it as a component of the mitochondrial fission machinery. Yeast two-hybrid; gene disruption; immunofluorescence and immunoelectron microscopy co-localization Molecular biology of the cell High 11179417
2005 Mdv1 interacts with assembled Dnm1 on the outer mitochondrial membrane to promote mitochondrial division; structure-function analysis shows dynamic Mdv1–Dnm1 interactions regulate Dnm1 self-assembly, and Mdv1 acts as an adaptor linking Fis1 with Dnm1. Structure-function mutagenesis; co-immunoprecipitation; fluorescence microscopy of mitochondrial dynamics The Journal of biological chemistry High 16272155
2006 The minimum oligomeric form of cytoplasmic Dnm1p is a dimer; dimeric Dnm1G385Dp can be recruited to the outer mitochondrial membrane via interaction with Mdv1p·Fis1p complexes without forming puncta, but multimerization of Dnm1p is subsequently required to reorganize Mdv1p into punctate fission complexes and promote division. In vivo fluorescence microscopy; biochemical oligomerization assays; genetic analysis of assembly mutants The Journal of biological chemistry High 16601120
2006 GTP hydrolysis by dynamin drives twisting of dynamin-coated lipid tubules and supercoiling, generating longitudinal tension that is released by tubule breakage, demonstrating a mechanoenzyme activity in membrane fission; fission requires both constriction and tension. Real-time observation of nucleotide effects on dynamin-coated lipid tubules; bead rotation assays; kinesin-generated tubule fission assay Nature High 16648839
2007 S. cerevisiae Fis1 binds directly to Dnm1 (not through Mdv1 as adaptor); the Fis1 N-terminal arm acts in an autoinhibitory manner reducing Dnm1 binding >100-fold, and the concave surface of the Fis1 TPR-like domain is the evolutionarily conserved site for Dnm1 binding. In vitro binding assays; site-directed mutagenesis of Fis1; quantitative binding measurements The Journal of biological chemistry High 17884824
2007 WD40 protein Mda1 is purified with Dnm1 from mitochondrial division machinery of C. merolae; Mda1 forms a stable homo-oligomeric ring before Dnm1 at division sites, and addition of GTP independently releases Dnm1 from the complex, indicating Dnm1 acts downstream of Mda1 in the division process. Biochemical purification of mitochondrial division machinery; immunofluorescence and immunoelectron microscopy; in vitro GTP-induced disassembly Proceedings of the National Academy of Sciences of the United States of America High 17360593
2008 miR-140* targets the 3'-UTR of Dnm1 mRNA by direct base-pairing; nicotine-increased miR-140* expression represses Dnm1 protein translation and induces Dnm1 mRNA degradation, demonstrating post-transcriptional regulation of dynamin 1 expression via the miRNA pathway. miRNA microarray; luciferase reporter assay; Dnm1 mRNA and protein expression analysis in PC12 cells The international journal of neuropsychopharmacology Medium 18845019
2008 A missense mutation R256L in canine DNM1 causes exercise-induced collapse in Labrador retrievers, the first documented mammalian DNM1 mutation, establishing that DNM1 is essential for normal neuromuscular function and neurotransmission in vivo. Genome-wide linkage mapping; SNP association; haplotype analysis; sequencing Nature genetics Medium 18806795
2009 In fission yeast, Dnm1 mediates mitochondrial fission during interphasic growth and at cell division in a microtubule-dependent manner; Dnm1-YFP localizes to foci at sites of mitochondrial severing between adjacent nucleoids, and dnm1Δ cells show aggregated nucleoids, linking Dnm1 fission activity to mitochondrial DNA organization. Live fluorescence microscopy; microtubule depolymerization; dnm1Δ deletion analysis Cell motility and the cytoskeleton Medium 19373772
2010 Cryo-EM structure of yeast Dnm1 in GTP-bound state reveals a unique helical assembly distinct from dynamin; upon GTP hydrolysis Dnm1 constricts liposomes with a magnitude substantially larger than dynamin and then dissociates, supporting a mechanochemical contractile role in mitochondrial division. Cryo-electron microscopy; 3D reconstruction; liposome constriction assay Nature structural & molecular biology High 21170049
2012 The Insert B domain of yeast Dnm1 contains a novel motif required for association with the mitochondrial adaptor Mdv1; mutation of this conserved motif specifically disrupts Dnm1-Mdv1 interaction, blocking mitochondrial recruitment and fission; suppressor mutations in Mdv1 identify the β-propeller domain as the binding interface. Site-directed mutagenesis; suppressor screen; fluorescence microscopy; co-immunoprecipitation The Journal of cell biology High 23148233
2014 Blm10-proteasomes degrade Dnm1 in yeast; loss of BLM10 impairs Dnm1 degradation in vitro and in vivo, leading to elevated Dnm1 levels, increased mitochondrial fragmentation, reduced respiratory capacity, and increased oxidative damage; constitutive DNM1 overexpression phenocopies blm10Δ cells. In vitro and in vivo protein degradation assays; mitochondrial morphology analysis; respiratory capacity measurement; genetic epistasis The Journal of biological chemistry High 24604417
2015 Epileptic encephalopathy-causing DNM1 mutations (A177P, K206N, G359A) impair synaptic vesicle endocytosis in a dominant-negative manner; K206N decreases protein levels, G359A disrupts higher-order oligomerization, and all three mutations produce vesicle defects visible by electron microscopy consistent with impaired vesicle scission activity. Transferrin endocytosis assay; high-content imaging; Western blotting; electron microscopy of transfected cells and mouse brain Neurology. Genetics High 27066543
2017 DNM1 mutations cluster within the GTPase or middle domains; structural modeling and functional data support a dominant-negative mechanism whereby mutant DNM1 impairs vesicle fission at presynaptic terminals; the recurrent p.Arg237Trp variant is predicted to destabilize the G domain and impair GTPase activity. Biomolecular structural modeling; review of existing functional data; cohort genotype-phenotype analysis Neurology Medium 28667181
2017 Dnm1 synergistically induces negative Gaussian curvature (saddle-shaped curvature) in membranes via a conserved helical domain, capable of generating fission necks of ~12.6 nm diameter; Fis1 inhibits this pro-fission membrane activity of Dnm1, revealing antagonistic roles in regulating mitochondrial fission. Synchrotron small-angle X-ray scattering (SAXS); machine learning-based sequence analysis; in silico mutational analysis ACS central science High 29202017
2019 Association of mitochondria with microtubules physically impedes assembly of Dnm1 around mitochondria, resulting in inhibition of mitochondrial fission; perturbation of microtubule dynamics alters mitochondrial fission frequency and copy number for faithful segregation during mitosis. Deletion of kinesin-like proteins; high-resolution and time-lapse fluorescence microscopy; Dnm1 assembly assays The Journal of biological chemistry High 30602572
2019 Glucose starvation promotes Dnm1 localization to mitochondria and increases mitochondrial fission frequency in fission yeast; low PKA activity enhances glucose starvation-induced Dnm1-dependent fragmentation, while high PKA activity confers resistance, placing Dnm1 downstream of PKA signaling in nutrient-stress mitochondrial remodeling. Live-cell microscopy; genetic manipulation of PKA activity; Dnm1 localization assays; reactive oxygen species measurement The Journal of biological chemistry Medium 31562247
2022 A recurrent de novo splice site variant (c.1197-8G>A) affecting DNM1 exon 10a causes an in-frame insertion of two amino acids that impairs oligomerization-dependent activity through a dominant-negative mechanism; neuropathological samples show accumulation of enlarged synaptic vesicles adherent to the plasma membrane, confirming impaired vesicular fission; the brain-predominant isoform includes exon 10a rather than 10b. RNA sequencing of pediatric brain; functional splice site analysis; neuropathology electron microscopy; structural modeling of oligomerization American journal of human genetics High 36413998
2023 The most prevalent pathogenic DNM1 mutation R237W disrupts dynamin-1 enzyme (GTPase) activity and endocytosis in central neurons; heterozygous R237W knock-in mice display dysfunctional endocytosis, altered excitatory neurotransmission, and seizure-like phenotypes; these phenotypes are corrected at cell, circuit, and in vivo levels by BMS-204352, a drug that accelerates endocytosis. Knock-in mouse model; electrophysiology; endocytosis assays in neurons; in vivo pharmacological rescue Nature communications High 37648685
2024 The mitochondrial intermembrane space protein Atg44 cooperates with Dnm1 to complete mitochondrial fission; Atg44-deficient cells accumulate enlarged mitochondria and rosary-like mitochondria with Dnm1 foci at constriction sites where both outer and inner membranes retain continuity, indicating Dnm1 cannot complete fission without Atg44 acting from inside the mitochondria. Fluorescence microscopy; correlative light and electron microscopy; genetic deletion analysis Autophagy High 38818923
2024 AAV9-based knockdown-replace gene therapy (RNAi against endogenous Dnm1 paired with RNAi-resistant Dnm1 cDNA) rescues lethal seizures and growth delay in a mouse model of dominant-negative DNM1 encephalopathy; mutant mice show abnormal expression of >1,000 genes in synaptic/neuronal clusters that are corrected by treatment; synaptic recordings reveal rectification of inhibitory-to-excitatory transmission alterations. AAV9 gene delivery; RNAi knockdown-replace strategy; RNA sequencing; synaptic electrophysiology; in vivo survival and growth measurement Molecular therapy High 39127888
2025 Dnm1 is required for the focal clustering of Fis1 on the mitochondrial outer membrane; in cells lacking Dnm1, Fis1 distributes diffusely rather than in discrete puncta, revealing that the downstream effector Dnm1 influences the spatial organization of its upstream receptor Fis1. CRISPR-Cas9 endogenous tagging of Fis1 with mNeonGreen; fluorescence microscopy in dnm1Δ yeast microPublication biology Medium 40910107
2026 MDA5 regulates DNM1 expression via an NF-κB-mediated axis; Mda5 knockout B cells show impaired actin polymerization and BCR signaling that can be rescued by the DNM1 activator Bis-T-23, placing DNM1 downstream of MDA5-NF-κB in governing cytoskeletal dynamics in B cells. Mda5 knockout mice; Bis-T-23 pharmacological activation of DNM1; actin polymerization assays; BCR signaling readouts Cellular & molecular immunology Medium 41476189

Source papers

Stage 0 corpus · 97 papers · ranked by NIH iCite citations
Year Title Journal Citations PMID
2005 A human protein-protein interaction network: a resource for annotating the proteome. Cell 1704 16169070
2002 Generation and initial analysis of more than 15,000 full-length human and mouse cDNA sequences. Proceedings of the National Academy of Sciences of the United States of America 1479 12477932
2015 The BioPlex Network: A Systematic Exploration of the Human Interactome. Cell 1118 26186194
2017 Architecture of the human interactome defines protein communities and disease networks. Nature 1085 28514442
2000 Proteomic analysis of NMDA receptor-adhesion protein signaling complexes. Nature neuroscience 977 10862698
2012 The mRNA-bound proteome and its global occupancy profile on protein-coding transcripts. Molecular cell 973 22681889
2004 Immunoaffinity profiling of tyrosine phosphorylation in cancer cells. Nature biotechnology 916 15592455
2003 Complete sequencing and characterization of 21,243 full-length human cDNAs. Nature genetics 754 14702039
2021 Dual proteome-scale networks reveal cell-specific remodeling of the human interactome. Cell 705 33961781
2011 Phylogenetic-based propagation of functional annotations within the Gene Ontology consortium. Briefings in bioinformatics 656 21873635
1993 Mutations in human dynamin block an intermediate stage in coated vesicle formation. The Journal of cell biology 634 8101525
2011 Mapping the NPHP-JBTS-MKS protein network reveals ciliopathy disease genes and pathways. Cell 507 21565611
1994 Oligo-capping: a simple method to replace the cap structure of eukaryotic mRNAs with oligoribonucleotides. Gene 492 8125298
2022 OpenCell: Endogenous tagging for the cartography of human cellular organization. Science (New York, N.Y.) 432 35271311
2005 Diversification of transcriptional modulation: large-scale identification and characterization of putative alternative promoters of human genes. Genome research 409 16344560
2015 Panorama of ancient metazoan macromolecular complexes. Nature 407 26344197
2001 GTPase activity of dynamin and resulting conformation change are essential for endocytosis. Nature 384 11242086
2006 GTP-dependent twisting of dynamin implicates constriction and tension in membrane fission. Nature 380 16648839
2002 The endophilin-CIN85-Cbl complex mediates ligand-dependent downregulation of c-Met. Nature 371 11894096
2010 FCHo proteins are nucleators of clathrin-mediated endocytosis. Science (New York, N.Y.) 361 20448150
2014 De novo mutations in synaptic transmission genes including DNM1 cause epileptic encephalopathies. American journal of human genetics 360 25262651
2007 Functional specialization of beta-arrestin interactions revealed by proteomic analysis. Proceedings of the National Academy of Sciences of the United States of America 360 17620599
2010 Conformational changes in Dnm1 support a contractile mechanism for mitochondrial fission. Nature structural & molecular biology 345 21170049
1997 The SH3p4/Sh3p8/SH3p13 protein family: binding partners for synaptojanin and dynamin via a Grb2-like Src homology 3 domain. Proceedings of the National Academy of Sciences of the United States of America 335 9238017
1990 Molecular cloning of the microtubule-associated mechanochemical enzyme dynamin reveals homology with a new family of GTP-binding proteins. Nature 329 2144893
2007 Huntingtin interacting proteins are genetic modifiers of neurodegeneration. PLoS genetics 325 17500595
1998 In mouse brain profilin I and profilin II associate with regulators of the endocytic pathway and actin assembly. The EMBO journal 296 9463375
2008 Molecular genetics of adult ADHD: converging evidence from genome-wide association and extended pedigree linkage studies. Journal of neural transmission (Vienna, Austria : 1996) 295 18839057
2020 Phosphorylated tau interactome in the human Alzheimer's disease brain. Brain : a journal of neurology 290 32812023
2001 The SH2/SH3 adaptor Grb4 transduces B-ephrin reverse signals. Nature 281 11557983
1998 Role of phosphorylation in regulation of the assembly of endocytic coat complexes. Science (New York, N.Y.) 280 9694653
2012 A high-throughput approach for measuring temporal changes in the interactome. Nature methods 273 22863883
2001 Division of mitochondria requires a novel DNM1-interacting protein, Net2p. Molecular biology of the cell 130 11179417
2005 Mdv1 interacts with assembled dnm1 to promote mitochondrial division. The Journal of biological chemistry 126 16272155
1995 DNM1, a dynamin-related gene, participates in endosomal trafficking in yeast. The Journal of cell biology 115 7622557
2017 DNM1 encephalopathy: A new disease of vesicle fission. Neurology 90 28667181
2008 Nicotine modulates expression of miR-140*, which targets the 3'-untranslated region of dynamin 1 gene (Dnm1). The international journal of neuropsychopharmacology 76 18845019
2007 Direct binding of the dynamin-like GTPase, Dnm1, to mitochondrial dynamics protein Fis1 is negatively regulated by the Fis1 N-terminal arm. The Journal of biological chemistry 54 17884824
2006 Dimeric Dnm1-G385D interacts with Mdv1 on mitochondria and can be stimulated to assemble into fission complexes containing Mdv1 and Fis1. The Journal of biological chemistry 53 16601120
2015 Epileptic encephalopathy-causing mutations in DNM1 impair synaptic vesicle endocytosis. Neurology. Genetics 46 27066543
2008 A canine DNM1 mutation is highly associated with the syndrome of exercise-induced collapse. Nature genetics 45 18806795
2019 Association of mitochondria with microtubules inhibits mitochondrial fission by precluding assembly of the fission protein Dnm1. The Journal of biological chemistry 43 30602572
2009 The dynamin related protein Dnm1 fragments mitochondria in a microtubule-dependent manner during the fission yeast cell cycle. Cell motility and the cytoskeleton 34 19373772
2017 Molecular Motor Dnm1 Synergistically Induces Membrane Curvature To Facilitate Mitochondrial Fission. ACS central science 33 29202017
2014 Proteasomes associated with the Blm10 activator protein antagonize mitochondrial fission through degradation of the fission protein Dnm1. The Journal of biological chemistry 31 24604417
2015 De novo DNM1 mutations in two cases of epileptic encephalopathy. Epilepsia 30 26611353
2022 A recurrent de novo splice site variant involving DNM1 exon 10a causes developmental and epileptic encephalopathy through a dominant-negative mechanism. American journal of human genetics 27 36413998
2019 Glucose starvation induces mitochondrial fragmentation depending on the dynamin GTPase Dnm1/Drp1 in fission yeast. The Journal of biological chemistry 27 31562247
2012 A novel motif in the yeast mitochondrial dynamin Dnm1 is essential for adaptor binding and membrane recruitment. The Journal of cell biology 27 23148233
2007 WD40 protein Mda1 is purified with Dnm1 and forms a dividing ring for mitochondria before Dnm1 in Cyanidioschyzon merolae. Proceedings of the National Academy of Sciences of the United States of America 27 17360593
2009 The a2 mating-type-locus gene lga2 of Ustilago maydis interferes with mitochondrial dynamics and fusion, partially in dependence on a Dnm1-like fission component. Journal of cell science 26 19531588
2018 Mutations in the PH Domain of DNM1 are associated with a nonepileptic phenotype characterized by developmental delay and neurobehavioral abnormalities. Molecular genetics & genomic medicine 24 29397573
1994 Introduction of oxygen into the alkyl chain of N-decyl-dNM decreases lipophilicity and results in increased retention of glucose residues on N-linked oligosaccharides. Glycobiology 20 8054714
2016 A dividing matter: Drp1/Dnm1-independent mitophagy. The Journal of cell biology 16 27903605
2012 The mitochondrial Dnm1-like fission component is required for lgA2-induced mitophagy but dispensable for starvation-induced mitophagy in Ustilago maydis. Eukaryotic cell 16 22843561
2024 Effective knockdown-replace gene therapy in a novel mouse model of DNM1 developmental and epileptic encephalopathy. Molecular therapy : the journal of the American Society of Gene Therapy 15 39127888
2021 Loss-of-function variants in DNM1 cause a specific form of developmental and epileptic encephalopathy only in biallelic state. Journal of medical genetics 15 34172529
2019 Proteomics Analysis of Candida albicans dnm1 Haploid Mutant Unraveled the Association between Mitochondrial Fission and Antifungal Susceptibility. Proteomics 14 31811746
2011 Presence and impact of the exercise-induced collapse associated DNM1 mutation in Labrador retrievers and other breeds. Veterinary journal (London, England : 1997) 13 21782486
2023 Reversal of cell, circuit and seizure phenotypes in a mouse model of DNM1 epileptic encephalopathy. Nature communications 12 37648685
2020 DNM1, a Dynamin-Related Protein That Contributes to Endocytosis and Peroxisome Fission, Is Required for the Vegetative Growth, Sporulation, and Virulence of Metarhizium robertsii. Applied and environmental microbiology 12 32631867
2018 Core/Shell Structured Fe₃O₄@TiO₂-DNM Nanospheres as Multifunctional Anticancer Platform: Chemotherapy and Photodynamic Therapy Research. Journal of nanoscience and nanotechnology 12 29442618
2014 Classical dynamin DNM1 and DNM3 genes attain maximum expression in the normal human central nervous system. BMC research notes 11 24673776
2024 Atg44/Mdi1/mitofissin facilitates Dnm1-mediated mitochondrial fission. Autophagy 10 38818923
2022 Clinical, Radiological, and Genetic Characterization of a Patient with a Novel Homoallelic Loss-of-Function Variant in DNM1. Genes 8 36553519
2021 Mimicking human Drp1 disease-causing mutations in yeast Dnm1 reveals altered mitochondrial dynamics. Mitochondrion 8 34157431
2023 DNA Nanomachine (DNM) Biplex Assay for Differentiating Bacillus cereus Species. International journal of molecular sciences 7 36901903
2021 dnm1 deletion blocks mitochondrial fragmentation in Δfzo1 cells. Yeast (Chichester, England) 7 33125774
2021 Altered Fast Synaptic Transmission in a Mouse Model of DNM1-Associated Developmental Epileptic Encephalopathy. eNeuro 7 33372033
2021 Pathogenic DNM1 Gene Variant Presenting With Unusually Nonsevere Neurodevelopmental Phenotype: A Case Report. Neurology. Genetics 7 34386584
2018 Ramalin, an antioxidant compound derived from Antarctic lichen, prevents progression of liver fibrosis induced by dimethylnitrosamine (DNM) in rats. Biochemical and biophysical research communications 7 30172374
2018 DNM1 Mutation in a child associated with progressive bilateral mesial temporal sclerosis. Clinical case reports 7 30455886
2017 DNM1 mutation status, sex, and sterilization status of a cohort of Labrador retrievers with and without cranial cruciate ligament rupture. Canine genetics and epidemiology 7 28168039
2012 Mitochondrial fission proteins Fis1 and Mdv1, but not Dnm1, play a role in maintenance of heteroplasmy in budding yeast. FEBS letters 6 22575664
2023 Genotypes and phenotypes of DNM1 encephalopathy. Journal of medical genetics 5 37248033
1997 Assignment of the dynamin-1 gene (DNM1) to human chromosome 9q34 by fluorescence in situ hybridization and somatic cell hybrid analysis. Genomics 5 9143509
2025 Expanding the phenotypic spectrum of DNM1-related disorders: novel GTPase domain variants and their diverse neurological outcomes. Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology 3 39954101
2023 A deep intronic variant in DNM1 in a patient with developmental and epileptic encephalopathy creates a splice acceptor site and affects only transcript variants including exon 10a. Neurogenetics 3 37039969
2023 Autism and mild epilepsy associated with a de novo missense pathogenic variant in the GTPase effector domain of DNM1. American journal of medical genetics. Part C, Seminars in medical genetics 3 37132416
2023 Truncated DNM1 variant underlines developmental delay and epileptic encephalopathy. Frontiers in pediatrics 3 37900685
2024 Study on the mechanism and degradation behavior of Encifer adhaerens DNM-S1 capturing dimethyl phthalate. Chemosphere 2 38641291
2025 Yeast Dnm1G178R causes altered organelle dynamics and sheds light on the human DRP1G149R disease mechanism. Mitochondrion 1 39788359
2024 Spontaneous Remission of Epileptic Seizures Following Norovirus Infection in a Patient With DNM1 Encephalopathy. Cureus 1 38903324
2023 Insights into the role of the conserved GTPase domain residues T62 and S277 in yeast Dnm1. International journal of biological macromolecules 1 37838106
1997 Dynamin genes Dnm1 and Dnm2 are located on proximal mouse chromosomes 2 and 9, respectively. Genomics 1 9143510
2026 MDA5 regulates BCR signaling and B-cell function via NF-κB-mediated DNM1. Cellular & molecular immunology 0 41476189
2026 DNM1-related disorder is characterized by recurrent variants and phenotypic homogeneity. medRxiv : the preprint server for health sciences 0 42006794
2026 DNA nanotechnology for nucleic acid analysis: detection of RNA and dsDNA amplicons using a multifunctional DNAzyme nanomachine (DNM). The Analyst 0 42046964
2025 Progressive Cone-Rod Synaptic Dysfunction in Dynamin-1 ( DNM1 ) Related Developmental and Epileptic Encephalopathy: A Distinct Retinal Phenotype in Human. Clinical genetics 0 39925045
2025 Reversal of metformin's anti-proliferative effect in fission yeast efr3 and dnm1 (DRP1) mutants with elongated mitochondria. npj metabolic health and disease 0 39991135
2025 Interpretation of de novo mutations (DNM) and genetic counseling for sporadic hearing loss based on family trio-based sequencing. Yi chuan = Hereditas 0 40068948
2025 The Yeast Parkinson's Disease Model Exhibits An Increase in Peroxisome Number Independent of the Division Proteins Vps1 and Dnm1. Molecular neurobiology 0 40702290
2025 Dnm1 Is Required for the Focal Clustering of Fis1 on the Mitochondrial Outer Membrane. microPublication biology 0 40910107
2025 Long-Read Genome Sequencing Establishes Biallelic Pathogenic Variants in DNM1 With Distinct Functional Effects as the Cause of Early Infantile Developmental and Epileptic Encephalopathy. American journal of medical genetics. Part A 0 41340537
2022 Generation of an induced pluripotent stem cell line (FDCHI007-A) derived from a patient with developmental and epileptic encephalopathy Type 31 carrying heterozygous c.545C > A mutation in DNM1 gene. Stem cell research 0 35184021
2020 Isolation and Analysis of Mitochondrial Fission Enzyme DNM1 from Saccharomyces cerevisiae. Methods in molecular biology (Clifton, N.J.) 0 32529359
2014 WITHDRAWN: Blm10-proteasomes antagonize mitochondrial fission through degradation of Dnm1. The Journal of biological chemistry 0 24285543