Affinage

BICD2

Protein bicaudal D homolog 2 · UniProt Q8TD16

Length
824 aa
Mass
93.5 kDa
Annotated
2026-06-09
57 papers in source corpus 26 papers cited in narrative 27 extracted findings
Cross-family judge vs UniProt: Affinage preferred faithfulness: 7/7 claims corpus-supported (100%)

Mechanistic narrative

Synthesis pass · prose summary of the discoveries below

BICD2 is a coiled-coil cargo adaptor that links the cytoplasmic dynein-dynactin motor to diverse cargoes and switches its cargo selection across the cell cycle (PMID:30345745). It is held in an autoinhibited state that is released by GTP-loaded Rab6a to assemble processive dynein-dynactin-BICD2 (DDB) complexes that drive retrograde transport (PMID:28883039); the Rab6GTP interface is reconfigured by the GDP-to-GTP transition, and disrupting it abolishes BICD2 co-migration with and motility of Rab6-positive secretory vesicles (PMID:38719748). Through this activity BICD2 stabilizes GTP-Rab6A on Golgi membranes, supports COPI-independent Golgi-to-ER retrograde transport, and is required for constitutive secretory cargo flow from the TGN to the plasma membrane, thereby maintaining Golgi integrity (PMID:25962623, PMID:32183910, PMID:23664116, PMID:23664119, PMID:23664120). In a cell-cycle-controlled switch, CDK1 and PLK1 phosphorylate BICD2 in G2, releasing autoinhibition and directing it to the nuclear envelope, where it engages CDK1-phosphorylated RanBP2/Nup358 to tether and separate centrosomes (PMID:37105961); a Nup358 cargo-recognition segment undergoes a coil-to-helix transition that activates DDB motility (PMID:35229716). At the nuclear envelope BICD2 also binds Nesprin-2, and RanBP2 and Nesprin-2 compete for mutually exclusive BICD2 interactions that drive successive phases of brain development—interkinetic nuclear migration in radial glia via RanBP2 and post-mitotic neuronal migration via Nesprin-2 (PMID:32665036, PMID:32619477, PMID:36930595). BICD2 additionally serves as a capsid-specific dynein adaptor co-opted by HIV-1 and HPV16 for retrograde transport to the nucleus (PMID:30068656, PMID:38829892), and has dynein-independent centriolar functions: it binds CP110 at the mother centriole to promote its removal and ciliogenesis (PMID:41102520) and controls mother-daughter centriole engagement. Dominant SMALED2/SMA-causing mutations increase BICD2 affinity for dynein-dynactin and hyperactivate DDB motility, increase microtubule stability, and remodel cargo-specific interactomes, impairing neuronal migration and motor neuron development (PMID:23664119, PMID:23664120, PMID:28883039, PMID:29528393, PMID:41334889).

Mechanistic history

Synthesis pass · year-by-year structured walk · 11 steps
  1. 2002 Medium

    Established BICD2 as a microtubule-associated, cytoskeleton-localized protein subject to kinase regulation, raising the question of how its localization is controlled.

    Evidence In vitro Nek8 kinase assay, Co-IP, and immunofluorescence with nocodazole in cells

    PMID:11864968

    Open questions at the time
    • No functional consequence of Nek8 phosphorylation defined
    • No cargo or motor link established at this stage
  2. 2013 Medium

    Linked BICD2 to dominant human motor neuron disease and to Golgi maintenance, showing mutations enhance dynein-dynactin binding while altering Rab6A association.

    Evidence Transient transfection of mutant cDNAs and patient fibroblast/lymphoblast IF and Co-IP in HeLa and SH-SY5Y cells

    PMID:23664116 PMID:23664119 PMID:23664120

    Open questions at the time
    • Mechanism linking enhanced dynein binding to disease not resolved
    • Whether Golgi fragmentation is cause or consequence unclear
  3. 2015 Medium

    Defined a biochemical role for BICD2 as a cytosolic factor that targets and stabilizes GTP-Rab6A on Golgi membranes, supporting COPI-independent retrograde transport.

    Evidence Golgi-targeting reconstitution in SLO-permeabilized cells, BICD2 knockdown, and FRAP of GFP-Rab6A

    PMID:25962623

    Open questions at the time
    • Did not reconstitute motility with purified dynein
    • Direct vs. indirect Rab6A stabilization not separated
  4. 2017 High

    Demonstrated with purified components that BICD2 is autoinhibited and that Rab6a-GTP releases it to form processive DDB complexes, and that disease mutants hyperactivate this motility.

    Evidence In vitro single-molecule motility with purified DDB and liposome-bound Rab6a-GTP, inducible organelle transport, and neuronal overexpression

    PMID:28883039

    Open questions at the time
    • Structural basis of autoinhibition not visualized
    • How hyperactivation translates to neuronal pathology not fully defined
  5. 2018 Medium

    Showed BICD2 cargo selection is cell-cycle-gated—Rab6GTP vesicles in G1/S versus the nucleus via Nup358 in G2—with defined stoichiometry and affinities and RanGTP as a negative regulator.

    Evidence Quantitative fluorescence binding assays, oligomeric state determination, and RanGTP competition

    PMID:30345745

    Open questions at the time
    • In vitro affinities not validated against the in-cell switch
    • Kinase-driven control of the switch not addressed here
  6. 2018 High

    Extended BICD2's adaptor role to viral hijacking and to disease-relevant cytoskeletal changes, identifying it as a capsid-specific dynein adaptor for HIV-1 and showing mutations increase microtubule stability and impair neuromuscular function.

    Evidence In vitro CA-binding and immunodepletion with siRNA and nuclear DNA PCR for HIV-1; microtubule stability assays and Drosophila neuron/muscle-specific expression for SMALED2

    PMID:29528393 PMID:30068656

    Open questions at the time
    • Capsid recognition determinants on BICD2 not mapped
    • Link between microtubule hyperstability and motor neuron loss mechanistic chain incomplete
  7. 2020 High

    Resolved the nuclear-envelope arm of BICD2 function, showing Nesprin-2 recruits BICD2 and dynein/kinesin-1 to drive neuronal nuclear migration, and demonstrating cell-intrinsic neuronal and non-cell-autonomous muscle requirements in mice.

    Evidence In utero electroporation in mouse/rat brain, Co-IP and LEWD/KASH mutagenesis, conditional neuron- and muscle-specific Bicd2 KO mice, and VSV-G secretory reporter in patient fibroblasts

    PMID:31655624 PMID:32183910 PMID:32619477 PMID:32665036

    Open questions at the time
    • How dynein/kinesin-1 directional balance is set not fully defined
    • Molecular basis of non-cell-autonomous muscle contribution unresolved
  8. 2022 High

    Defined the kinase logic and structural transitions activating BICD2 for nuclear/centrosomal cargo, showing CDK1 then PLK1 phosphorylation switches it to a dynein-competent state engaging phospho-RanBP2, with a Nup358 coil-to-helix transition activating motility.

    Evidence In vitro kinase assays with residue mapping, phospho-mutant centrosome separation assays, NMR/CD on the Nup358 helix, and in vitro single-molecule motility; cryo-EM of full-length BicD2

    PMID:35229716 PMID:36150379 PMID:37105961

    Open questions at the time
    • High-resolution structure of the activated DDB-BICD2 complex lacking
    • Full-length cryo-EM gave limited functional/mutagenesis validation
  9. 2023 High

    Established that mutually exclusive RanBP2 versus Nesprin-2 binding partitions BICD2 between successive developmental tasks, and uncovered a degradation route and a non-transport signaling function.

    Evidence In vitro competition assays and in utero electroporation with binding-selective mutations; VHL ubiquitination/Co-IP and STAT1 nuclear translocation assays; AlphaFold/docking of the Nup358 interface

    PMID:36930595 PMID:37833251 PMID:37892127

    Open questions at the time
    • Nup358 interface model not confirmed by direct structure (Low-confidence prediction)
    • How VHL-mediated turnover integrates with cell-cycle regulation unknown
  10. 2024 Medium

    Broadened BICD2's cargo repertoire and regulation, identifying HPV16 L2/Rab6a-dependent transport, a refined Rab6GTP interface, and m6Am-mediated mRNA control linking BICD2 to ciliogenesis.

    Evidence In vitro L2/Rab6a binding, siRNA and fractionated Co-IP and entry assays; AlphaFold Rab6 model with mutagenesis and vesicle motility; PCIF1 LC-MS m6Am mapping with knockdown epistasis and ciliogenesis assays

    PMID:38526325 PMID:38719748 PMID:38829892 PMID:39431827

    Open questions at the time
    • Rab6GTP interface based on computational model with cell validation only
    • How m6Am modification tunes BICD2 levels mechanistically incomplete
  11. 2025 High

    Revealed dynein-independent centriolar functions and further disease/regulatory mechanisms, showing BICD2 binds CP110 to promote ciliogenesis, controls centriole engagement, and that mutations remodel its interactome.

    Evidence Direct CP110 binding with knockdown epistasis and zebrafish rescue; centriole engagement live imaging with phospho-mutants (preprint); AP-MS interactome with motility assays for R747C; PACS1-HDAC6 Co-IP and motility assays

    PMID:41102520 PMID:41334889 PMID:41888583

    Open questions at the time
    • How a transport adaptor performs dynein-independent centriolar roles mechanistically unclear
    • Centriole-engagement role rests on preprint without peer review

Open questions

Synthesis pass · forward-looking unresolved questions
  • How the kinase-, Rab6-, and competition-based regulatory inputs are integrated in vivo to coordinate BICD2's transport and dynein-independent centriolar/ciliary functions, and how disease mutations selectively perturb specific cargo arms, remains unresolved.
  • No unified structural model of activated vs. autoinhibited BICD2 with each cargo
  • Tissue-specific basis of motor neuron vulnerability not established
  • Mechanism coupling transport adaptor activity to centriole engagement undefined

Mechanism profile

Synthesis pass · controlled-vocabulary classification · explore literature graph →
Molecular activity
GO:0060090 molecular adaptor activity 4 GO:0008092 cytoskeletal protein binding 3 GO:0038024 cargo receptor activity 3
Localization
GO:0005635 nuclear envelope 4 GO:0005794 Golgi apparatus 3 GO:0005815 microtubule organizing center 3 GO:0005829 cytosol 1 GO:0005856 cytoskeleton 1
Pathway
R-HSA-1266738 Developmental Biology 4 R-HSA-1643685 Disease 4 R-HSA-5653656 Vesicle-mediated transport 4 R-HSA-9609507 Protein localization 3 R-HSA-1640170 Cell Cycle 2 R-HSA-1852241 Organelle biogenesis and maintenance 2
Partners
CP110DCTN1HDAC6PLK1RAB6ARANBP2SYNE2VHL
Complex memberships
HOPS complex (cargo)dynein-dynactin-BICD2 (DDB)

Evidence

Reading pass · 27 per-paper findings extracted from the source corpus
Year Finding Method Journal Conf PMIDs
2013 BICD2 disease-causing mutations (p.Ser107Leu, p.Asn188Thr, p.Thr703Met) cause Golgi fragmentation when transiently transfected in HeLa cells, and BICD2 levels are reduced and trapped within the fragmented Golgi in patient fibroblasts, demonstrating the protein's role in maintaining Golgi integrity. Transient transfection of HeLa cells with mutant BICD2 cDNAs; immunofluorescence of patient fibroblasts American journal of human genetics Medium 23664116 23664119 23664120
2013 BICD2 mutations (p.Ser107Leu and p.Glu774Gly) that cause dominant SMA increase BICD2 binding affinity to the cytoplasmic dynein-dynactin complex; p.Ser107Leu causes accumulation of BICD2 at the MTOC and reduced colocalization with RAB6A, while p.Glu774Gly impairs interaction with RAB6A. Co-immunoprecipitation; immunofluorescence in HeLa, SH-SY5Y cells and patient lymphoblasts American journal of human genetics Medium 23664119 23664120
2002 BICD2 is phosphorylated by the NIMA-related kinase Nek8 in vitro, and the endogenous proteins associate in vivo; BICD2 localizes to cytoskeletal structures in a microtubule-dependent manner, with nocodazole treatment causing dramatic reorganization of BICD2 correlating with Nek8 phosphorylation. In vitro kinase assay; co-immunoprecipitation; immunofluorescence with nocodazole treatment The Journal of biological chemistry Medium 11864968
2017 Disease-causing BICD2 mutants show enhanced ability to form motile dynein-dynactin-BICD2 (DDB) complexes in vitro; Rab6a-GTP releases BICD2 from an autoinhibited state to promote processive retrograde transport; BICD2 mutants display increased retrograde transport in cells and, when overexpressed in rat hippocampal neurons, decrease neurite growth. In vitro single-molecule motility assays with purified DDB and Rab6a-GTP; inducible organelle transport assay in cells; neuronal overexpression The Journal of cell biology High 28883039
2015 BICD2 functions as a cytosolic factor required for Golgi targeting of Rab6A in semi-intact (SLO-permeabilized) HeLa cells; BICD2 stabilizes the GTP-bound form of Rab6A on Golgi membranes (shown by FRAP), and BICD2/Rab6A play concerted roles in COPI-independent Golgi-to-ER retrograde transport. Golgi-targeting reconstitution assay in streptolysin O-permeabilized cells; BICD2 knockdown; FRAP analysis of GFP-Rab6A; brefeldin A vesicle transport assay Biochimica et biophysica acta Medium 25962623
2018 HIV-1 engages BICD2 as a capsid-specific dynein adaptor: purified recombinant BICD2 binds HIV-1 CA assemblies in vitro; depletion of BICD2 by siRNA reduces HIV-1 nuclear DNA accumulation and retrograde viral movement; immunodepletion of BICD2 from cell extracts reduces dynein association with CA assemblies. siRNA knockdown; in vitro binding assay with purified recombinant BICD2 and CA assemblies; immunodepletion; quantitative nuclear HIV-1 DNA PCR Journal of virology High 30068656
2018 SMALED2-causing BICD2 mutations increase microtubule stability in patient fibroblasts and in motor neurons upon overexpression; in a Drosophila model, neuron-specific expression of BICD2 mutants reduces neuromuscular junction size and impairs locomotion, while muscle-specific expression has no effect. Microtubule stability assays in patient fibroblasts; Drosophila transgenic neuron/muscle-specific expression; NMJ morphology analysis; locomotion assays Human molecular genetics Medium 29528393
2020 BICD2 is required for physiological flow of constitutive secretory cargoes from the Trans Golgi Network to the plasma membrane (VSV-G reporter assay in patient fibroblasts); muscle-specific knockout of Bicd2 in mice reduces L4 ventral motor axons comparably to global knockout, implicating muscle BICD2 in non-cell autonomous motor neuron maintenance. VSV-G secretory cargo reporter assay in SMALED2 patient fibroblasts; conditional muscle-specific Bicd2 knockout mice; L4 ventral root axon counting Acta neuropathologica communications Medium 32183910
2020 BICD2 localizes to the nuclear envelope (NE) through interaction with Nesprin-2; a truncating BICD2 variant (K775X) disrupts this interaction, prevents NE recruitment of BICD2 and dynein, and causes severe neuronal migration failure without affecting centrosome movement in mouse embryos; restoring NE localization via KASH fusion rescues migration. In utero electroporation in mouse embryos; co-immunoprecipitation; live imaging of centrosome/nucleus movement; domain fusion rescue experiments Acta neuropathologica communications High 32665036
2020 Nesprin-2 recruits BicD2 to the nuclear envelope in both migrating neurons and non-mitotic fibroblasts; BicD2 mediates interaction of Nesprin-2 with both dynein and kinesin-1; mutation of the Nesprin-2 LEWD sequence disrupts BicD2 binding; kinesin-1 inhibition accelerates neuronal migration while dynein inhibition blocks forward nuclear movement. In utero electroporation in rat brain; co-immunoprecipitation; dominant-negative constructs; live imaging of nuclear and centrosome movement Current biology : CB High 32619477
2019 BICD2 is required cell-intrinsically in post-mitotic cortical neurons for bipolar locomotion and radial migration; neuron-specific conditional Bicd2 KO mice show severely impaired radial migration of late-born upper-layer neurons; disease-associated point mutation in the RAB6/RANBP2-binding domain fails to rescue cortical neuron migration. Neuron-specific conditional Bicd2 KO mice; single-neuron labeling; rescue experiments with WT and mutant BICD2 constructs Acta neuropathologica communications High 31655624
2022 In G2, CDK1 phosphorylates BICD2 to promote its interaction with PLK1; PLK1 then phosphorylates a single N-terminal residue of BICD2, causing a structural change that facilitates interaction with dynein and dynactin and formation of active motor complexes; phospho-BICD2 preferentially interacts with CDK1-phosphorylated RanBP2 at the nuclear envelope, driving centrosome tethering and separation in G2/M. In vitro kinase assays; phospho-specific antibodies; co-immunoprecipitation; BICD2 phospho-mutants; centrosome separation assays in cells Nature communications High 37105961
2022 A minimal Nup358 domain (residues 2162-2184) undergoes coil-to-α-helix transition upon BicD2 binding and activates dynein/dynactin/BicD2 for processive motility on microtubules; mutations in this Nup358 'cargo recognition α-helix' decrease Nup358/BicD2 interaction, dynein recruitment, and motility. NMR titration and CEST; circular dichroism; mutagenesis; in vitro single-molecule motility assay eLife High 35229716
2023 Nesprin-2 and RanBP2 compete for BICD2 binding in vitro, and mutually exclusive RanBP2-BICD2 vs. Nesprin-2-BICD2 interactions at the nuclear envelope play successive roles in interkinetic nuclear migration in radial glial progenitors (via RanBP2) and post-mitotic neuronal migration (via Nesprin-2). In vitro biochemical competition assays; in utero electroporation-based brain developmental assays with BICD2 mutations affecting RanBP2 vs. Nesprin-2 binding PLoS genetics High 36930595
2018 BicD2 switches cargo selection based on cell cycle phase: it selects Rab6GTP-positive secretory/Golgi vesicles in G1/S (via Rab6GTP binding) and selects the nucleus in G2 (via Nup358 binding); BicD2 and cargo form predominantly 2:2 complexes; the affinity of BicD2 for Nup358 is higher than for Rab6GTP; RanGTP is a negative regulator of the Nup358/BicD2 interaction. Quantitative binding assays (fluorescence-based); oligomeric state determination; concentration-dependent affinity measurements; RanGTP competition assay Biochemistry Medium 30345745
2022 Full-length human BicD2 undergoes pH-dependent conformational changes; it forms dimers and higher-order oligomers; it binds RanBP2 in biochemical interaction studies; cryo-EM reveals structural flexibility consistent with autoinhibited and activated states. Recombinant full-length BicD2 production; cryo-EM; biophysical characterization; interaction studies with RanBP2 Structure (London, England : 1993) Medium 36150379
2024 BICD2 binds directly to the HPV16 L2 capsid protein during cell entry; a short segment near the C-terminus of L2 mediates direct BICD2 binding in vitro; BICD2 depletion causes HPV accumulation in endosome and TGN and inhibits infection; BICD2 recruits HPV to dynein for transport along the endosome-TGN/Golgi axis. siRNA knockdown; in vitro binding assays; cell-based HPV entry assays; co-localization studies PLoS pathogens Medium 38829892
2024 Rab6a facilitates HPV association with BICD2 and dynein specifically in the TGN (but not endosome) of infected cells; L2 capsid protein binds directly to GTP-Rab6a in vitro; Rab6a knockdown impairs HPV exit from TGN and intra-Golgi transport; cycling between GDP- and GTP-Rab6 states is required. siRNA knockdown of Rab6a; in vitro L2-Rab6a binding assay; co-immunoprecipitation of HPV with BICD2/dynein in TGN vs endosome fractions; HPV infection assays mBio Medium 39431827
2024 The binding site of BicD2 for Rab6GTP spans two regions of Rab6 that undergo structural changes upon GDP-to-GTP transition; hydrophobic interface residues are rearranged explaining increased affinity for GTP-bound Rab6; mutations abolishing Rab6GTP binding to BicD2 reduce co-migration and severely diminish motility of Rab6-positive vesicles in cells. AlphaFold structure prediction; mutagenesis; co-migration assays in cells; live-cell vesicle motility assays Life science alliance Medium 38719748
2023 The Nup358 cargo-recognition α-helix binds to BicD2 between residues 747-774 in an antiparallel manner forming a helical bundle; two intermolecular salt bridges stabilize this interface; a secondary interface involves an intrinsically disordered Nup358 region binding BicD2 residues 774-800, overlapping with the Rab6 binding site, explaining competitive cargo selection. AlphaFold2 structure prediction; HADDOCK and ClusPro docking; mutagenesis Biomolecules Low 37892127
2023 BICD2 interacts with VHL; VHL promotes K48-linked polyubiquitination of BICD2, leading to its proteasomal degradation; BICD2 promotes STAT1 nuclear translocation to facilitate IFNγ signaling and enhance IFNγ-mediated suppression of system Xc-, increasing ferroptosis sensitivity in renal epithelial cells. Co-immunoprecipitation; ubiquitination assays; overexpression/knockdown experiments; STAT1 nuclear localization assays Cell death & disease Medium 37833251
2024 PCIF1 methyltransferase activity modifies BICD2 mRNA at a specific m6Am site, reducing BICD2 mRNA stability and translation efficiency; BICD2 depletion reduces ciliogenesis; the ciliogenesis defect in PCIF1-depleted cells (which have elevated BICD2) is rescued by BICD2 knockdown. Quantitative proteomics; siRNA knockdown of PCIF1 and BICD2; single-base LC-MS m6Am site identification; ciliogenesis assays in RPE-1 cells The Journal of cell biology Medium 38526325
2025 BICD2 promotes ciliogenesis by directly binding CP110 at the mother centriole and facilitating CP110 removal; BICD2 is recruited to the mother centriole during ciliogenesis; BICD2 depletion inhibits CP110 removal and ciliogenesis; CP110 knockdown rescues ciliogenesis in BICD2-depleted cells; zebrafish bicd2 morphants show ciliogenesis defects reversed by bicd2 mRNA or Cp110 depletion. Co-immunoprecipitation/direct binding assays; siRNA/shRNA knockdown; ciliogenesis assays in RPE-1 cells; zebrafish morpholino knockdown with rescue EMBO reports High 41102520
2025 SMALED2-linked BICD2 mutations (including R747C) cause dynein hyperactivation; R747C is deficient in binding HOPS complex components and RANBP2; R747C displays gain-of-function interaction with GRAMD1A leading to its mislocalization; wild-type BICD2 interacts with HOPS complex components as novel cargo. Interactome mass spectrometry (AP-MS); dynein motility assays; co-immunoprecipitation; immunofluorescence of GRAMD1A localization eLife Medium 41334889
2025 BICD2 is a centriolar protein with a dynein-independent role in controlling mother-daughter centriole engagement; BICD2 removal causes premature centriole disengagement in G2/early M and centriole amplification; BICD2 centriolar localization is controlled by phosphorylation. Live-cell imaging; BICD2 depletion; centriole engagement/disengagement assays; phospho-mutant analysis; centrosome amplification quantification bioRxivpreprint Medium
2025 PACS1R203W-HDAC6 recruits BICD2 to form a complex that disperses the Golgi and impairs dynein function; BICD2 in this complex reduces dynein initiation frequency and velocity (rescued by HDAC6 inhibition or Lis1 expression), placing BICD2 downstream of the PACS1-HDAC6 axis in dynein-driven Golgi transport. Co-immunoprecipitation; cargo motility assays; HDAC6 inhibition; Golgi morphology assays; dynein initiation frequency measurements Communications biology Medium 41888583
2025 Mechanical tension recruits a third dynein motor to DDB complexes via an auxiliary BicD2 adaptor binding the light intermediate chain; complexes with two or three dyneins generate ~7 pN and ~9 pN respectively; under load DDB primarily takes ~8 nm steps. Single-molecule optical tweezers force measurements; iSCAT tracking; purified DDB complexes from brain lysate bioRxivpreprint Medium

Source papers

Stage 0 corpus · 57 papers · ranked by NIH iCite citations
Year Title Journal Citations PMID
2013 Mutations in BICD2, which encodes a golgin and important motor adaptor, cause congenital autosomal-dominant spinal muscular atrophy. American journal of human genetics 141 23664116
2013 Mutations in BICD2 cause dominant congenital spinal muscular atrophy and hereditary spastic paraplegia. American journal of human genetics 138 23664120
2013 Molecular defects in the motor adaptor BICD2 cause proximal spinal muscular atrophy with autosomal-dominant inheritance. American journal of human genetics 97 23664119
2014 Phenotypic and molecular insights into spinal muscular atrophy due to mutations in BICD2. Brain : a journal of neurology 74 25497877
2018 HIV-1 Engages a Dynein-Dynactin-BICD2 Complex for Infection and Transport to the Nucleus. Journal of virology 63 30068656
2017 Disease-associated mutations in human BICD2 hyperactivate motility of dynein-dynactin. The Journal of cell biology 62 28883039
2020 Nesprin-2 Recruitment of BicD2 to the Nuclear Envelope Controls Dynein/Kinesin-Mediated Neuronal Migration In Vivo. Current biology : CB 51 32619477
2002 Purification, cloning, and characterization of Nek8, a novel NIMA-related kinase, and its candidate substrate Bicd2. The Journal of biological chemistry 51 11864968
2016 Recurrent de novo BICD2 mutation associated with arthrogryposis multiplex congenita and bilateral perisylvian polymicrogyria. Neuromuscular disorders : NMD 48 27751653
2015 Dominant spinal muscular atrophy is caused by mutations in BICD2, an important golgin protein. Frontiers in neuroscience 42 26594138
2020 Loss of BICD2 in muscle drives motor neuron loss in a developmental form of spinal muscular atrophy. Acta neuropathologica communications 40 32183910
2020 Impairment in dynein-mediated nuclear translocation by BICD2 C-terminal truncation leads to neuronal migration defect and human brain malformation. Acta neuropathologica communications 33 32665036
2022 Elevated BICD2 DNA methylation in blood of major depressive disorder patients and reduction of depressive-like behaviors in hippocampal Bicd2-knockdown mice. Proceedings of the National Academy of Sciences of the United States of America 31 35858435
2017 DT-13 synergistically enhanced vinorelbine-mediated mitotic arrest through inhibition of FOXM1-BICD2 axis in non-small-cell lung cancer cells. Cell death & disease 28 28542137
2016 Expanding the phenotype of BICD2 mutations toward skeletal muscle involvement. Neurology 28 27784775
2020 The Genotypic and Phenotypic Spectrum of BICD2 Variants in Spinal Muscular Atrophy. Annals of neurology 25 32057122
2015 Reconstitution of the targeting of Rab6A to the Golgi apparatus in semi-intact HeLa cells: A role of BICD2 in stabilizing Rab6A on Golgi membranes and a concerted role of Rab6A/BICD2 interactions in Golgi-to-ER retrograde transport. Biochimica et biophysica acta 25 25962623
2022 Coil-to-α-helix transition at the Nup358-BicD2 interface activates BicD2 for dynein recruitment. eLife 22 35229716
2023 BICD2 phosphorylation regulates dynein function and centrosome separation in G2 and M. Nature communications 18 37105961
2018 Novel insights into SMALED2: BICD2 mutations increase microtubule stability and cause defects in axonal and NMJ development. Human molecular genetics 18 29528393
2016 Autosomal dominant spinal muscular atrophy with lower extremity predominance: A recognizable phenotype of BICD2 mutations. Muscle & nerve 18 26998597
2019 Dynein activating adaptor BICD2 controls radial migration of upper-layer cortical neurons in vivo. Acta neuropathologica communications 17 31655624
2020 circ_BICD2 acts as a ceRNA to promote tumor progression and Warburg effect in oral squamous cell carcinoma by sponging miR-107 to enhance HK2. American journal of translational research 16 32774714
2023 Role of Nesprin-2 and RanBP2 in BICD2-associated brain developmental disorders. PLoS genetics 15 36930595
2018 In-frame de novo mutation in BICD2 in two patients with muscular atrophy and arthrogryposis. Cold Spring Harbor molecular case studies 15 30054298
2018 A Quantitative Model for BicD2/Cargo Interactions. Biochemistry 13 30345745
2020 Clinical spectrum of BICD2 mutations. European journal of neurology 11 32056343
2023 Critical role of VHL/BICD2/STAT1 axis in crystal-associated kidney disease. Cell death & disease 9 37833251
2022 In vitro characterization of the full-length human dynein-1 cargo adaptor BicD2. Structure (London, England : 1993) 9 36150379
2024 m6Am methyltransferase PCIF1 negatively regulates ciliation by inhibiting BICD2 expression. The Journal of cell biology 8 38526325
2024 The BICD2 dynein cargo adaptor binds to the HPV16 L2 capsid protein and promotes HPV infection. PLoS pathogens 8 38829892
2017 Novel BICD2 mutation in a Japanese family with autosomal dominant lower extremity-predominant spinal muscular atrophy-2. Brain & development 7 29273277
2023 A Structural Model for the Core Nup358-BicD2 Interface. Biomolecules 6 37892127
2024 Molecular mechanism for recognition of the cargo adapter Rab6GTP by the dynein adapter BicD2. Life science alliance 5 38719748
2021 Roles of the multivalent dynein adaptors BicD2 and RILP in neurons. Neuroscience letters 5 33667600
2020 SMALED2 with BICD2 gene mutations: Report of two cases and portrayal of a classical phenotype. Neuromuscular disorders : NMD 5 32709491
2018 BICD2 mutational analysis in hereditary spastic paraplegia and hereditary motor and sensory neuropathy. Muscle & nerve 5 30536747
2024 Rab6a enables BICD2/dynein-mediated trafficking of human papillomavirus from the trans-Golgi network during virus entry. mBio 4 39431827
2022 Biallelic BICD2 variant is a novel candidate for Cohen-like syndrome. Journal of human genetics 4 35338243
2020 A case of severe autosomal dominant spinal muscular atrophy with lower extremity predominance caused by a de novo BICD2 mutation. Brain & development 4 32888736
2019 Adult-onset SMALED2 due to a novel BICD2 mutation presenting with asymmetrical lower limb involvement. Clinical neuropathology 4 30738493
2022 A homozygous loss-of-function variant in BICD2 is associated with lissencephaly and cerebellar hypoplasia. Journal of human genetics 3 35896821
2022 Identification and functional characterization of BICD2 as a candidate disease gene in an consanguineous family with dilated cardiomyopathy. BMC medical genomics 3 36068540
2021 An infant with congenital respiratory insufficiency and diaphragmatic paralysis: A novel BICD2 phenotype? American journal of medical genetics. Part A 3 34825470
2025 Dominant spinal muscular atrophy linked mutations in the cargo binding domain of BICD2 result in altered interactomes and dynein hyperactivity. bioRxiv : the preprint server for biology 2 40462900
2023 Microscopic and Biochemical Hallmarks of BICD2-Associated Muscle Pathology toward the Evaluation of Novel Variants. International journal of molecular sciences 2 37047781
2023 Likely pathogenic variant in the BICD2 gene in fetus presenting with non-immune hydrops. Prenatal diagnosis 2 37173812
2025 Hydrops, Arthrogryposis, and Cerebellar Hypoplasia in a Fetus With a de Novo BICD2 Variant: Expanding the Prenatal Phenotype of SMALED2B. Prenatal diagnosis 1 41249097
2025 Dominant spinal muscular atrophy linked mutations in the cargo binding domain of BICD2 result in altered interactomes and dynein hyperactivity. eLife 1 41334889
2023 High-Resolution Tracking of Dynein-Dynactin-BicD2 Complexes. Methods in molecular biology (Clifton, N.J.) 1 36602686
2021 A novel BICD2 mutation of a patient with Spinal Muscular Atrophy Lower Extremity Predominant 2. Intractable & rare diseases research 1 33996355
2026 Structural characterization of a minimal KLC2/Nup358/BicD2 complex. bioRxiv : the preprint server for biology 0 41648486
2026 PACS1 syndrome mutation disrupts dynein-mediated cargo transport via HDAC6 and BICD2. Communications biology 0 41888583
2025 BICD2 promotes ciliogenesis by facilitating CP110 removal from the mother centriole. EMBO reports 0 41102520
2025 Antisense oligonucleotide of cargo adaptor BICD2 long-term effectively alleviates neuropathic pain via activation of PSD95 in the mouse spinal dorsal horn. Neuropharmacology 0 41349793
2022 Long-term follow-up of a patient with autosomal dominant lower extremity-predominant spinal muscular atrophy-2 due to a BICD2 variant. Brain & development 0 35527075
2021 [Effect and mechanism of circular RNA BICD2 on the biological behavior of oral squamous cell carcinoma cells]. Zhonghua kou qiang yi xue za zhi = Zhonghua kouqiang yixue zazhi = Chinese journal of stomatology 0 34763405

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