Affinage

DNM1

Dynamin-1 · UniProt Q05193

Length
864 aa
Mass
97.4 kDa
Annotated
2026-06-09
67 papers in source corpus 23 papers cited in narrative 26 extracted findings
Cross-family judge vs UniProt: Affinage preferred faithfulness: 8/8 claims corpus-supported (100%)

Mechanistic narrative

Synthesis pass · prose summary of the discoveries below

DNM1 (dynamin-1) is a self-assembling mechanochemical GTPase that couples GTP hydrolysis to membrane constriction and scission, acting both in synaptic vesicle endocytosis in neurons and, in its yeast ortholog Dnm1, as the core mitochondrial fission GTPase (PMID:21170049, PMID:27066543). In the mitochondrial fission context, the protein assembles into a unique helical lattice that, upon GTP hydrolysis, constricts liposomes and then dissociates from the bilayer (PMID:21170049); assembly-stimulated GTP hydrolysis and conserved GTPase-domain residues (G2 motif T62, G5 motif S277) are required for both catalytic activity and fission function (PMID:32529359, PMID:37838106). Dnm1 generates the negative Gaussian (saddle) curvature characteristic of scission necks, narrowing the membrane to a ~12.6 nm neck via a helical membrane-restructuring domain (PMID:29202017). Its minimal cytoplasmic unit is a dimer, and higher-order multimerization is needed to reorganize the adaptor into punctate fission complexes (PMID:16601120). Recruitment to the outer mitochondrial membrane proceeds through the receptor Fis1 — which binds Dnm1 via the concave face of its TPR-like domain and is held under N-terminal autoinhibition — and the adaptor Mdv1/Net2p, which links Fis1 to Dnm1 and regulates Dnm1 self-assembly; an Insert B motif in Dnm1 mediates the Mdv1 contact (PMID:17884824, PMID:11179417, PMID:16272155, PMID:23148233). Completion of division requires cooperation with the intermembrane-space factor Atg44, with Dnm1 acting from the cytosolic face and Atg44 from inside (PMID:38818923). Dnm1 levels and fission output are tuned by Blm10-proteasome degradation and by inputs including microtubule association, glucose/PKA signaling, and cell-cycle cues (PMID:24604417, PMID:30602572, PMID:31562247). In humans, dominant-negative de novo DNM1 mutations cause epileptic encephalopathy: variants such as A177P, K206N, G359A, R237W and a recurrent splice variant impair oligomerization and vesicle scission, producing accumulation of enlarged synaptic vesicles and altered synaptic transmission, with the brain-predominant exon-10a isoform (DNM1A) associated with more severe disease (PMID:27066543, PMID:37648685, PMID:36413998, PMID:33372033).

Mechanistic history

Synthesis pass · year-by-year structured walk · 14 steps
  1. 1995 Medium

    Established that the yeast DNM1 gene product is a dynamin-related GTPase acting in a defined endocytic step, anchoring its role in membrane trafficking before its fission function was known.

    Evidence Gene disruption with receptor half-life, ligand internalization, and vacuolar sorting assays in yeast

    PMID:7622557

    Open questions at the time
    • Did not identify the membrane-scission mechanism
    • No structural or biochemical characterization of GTPase activity
  2. 2001 High

    Identified the adaptor Net2p/Mdv1 as a Dnm1-interacting fission factor that localizes to constriction sites, revealing how Dnm1 is recruited to mitochondria.

    Evidence Yeast two-hybrid, fluorescence and immunoelectron microscopy, genetic disruption with morphology readout

    PMID:11179417

    Open questions at the time
    • Molecular interfaces of the interaction not defined
    • Order of recruitment relative to Fis1 unresolved
  3. 2006 High

    Defined the assembly hierarchy showing the cytoplasmic Dnm1 dimer as the minimal unit and that multimerization is required to convert diffuse Mdv1 into punctate fission complexes.

    Evidence In vivo/in vitro oligomerization assays, Co-IP, fluorescence microscopy, genetics in yeast

    PMID:16601120

    Open questions at the time
    • Stoichiometry of the assembled complex not determined
    • Trigger for multimerization on the membrane unclear
  4. 2007 High

    Mapped the direct Fis1-Dnm1 interaction to the Fis1 TPR concave surface and identified N-terminal autoinhibition as a regulatory switch controlling Dnm1 access.

    Evidence In vitro binding assays, site-directed mutagenesis, quantitative affinity measurements

    PMID:17884824

    Open questions at the time
    • What relieves Fis1 autoinhibition in vivo not identified
    • Direct vs adaptor-mediated Fis1-Dnm1 contact in the complex not fully resolved
  5. 2007 Medium

    Showed in a divergent alga that a distinct WD40/coiled-coil protein (Mda1) forms a pre-existing ring recruiting Dnm1 late in division, demonstrating conserved late-stage Dnm1 action and GTP-dependent release.

    Evidence Biochemical purification of the division machinery, immuno-EM, GTP-induced disassembly and sedimentation

    PMID:17360593

    Open questions at the time
    • Relationship of Mda1 to Mdv1/Fis1 adaptors unclear
    • Mechanism of Dnm1 release upon GTP addition not structurally defined
  6. 2010 High

    Provided the structural basis for fission, showing Dnm1 forms a unique helix that drives large GTP-hydrolysis-coupled liposome constriction followed by membrane dissociation.

    Evidence Cryo-EM 3D structure and liposome constriction assay

    PMID:21170049

    Open questions at the time
    • In-cell constriction dimensions not directly measured
    • Coupling between conformational change and scission completion incomplete
  7. 2012 High

    Pinpointed the Insert B motif of Dnm1 as the structural determinant for Mdv1 binding, explaining how Dnm1 is physically tethered to the mitochondrial adaptor.

    Evidence Site-directed mutagenesis, suppressor screen, Co-IP, fluorescence microscopy in yeast

    PMID:23148233

    Open questions at the time
    • Exact Mdv1 beta-propeller contact residues only inferred from suppressors
    • Whether Insert B contacts other partners not addressed
  8. 2014 Medium

    Identified Blm10-proteasome degradation of Dnm1 as a homeostatic brake on fission, linking Dnm1 protein levels to mitochondrial integrity.

    Evidence In vitro and in vivo degradation assays, yeast genetics, mitochondrial morphology and respiration measurements

    PMID:24604417

    Open questions at the time
    • Signals targeting Dnm1 for Blm10-dependent turnover unknown
    • Whether degradation is regulated during the cell cycle unclear
  9. 2015 High

    Demonstrated that human epileptic-encephalopathy DNM1 mutations act dominant-negatively by impairing oligomerization and endocytic vesicle scission, establishing the disease mechanism.

    Evidence Transferrin endocytosis assays, oligomerization analysis, EM in cells and Dnm1-Ftfl mouse brain

    PMID:27066543

    Open questions at the time
    • Synapse-level consequences not yet measured
    • Therapeutic correction not addressed
  10. 2017 Medium

    Defined the biophysical mechanism of scission, showing Dnm1 generates negative Gaussian curvature to form a ~12.6 nm fission neck via a helical membrane-restructuring domain, with Fis1 inhibiting this activity.

    Evidence Synchrotron SAXS, machine-learning domain identification, in silico mutagenesis

    PMID:29202017

    Open questions at the time
    • Helical domain function not validated by in-cell mutagenesis
    • How Fis1 inhibits membrane restructuring mechanistically unclear
  11. 2019 Medium

    Connected cytoskeletal and metabolic cues to fission, showing microtubule association impedes Dnm1 assembly while glucose starvation and low PKA promote Dnm1 recruitment.

    Evidence Kinesin and pathway gene deletions, live-cell and time-lapse imaging of Dnm1 in fission yeast

    PMID:30602572 PMID:31562247

    Open questions at the time
    • Molecular link between PKA activity and Dnm1 recruitment not defined
    • Whether microtubule effect is steric or signaled is unresolved
  12. 2023 High

    Defined GTPase-domain residues T62 and S277 as essential for catalysis and fission, with S277 variants acting dominant-negatively, and modeled the prevalent human R237W mutation in a knock-in mouse rescued pharmacologically.

    Evidence Site-directed mutagenesis with GTPase assays in yeast; R237W knock-in mouse with electrophysiology, endocytosis assays, and BMS-204352 rescue

    PMID:37648685 PMID:37838106

    Open questions at the time
    • Physiological kinases for T62/S277 not identified
    • Long-term durability of pharmacological rescue unknown
  13. 2024 Medium

    Showed fission completion requires the intermembrane-space factor Atg44 cooperating with cytosolic Dnm1, and that Dnm1 in turn drives focal clustering of its receptor Fis1, revealing reciprocal spatial organization.

    Evidence atg44 deletion with CLEM and live imaging; CRISPR endogenous Fis1 tagging with dnm1 deletion in yeast

    PMID:38818923 PMID:40910107

    Open questions at the time
    • How inner- and outer-membrane scission events are coordinated mechanistically unclear
    • Molecular basis of Dnm1-dependent Fis1 clustering not defined
  14. 2024 High

    Established the disease isoform and a corrective gene-therapy principle, identifying the brain-predominant DNM1A isoform and showing that combined knockdown-replace is required to rescue the dominant-negative mutant.

    Evidence Brain RNA-seq isoform analysis with splice variant functional assay and neuropathological EM; AAV9 bivalent knockdown-replace in mouse with electrophysiology and transcriptomics

    PMID:33372033 PMID:36413998 PMID:39127888

    Open questions at the time
    • Isoform-specific structural differences not resolved
    • Clinical translation of knockdown-replace not established

Open questions

Synthesis pass · forward-looking unresolved questions
  • How DNM1's dual roles in synaptic endocytosis and mitochondrial fission are coordinated within a cell, and whether proposed roles in redox sensing, B-cell actin dynamics, and cancer cell N-cadherin recycling reflect direct DNM1 mechanisms, remain open.
  • Redox-sensing and cancer/immune roles rest on Low-confidence or preprint evidence
  • Direct molecular mechanism in these contexts not demonstrated

Mechanism profile

Synthesis pass · controlled-vocabulary classification · explore literature graph →
Molecular activity
GO:0003924 GTPase activity 3 GO:0140657 ATP-dependent activity 2 GO:0008289 lipid binding 1
Localization
GO:0005739 mitochondrion 3 GO:0005829 cytosol 1
Pathway
R-HSA-1643685 Disease 3 R-HSA-1852241 Organelle biogenesis and maintenance 3 R-HSA-112316 Neuronal System 2 R-HSA-5653656 Vesicle-mediated transport 2
Partners
ATG44BLM10FIS1MDA1MDV1
Complex memberships
mitochondrial division machinery (Dnm1-Mdv1-Fis1)

Evidence

Reading pass · 26 per-paper findings extracted from the source corpus
Year Finding Method Journal Conf PMIDs
2010 Cryo-EM structural analysis of yeast Dnm1 in a GTP-bound state revealed it adopts a unique helical assembly distinct from dynamin. Upon GTP hydrolysis, Dnm1 constricted liposomes with a substantially larger conformational change than dynamin, and subsequently dissociated from the lipid bilayer, supporting a mechanochemical role during mitochondrial division. Cryo-EM 3D structure determination; liposome constriction assay Nature structural & molecular biology High 21170049
2001 Yeast Net2p (Mdv1/Caf4 homolog) was identified as a Dnm1p-interacting protein required for mitochondrial fission. Net2p localizes to dot-like structures on the mitochondrial surface that colocalize with Dnm1p, preferentially at constriction sites along mitochondrial tubules. Yeast two-hybrid; co-localization by fluorescence and immunoelectron microscopy; genetic disruption with mitochondrial morphology readout Molecular biology of the cell High 11179417
2005 Structure-function analysis of Mdv1 showed that dynamic interactions between Mdv1 and assembled Dnm1 regulate Dnm1 self-assembly at the mitochondrial outer membrane, and that Mdv1 functions as an adaptor linking Fis1 with Dnm1 to promote mitochondrial division. Structure-function mutagenesis of Mdv1; co-immunoprecipitation; yeast genetics with mitochondrial morphology readout The Journal of biological chemistry High 16272155
2007 S. cerevisiae Fis1 directly binds Dnm1 via the concave surface of its tetratricopeptide repeat-like domain. The Fis1 N-terminal arm acts in an autoinhibitory manner, decreasing Fis1-Dnm1 binding affinity by more than 100-fold, suggesting regulated access to the Dnm1 binding site. In vitro binding assays; site-directed mutagenesis of Fis1; quantitative binding measurements The Journal of biological chemistry High 17884824
2006 The minimum oligomeric form of cytoplasmic Dnm1p is a dimer. Dimeric Dnm1G385Dp stably interacts with Mdv1p on the outer mitochondrial membrane independently of higher-order assembly, but multimerization of Dnm1p is required to reorganize Mdv1p from uniform mitochondrial localization into punctate fission complexes. Fis1p is present in assembled fission complexes. In vivo and in vitro oligomerization assays; co-immunoprecipitation; fluorescence microscopy; genetic analysis The Journal of biological chemistry High 16601120
2012 A novel motif in the Insert B domain of yeast Dnm1 is required for association with the mitochondrial adaptor Mdv1. Mutation of this motif specifically disrupts Dnm1-Mdv1 interactions, blocking Dnm1 recruitment to mitochondria and mitochondrial fission. Suppressor mutations in Mdv1 identified potential binding interfaces on the Mdv1 β-propeller domain. Site-directed mutagenesis; suppressor screen; co-immunoprecipitation; fluorescence microscopy; yeast genetics The Journal of cell biology High 23148233
2015 Epileptic encephalopathy-causing de novo mutations in human DNM1 (A177P, K206N, G359A) impair endocytosis in a dominant-negative manner. K206N decreased protein levels; G359A disrupted higher-order DNM1 oligomerization; all three mutations caused vesicle defects consistent with impaired vesicle scission activity. Transferrin endocytosis assay in transfected HeLa/COS-7 cells; high-content imaging; Western blotting; electron microscopy of transfected cells and Dnm1-Ftfl mouse brain Neurology. Genetics High 27066543
2017 Synchrotron small-angle X-ray scattering (SAXS) revealed that Dnm1 restructures membranes into phases rich in negative Gaussian curvature (saddle-shaped curvature found at scission necks), inducing a fission neck of ~12.6 nm diameter. A helical domain of Dnm1 identified by machine learning was shown to be responsible for this membrane curvature generation. Fis1 inhibits this pro-fission membrane activity of Dnm1. Synchrotron SAXS; machine learning (SVM) identification of membrane-restructuring domain; in silico mutagenesis; phylogenetic analysis ACS central science Medium 29202017
2014 Proteasomes associated with the Blm10 activator protein degrade Dnm1, thereby antagonizing mitochondrial fission. In the absence of BLM10, Dnm1 degradation is impaired in vitro and in vivo, leading to elevated Dnm1 levels, increased mitochondrial fragmentation, and mitochondrial dysfunction. In vitro and in vivo proteasome degradation assays; yeast genetics; mitochondrial morphology analysis; respiratory capacity measurements The Journal of biological chemistry Medium 24604417
2019 In fission yeast, association of mitochondria with microtubules physically impedes the assembly of Dnm1 around mitochondria, thereby inhibiting mitochondrial fission. Disruption of microtubule dynamics (via kinesin-like protein deletions) alters mitochondrial lengths by modulating Dnm1 assembly, and increased mitochondrial fission upon mitotic spindle formation reduces partitioning errors during mitochondrial segregation. Gene deletions of kinesin-like proteins; high-resolution and time-lapse fluorescence microscopy; live-cell imaging of Dnm1 assembly The Journal of biological chemistry Medium 30602572
2009 In fission yeast, Dnm1 mediates mitochondrial fission in a microtubule-dependent manner. Dnm1-YFP localizes to foci at sites of mitochondrial severing at interfaces between adjacent nucleoids. Microtubule depolymerization causes mitochondrial fragmentation only when Dnm1 is present; mitochondrial fusion is microtubule-independent. Gene deletion; fluorescence microscopy with YFP-tagged Dnm1; microtubule depolymerization experiments Cell motility and the cytoskeleton Medium 19373772
2007 In Cyanidioschyzon merolae, Dnm1 co-purifies with the WD40/coiled-coil protein Mda1 as the two major proteins in the purified mitochondrial division machinery from M-phase arrested cells. Mda1 forms a ring on the mitochondrial outer surface preceding Dnm1 recruitment; Dnm1 colocalizes with Mda1 only in late division stages. Addition of GTP to the purified machinery releases Dnm1 independently, suggesting Mda1 forms a stable homo-oligomeric core structure. Biochemical purification of mitochondrial division machinery; immunofluorescence and immunoelectron microscopy; GTP-induced disassembly assay; sedimentation analysis Proceedings of the National Academy of Sciences of the United States of America Medium 17360593
2019 In fission yeast, glucose starvation promotes Dnm1 localization to mitochondria and increases mitochondrial fission frequency. Low PKA activity enhances glucose starvation-induced mitochondrial fragmentation, while high PKA activity confers resistance. AMPK is not involved in this process. Live-cell fluorescence microscopy; genetic manipulation of PKA and AMPK; profusion chamber experiments The Journal of biological chemistry Medium 31562247
2023 The R237W mutation in human DNM1 (most prevalent pathogenic variant) disrupts dynamin-1 enzyme activity and endocytosis when overexpressed in central neurons. Neurons from heterozygous R237W knock-in mice display dysfunctional endocytosis, altered excitatory neurotransmission, and seizure-like phenotypes. Treatment with BMS-204352, which accelerates endocytosis, corrects these phenotypes at cell, circuit, and in vivo levels. Knock-in mouse model; electrophysiology; endocytosis assays in neurons; in vivo pharmacological rescue Nature communications High 37648685
2022 A recurrent de novo splice site variant (c.1197-8G>A) in DNM1 intron 9 causes insertion of two amino acids predicted to impair oligomerization-dependent activity, acting through a dominant-negative mechanism. Neuropathological samples show accumulation of enlarged synaptic vesicles adherent to the plasma membrane, consistent with impaired vesicular fission. The brain predominantly expresses the exon 10a-containing isoform (DNM1A), and variants affecting this isoform cause more severe disease. RNA sequencing of pediatric brain samples; RT-PCR and Sanger sequencing of patient fibroblast cDNA; neuropathological electron microscopy; functional variant analysis American journal of human genetics High 36413998
2021 In mouse cortical neurons carrying the Dnm1-Ftfl dominant-negative variant, miniature and spontaneous EPSCs and IPSCs are larger but less frequent at all synapse types, with reduced excitatory and inhibitory synaptic vesicle markers. Baseline evoked transmission is reduced specifically at inhibitory synapses onto excitatory neurons due to a smaller pool of releasable synaptic vesicles. Paired whole-cell electrophysiology recordings from cultured cortical neurons; synaptic vesicle marker quantification eNeuro Medium 33372033
2024 Atg44 (mitofissin) is required to complete Dnm1-mediated mitochondrial fission in yeast. In Atg44-deficient cells, Dnm1 accumulates at mitochondrial constriction sites where both outer and inner membranes remain continuous, indicating Dnm1 cannot complete fission without Atg44. Atg44 and Dnm1 cooperatively execute fission from inside (IMS) and outside (cytoplasm) the mitochondria, respectively. Yeast genetics (atg44 deletion); correlative light and electron microscopy (CLEM); live fluorescence microscopy of Dnm1 localization Autophagy Medium 38818923
2023 Two GTPase domain residues of yeast Dnm1, T62 (G2 motif) and S277 (G5 motif), are functionally important: non-phosphorylatable and phosphomimetic variants of both residues abolished GTPase activity and mitochondrial fission function without altering secondary structure. S277 variants act in a dominant-negative manner without altering Dnm1 localization, while T62 variants alter localization. Both residues are putatively phosphorylated. Site-directed mutagenesis; in vivo and in vitro GTPase activity assays; yeast genetics with mitochondrial morphology readout; fluorescence microscopy International journal of biological macromolecules Medium 37838106
2025 In yeast, Dnm1 is required for the focal clustering of Fis1 on the mitochondrial outer membrane: Fis1 appears as discrete puncta dependent on Dnm1 presence, in addition to diffuse signal. This reveals a feedback mechanism where the downstream effector Dnm1 influences the spatial distribution of its upstream receptor Fis1. CRISPR-Cas9 endogenous mNeonGreen tagging of Fis1; fluorescence microscopy; dnm1 deletion analysis microPublication biology Medium 40910107
1995 Yeast DNM1 encodes a dynamin-related GTPase that participates in receptor-mediated endocytosis at a post-internalization step before fusion with the vacuole (late endosome). Disruption of DNM1 increased the half-life of the Ste3p pheromone receptor 2-3 fold and impeded delivery of internalized receptor to the vacuole during ligand-induced endocytosis, without affecting initial internalization or vacuolar protein sorting. Gene disruption; receptor half-life assay; ligand internalization assay; vacuolar protein sorting assay The Journal of cell biology Medium 7622557
2024 In a mouse model of DNM1 disease expressing a patient-based GABAergic neuron variant, AAV9-based bivalent knockdown-replace (RNAi + codon-optimized Dnm1 cDNA) corrected synaptic transmission alterations from inhibitory to excitatory neurons and abrogated disease-associated transcriptomic changes. RNAi or cDNA alone were insufficient, demonstrating that the dominant-negative mutant DNM1 must be eliminated and replaced with wild-type for rescue. AAV9 gene therapy in mouse model; electrophysiological recordings of cortical neurons; RNA sequencing and functional annotation clustering Molecular therapy Medium 39127888
2020 Purified S. cerevisiae Dnm1 exhibits assembly-stimulated hydrolysis of GTP, consistent with other fission dynamins. The yeast-purified enzyme shows different kinetics compared to enzyme isolated from non-native sources. Protein purification from native yeast source; in vitro GTPase activity assay Methods in molecular biology Medium 32529359
2024 In fission yeast, Mfi2 (mitofissin 2), a mitochondrial outer membrane microprotein, independently facilitates mitochondrial fission during mitophagy alongside Dnm1. Mfi2 binds lipid membranes and mediates membrane fission in vitro, and overexpression of a C-terminally truncated form of Mfi2 partially restores mitophagy in atg44Δ cells. Genetic analyses reveal Mfi2 and Dnm1 function independently in mitophagy-associated fission. In vitro membrane fission assay; yeast genetics; fluorescence microscopy; genetic epistasis bioRxivpreprint Low
2024 In fission yeast, cellular redox state (controlled by electron entry into the ETC) controls mitochondrial morphology through Dnm1: conditions causing oxidized cytosol lead to rapid mitochondrial fragmentation, and Dnm1 fission machinery responds within minutes to redox state changes, preceding the change in mitochondrial form. Live-cell fluorescence microscopy; genetic manipulation of ETC components; pharmacological redox manipulation; time-lapse imaging of Dnm1 bioRxivpreprint Low
2024 Human DNM1 promotes endocytic recycling of N-cadherin in ovarian cancer cells, maintaining a mesenchymal state. DNM1 upregulates N-cadherin by promoting its endocytosis and recycling, inducing cell polarization and motility. Loss of DNM1 suppresses peritoneal metastatic colonization in vivo. Master regulators algorithm; molecular assays (endocytosis/recycling); ATAC-seq and RNA-seq; in vivo metastasis model with DNM1 depletion bioRxivpreprint Low
2026 MDA5 regulates actin polymerization in B cells via an MDA5-NF-κB-DNM1 axis; impaired BCR signaling in Mda5 KO B cells can be rescued by treatment with the dynamin 1 (DNM1) activator Bis-T-23, placing DNM1 downstream of MDA5-NF-κB signaling in cytoskeletal dynamics regulation. Mda5 knockout B cells; pharmacological rescue with Bis-T-23; actin polymerization assay; BCR signaling assay Cellular & molecular immunology Low 41476189

Source papers

Stage 0 corpus · 67 papers · ranked by NIH iCite citations
Year Title Journal Citations PMID
2014 De novo mutations in synaptic transmission genes including DNM1 cause epileptic encephalopathies. American journal of human genetics 362 25262651
2010 Conformational changes in Dnm1 support a contractile mechanism for mitochondrial fission. Nature structural & molecular biology 345 21170049
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 93 28667181
2008 Nicotine modulates expression of miR-140*, which targets the 3'-untranslated region of dynamin 1 gene (Dnm1). The international journal of neuropsychopharmacology 77 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 47 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 44 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
2015 De novo DNM1 mutations in two cases of epileptic encephalopathy. Epilepsia 31 26611353
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
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 28 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 25 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
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 17 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 16 34172529
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
2019 Proteomics Analysis of Candida albicans dnm1 Haploid Mutant Unraveled the Association between Mitochondrial Fission and Antifungal Susceptibility. Proteomics 14 31811746
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 13 32631867
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
2018 Core/Shell Structured Fe₃O₄@TiO₂-DNM Nanospheres as Multifunctional Anticancer Platform: Chemotherapy and Photodynamic Therapy Research. Journal of nanoscience and nanotechnology 12 29442618
2024 Atg44/Mdi1/mitofissin facilitates Dnm1-mediated mitochondrial fission. Autophagy 11 38818923
2014 Classical dynamin DNM1 and DNM3 genes attain maximum expression in the normal human central nervous system. BMC research notes 11 24673776
2022 Clinical, Radiological, and Genetic Characterization of a Patient with a Novel Homoallelic Loss-of-Function Variant in DNM1. Genes 9 36553519
2023 DNA Nanomachine (DNM) Biplex Assay for Differentiating Bacillus cereus Species. International journal of molecular sciences 8 36901903
2021 Mimicking human Drp1 disease-causing mutations in yeast Dnm1 reveals altered mitochondrial dynamics. Mitochondrion 8 34157431
2018 DNM1 Mutation in a child associated with progressive bilateral mesial temporal sclerosis. Clinical case reports 8 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 8 28168039
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
2023 Genotypes and phenotypes of DNM1 encephalopathy. Journal of medical genetics 6 37248033
2012 Mitochondrial fission proteins Fis1 and Mdv1, but not Dnm1, play a role in maintenance of heteroplasmy in budding yeast. FEBS letters 6 22575664
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 4 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 4 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 4 37132416
2023 Truncated DNM1 variant underlines developmental delay and epileptic encephalopathy. Frontiers in pediatrics 4 37900685
2024 Study on the mechanism and degradation behavior of Encifer adhaerens DNM-S1 capturing dimethyl phthalate. Chemosphere 2 38641291
2024 Spontaneous Remission of Epileptic Seizures Following Norovirus Infection in a Patient With DNM1 Encephalopathy. Cureus 2 38903324
2025 Yeast Dnm1G178R causes altered organelle dynamics and sheds light on the human DRP1G149R disease mechanism. Mitochondrion 1 39788359
2025 Progressive Cone-Rod Synaptic Dysfunction in Dynamin-1 ( DNM1 ) Related Developmental and Epileptic Encephalopathy: A Distinct Retinal Phenotype in Human. Clinical genetics 1 39925045
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 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

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