Affinage

DNAJC7

DnaJ homolog subfamily C member 7 · UniProt Q99615

Length
494 aa
Mass
56.4 kDa
Annotated
2026-06-09
31 papers in source corpus 15 papers cited in narrative 15 extracted findings
Cross-family judge vs UniProt: Affinage preferred faithfulness: 6/6 claims corpus-supported (100%)

Mechanistic narrative

Synthesis pass · prose summary of the discoveries below

DNAJC7 (Tpr2) is an HSP40/J-domain co-chaperone that physically bridges the Hsp70 and Hsp90 chaperone machines, using two TPR domains to bind Hsp70 and Hsp90 simultaneously while its J domain stimulates Hsp70 ATPase activity and polypeptide binding (PMID:12853476). Unlike other co-chaperones, it induces ATP-independent dissociation of Hsp90 from chaperone-substrate complexes, mediating retrograde transfer of substrates from Hsp90 back onto Hsp70 (PMID:12853476), and it substitutes for J proteins in Hsp90-dependent maturation of clients such as the progesterone receptor and Chk1 (PMID:18620420). Through this co-chaperone activity DNAJC7 regulates the chaperoning and signaling of steroid and nuclear receptors, retaining the constitutive androstane receptor (CAR) in the cytoplasm and being itself degraded by the proteasome upon CAR activation (PMID:12853476, PMID:24789201), and acting in the nucleus where it is required for phenobarbital-induced H3K27 demethylation and RNA polymerase II recruitment at the Cyp2b10 promoter (PMID:25542016). A second major function is suppression of pathological protein aggregation: DNAJC7 binds natively folded tau via a single TPR domain recognizing a β-turn around the 275VQIINK280 amyloid motif and blocks tau aggregation and seeding in an Hsp70 ATPase-dependent manner (PMID:34504072, PMID:37387473), and it likewise suppresses TDP-43 condensate assembly and polyglutamine aggregation while promoting their disassembly/clearance (PMID:40802071, PMID:41708002). ALS-associated J-domain mutations (e.g., E425K) leave the protein structurally intact and preserve TPR-mediated holdase binding to clients, but selectively uncouple DNAJC7 from Hsp70 activation, abolishing client transfer and refolding (PMID:41531269), consistent with loss-of-function ALS mutations that increase client RBP insolubility and ablate the HSF1 stress-response pathway in motor neurons (PMID:39651147). DNAJC7 additionally stabilizes p53 by disrupting the p53–MDM2 interaction, an activity reinforced by USP19-mediated deubiquitination of DNAJC7 (PMID:23261415, PMID:42193933).

Mechanistic history

Synthesis pass · year-by-year structured walk · 12 steps
  1. 2001 Medium

    Established that DNAJC7's TPR region engages specific clients and that its J domain actively gates these interactions, hinting the protein is more than a passive scaffold.

    Evidence Yeast two-hybrid and in vitro/in vivo binding with HPD-motif mutagenesis against Rad9/Rad1/Hus1 checkpoint proteins

    PMID:11573955

    Open questions at the time
    • Functional consequence for the checkpoint clamp not resolved
    • Link to Hsp70/Hsp90 machinery not yet drawn
  2. 2003 High

    Defined the core biochemical mechanism: DNAJC7 bridges Hsp70 and Hsp90, stimulates Hsp70 ATPase, and uniquely drives retrograde substrate transfer from Hsp90 to Hsp70.

    Evidence In vitro ATPase and polypeptide-binding assays, cell-lysate GR folding assays, domain-binding studies

    PMID:12853476

    Open questions at the time
    • Physiological clients of retrograde transfer not enumerated
    • Structural basis of dual TPR/EEVD engagement not resolved
  3. 2008 High

    Showed DNAJC7 can replace J proteins in Hsp90-dependent client maturation but is distinct from Hop, clarifying its non-redundant role in steroid receptor chaperoning.

    Evidence Reciprocal Co-IP and in vitro/in vivo chaperoning assays for PR and Chk1 in HeLa cells

    PMID:18620420

    Open questions at the time
    • ATP-dependence of Hsp70 binding mechanistically unexplained
    • Range of clients beyond PR/Chk1 unknown
  4. 2012 Medium

    Identified a chaperone-independent role in tumor suppression, with DNAJC7 stabilizing p53 by displacing MDM2.

    Evidence Yeast two-hybrid, Co-IP, luciferase reporter, colony formation, and p53 half-life analysis

    PMID:23261415

    Open questions at the time
    • Direct vs chaperone-mediated mechanism of MDM2 displacement unclear
    • Whether Hsp70/Hsp90 participate not addressed
  5. 2014 High

    Connected DNAJC7 to nuclear receptor regulation, showing it retains CAR in the cytoplasm and is itself degraded upon CAR activation; in vivo it controls promoter epigenetics rather than receptor binding.

    Evidence Co-IP, ubiquitination/MG132 assays and reporters in HepG2 cells; knockout mice with ChIP at the Cyp2b10 promoter

    PMID:24789201 PMID:25542016

    Open questions at the time
    • How a cytoplasmic co-chaperone influences promoter H3K27 demethylation is mechanistically undefined
    • Identity of the ubiquitin ligase for DNAJC7 unknown
  6. 2016 Medium

    Revealed DNAJC7 as a substrate of polyglutamylation regulated by CCP6, a post-translational modification with potential as a cancer biomarker.

    Evidence Mass spectrometry, pulldown, immunohistochemistry and immunoassay in renal cell carcinoma

    PMID:26993597

    Open questions at the time
    • Functional effect of polyglutamylation on chaperone activity unknown
    • Single-lab biomarker observation
  7. 2021 High

    Defined the structural logic of DNAJC7 anti-aggregation activity: a single TPR domain recognizes the tau β-turn/amyloid motif to keep tau natively folded.

    Evidence In vitro aggregation and binding assays, domain mapping, peptide competition, cell-based aggregation

    PMID:34504072

    Open questions at the time
    • Dependence on Hsp70 not yet tested in this study
    • Whether TPR recognition generalizes to other amyloid clients unknown
  8. 2022 Medium

    Extended the bridging mechanism to antiviral defense, with DNAJC7 linking Hsp70 to a viral Hexon protein for autophagic degradation.

    Evidence LC-MS/MS interaction screen, Co-IP, domain deletion, autophagy inhibitor experiments

    PMID:35852354

    Open questions at the time
    • Generality beyond FAdV-4 unknown
    • Mechanism of Hsp70-to-autophagy handoff not detailed
  9. 2023 High

    Demonstrated that tau protection is specifically Hsp70-dependent: among all JDPs, only DNAJC7 loss impairs aggregate clearance, and both J-domain and substrate-binding mutations abolish protection.

    Evidence Proteomics, CRISPR knockout of all JDPs, tau seeding assays, colocalization imaging, J-domain mutagenesis

    PMID:37387473

    Open questions at the time
    • In vivo relevance in neurons not established here
    • Quantitative contribution of holdase vs Hsp70-coupled clearance unresolved
  10. 2024 Medium

    Linked DNAJC7 loss-of-function to ALS pathology in human motor neurons, showing client RBP insolubility and an ablated HSF1 stress response rescuable by exogenous HSF1.

    Evidence MS interactome, iPSC-derived motor neurons (R156X), solubility fractionation, HSF1 rescue, RNA-seq (preprint)

    PMID:39651147

    Open questions at the time
    • Preprint, single lab
    • Mechanism by which DNAJC7 haploinsufficiency disables HSF1 signaling unclear
  11. 2025 Medium

    Broadened the anti-aggregation role to TDP-43 condensate dynamics and identified a USP19 deubiquitination axis that stabilizes DNAJC7 to support p53.

    Evidence TDP-43 condensate assays with arsenite stress and zebrafish dnajc7 knockdown; USP19-DnaJC7 Co-IP, deubiquitination and p53 stability assays

    PMID:40802071 PMID:42193933

    Open questions at the time
    • Direct vs Hsp70-mediated control of TDP-43 condensates not separated
    • How USP19 regulation intersects with chaperone activity unknown
  12. 2026 High

    Resolved the molecular defect of ALS mutation E425K and confirmed broad anti-aggregation function: the mutant preserves structure and TPR-mediated holdase activity but selectively uncouples from Hsp70, while DNAJC7 also suppresses polyQ aggregation.

    Evidence NMR, in vitro binding and Hsp70 ATPase assays, TDP-43 aggregation assays; genome-wide CRISPRi chaperone screen with KD/OE polyQ aggregation validation

    PMID:41531269 PMID:41708002

    Open questions at the time
    • In vivo demonstration that holdase activity alone is insufficient in neurons lacking
    • Whether other ALS mutations act by the same uncoupling mechanism not exhaustively tested

Open questions

Synthesis pass · forward-looking unresolved questions
  • How DNAJC7's cytoplasmic co-chaperone activity is mechanistically coupled to its nuclear functions (Cyp2b10 promoter epigenetics, p53 stabilization) and how client specificity is partitioned between TPR holdase and Hsp70-coupled refolding across its many clients remain unresolved.
  • No unified model linking chaperone bridging to transcriptional/epigenetic roles
  • Structural basis of differential client recognition by TPR domains incomplete

Mechanism profile

Synthesis pass · controlled-vocabulary classification · explore literature graph →
Molecular activity
GO:0044183 protein folding chaperone 3 GO:0060090 molecular adaptor activity 3 GO:0098772 molecular function regulator activity 3 GO:0140096 catalytic activity, acting on a protein 3 GO:0140657 ATP-dependent activity 3
Localization
GO:0005829 cytosol 3 GO:0005634 nucleus 2
Pathway
R-HSA-392499 Metabolism of proteins 4 R-HSA-74160 Gene expression (Transcription) 2 R-HSA-8953897 Cellular responses to stimuli 2
Complex memberships
Hsp70-Hsp90 chaperone complex

Evidence

Reading pass · 15 per-paper findings extracted from the source corpus
Year Finding Method Journal Conf PMIDs
2003 Tpr2 (DNAJC7) contains two TPR domains that bind Hsp70 and Hsp90 simultaneously, and a J domain that stimulates Hsp70 ATPase activity and polypeptide binding. Unlike other co-chaperones, Tpr2 induces ATP-independent dissociation of Hsp90 (but not Hsp70) from chaperone-substrate complexes, mediating retrograde transfer of substrates from Hsp90 back onto Hsp70. Excess Tpr2 inhibits Hsp90-dependent folding of the glucocorticoid receptor (GR) in cell lysates. In vitro biochemical assays (ATPase stimulation, polypeptide binding), cell lysate GR folding assays, domain-binding studies, overexpression/knockdown in vivo The EMBO journal High 12853476
2008 Tpr2 (DNAJC7) associates with Hsp90 and Hsp70 complexes that also contain the progesterone receptor (PR); it can bind Hsp70 and Hsp90 simultaneously, but unlike Hop, its binding to Hsp70 in the presence of Hsp90 is ATP-dependent. Tpr2 cannot replace Hop in Hsp90 chaperoning in vitro or in vivo. Tpr2 replaces type I and II J proteins in Hsp90-dependent chaperoning of the PR and Chk1 kinase and promotes accumulation of Hsp70 in PR heterocomplexes in the presence of Hsp90. Immunoprecipitation, pulldown, in vitro and in vivo chaperoning assays, knockdown/overexpression in HeLa cells Biochemistry High 18620420
2001 Tpr2 (DNAJC7) binds Rad9, Rad1, and Hus1 (checkpoint clamp proteins) through its N-terminal TPR region. The J domain of Tpr2 negatively regulates its interaction with Rad9: deletion of the J domain or mutation of the conserved HPD motif greatly enhances Tpr2-Rad9 binding. Following heat-shock or UV treatment, Rad9 transiently dissociates from Tpr2 in a J-domain-dependent manner. The J domain also modulates the cellular localization of both Tpr2 and Rad9. Yeast two-hybrid screening, in vivo and in vitro binding assays, J-domain point mutation (HPD motif), cellular localization analysis Biochemical and biophysical research communications Medium 11573955
2012 DnaJC7/TPR2 binds p53 through its central DNA-binding domain (identified by yeast two-hybrid), confirmed by co-immunoprecipitation in mammalian cells. DnaJC7 enhances p53-dependent transcriptional and growth-suppressive activity, extends p53 half-life, and reduces the amount of p53/MDM2 complex, suggesting DnaJC7 dissociates MDM2 from p53 to stabilize p53. Yeast two-hybrid screening, co-immunoprecipitation, luciferase reporter assay, colony formation assay, pulse-chase/half-life analysis Biochemical and biophysical research communications Medium 23261415
2014 CCRP/DNAJC7 interacts with HSP70 (co-immunoprecipitation in HepG2 cells) and retains nuclear receptor CAR in the cytoplasm. DNAJC7 is ubiquitinated and degraded by the proteasome upon CAR activation; this ubiquitination of DNAJC7 (but not CAR itself) is increased by TCPOBOP in the presence of MG132. HSP70 induction by heat shock also increases cytoplasmic CAR levels and attenuates CAR transcriptional activation, phenocopying proteasome inhibition. Co-immunoprecipitation, proteasome inhibitor (MG132) treatment, ubiquitination assay, luciferase reporter assay, heat shock experiments in HepG2 cells PloS one Medium 24789201
2014 CCRP/DNAJC7 knockout mice show increased nuclear CAR accumulation after phenobarbital treatment but paradoxically attenuated Cyp2b10 gene activation. ChIP assays reveal that DNAJC7 is required for phenobarbital-induced de-methylation of H3K27 on the Cyp2b10 promoter and for RNA polymerase II recruitment, but not for CAR/RXRα binding to the promoter. DNAJC7 KO males also develop steatotic livers and altered cholesterol levels upon fasting. Knockout mouse model, ChIP assays (H3K27 methylation, RNA Pol II, CAR/RXRα), immunoblotting, gene expression analysis PloS one High 25542016
2016 Mass spectrometric and pulldown analysis identified DNAJC7 as a substrate of cytosolic carboxypeptidase 6 (CCP6). When CCP6 is reduced (as in RCC), DNAJC7 accumulates in a polyglutamylated form (polyE-DNAJC7) detectable in serum. Mass spectrometry, pulldown assay, immunohistochemistry, Western blot, electrochemiluminescence immunoassay Oncotarget Medium 26993597
2021 DnaJC7 binds preferentially to natively folded wild-type tau (not aggregated conformers) using a single TPR domain that recognizes a β-turn structural element containing the 275VQIINK280 amyloid motif. Disease-associated tau mutants that disrupt this β-turn reduce DnaJC7 binding affinity. DnaJC7 efficiently suppresses tau aggregation in vitro and in cells. In vitro aggregation assays, binding affinity measurements, domain mapping (single TPR domain), peptide competition assays, cell-based aggregation assays Nature communications High 34504072
2023 DnaJC7 co-purifies with insoluble tau and colocalizes with intracellular tau aggregates. Knockout of DnaJC7 (but not other JDPs) specifically decreases aggregate clearance and increases intracellular tau seeding. This protective activity depends on the ability of the DnaJC7 J domain to stimulate Hsp70 ATPase activity; J-domain mutations blocking Hsp70 interaction abolish protection. Disease-associated mutations in both the J domain and the substrate-binding site of DnaJC7 also abolish protective activity. Proteomics (co-purification with insoluble tau), CRISPR knockout of all JDPs individually, tau seeding assay, co-localization imaging, J-domain mutagenesis (HPD motif and ALS variants) eLife High 37387473
2022 DnaJC7 acts as a bridge between Hsp70 and viral Hexon protein: the J domain of DnaJC7 is required for inhibiting FAdV-4 replication, and DnaJC7 mediates the indirect interaction between Hsp70 and Hexon. Hsp70 subsequently suppresses Hexon through the autophagy pathway to restrict viral replication. LC-MS/MS protein interaction screen, Co-IP, domain deletion analysis (J domain of DnaJC7, NBD of Hsp70), overexpression and autophagy inhibitor (chloroquine) experiments Journal of virology Medium 35852354
2025 DNAJC7 knockdown impairs disassembly of TDP-43 condensates following arsenite-induced stress, while DNAJC7 overexpression suppresses assembly and promotes disassembly of arsenite-induced TDP-43 condensates. In a zebrafish ALS model, dnajc7 knockdown increases TDP-43 aggregation in motor neurons and reduces survival. Cell-based TDP-43 condensate assay with arsenite stress, DNAJC7 KD and OE, zebrafish dnajc7 morpholino knockdown, immunohistochemistry, RNA sequencing Acta neuropathologica Medium 40802071
2026 The ALS-associated E425K mutation in the DNAJC7 J domain does not alter protein structure (confirmed by NMR) but specifically disrupts J-domain–Hsp70 interaction, uncoupling DNAJC7 from Hsp70 activation. A second Hsp70-binding interface exists in the TPR domains (binding the C-terminal EEVD motif of Hsp70), which is preserved in E425K but cannot compensate for loss of J-domain function. The TPR domains of DNAJC7 directly bind TDP-43 and prevent its aggregation (holdase activity retained in E425K), but the mutant fails to support client transfer to Hsp70 and subsequent Hsp70-mediated substrate refolding. NMR spectroscopy (structural and interaction analysis), in vitro binding assays, Hsp70 ATPase activation assay, TDP-43 aggregation assay The FEBS journal High 41531269
2024 The DNAJC7 interactome in human motor neurons (iPSC-derived) is enriched for RNA-binding proteins (RBPs) and stress-response chaperones (mass spectrometry). The ALS-associated R156X loss-of-function mutation causes increased insolubility of client RBP HNRNPU and associated RNA metabolism alterations. DNAJC7 haploinsufficiency renders motor neurons susceptible to proteotoxic stress and cell death through an ablated HSF1 stress-response pathway; exogenous HSF1 expression rescues sensitivity to proteotoxic stress in DNAJC7-mutant motor neurons. Mass spectrometry interactome, iPSC-derived motor neurons with ALS mutation (R156X), protein solubility fractionation, HSF1 rescue experiment, RNA sequencing bioRxivpreprint Medium 39651147
2025 USP19 deubiquitinates DnaJC7, increasing its protein stability. Upregulation of both USP19 and DnaJC7 disrupts the p53-MDM2 interaction; knockdown of USP19 and DnaJC7 decreases p53 expression following cisplatin treatment, indicating that the USP19-DnaJC7 axis stabilizes p53. Co-immunoprecipitation (USP19-DnaJC7 interaction), deubiquitination assay, knockdown experiments, p53 stability and MDM2 interaction assays in cisplatin-treated cells Cells Medium 42193933
2026 DNAJC7 is a potent suppressor of polyglutamine (polyQ) aggregation, identified in a genome-wide CRISPRi screen of all chaperones. Physical interaction between DNAJC7 and polyQ-expanded protein was established. DNAJC7 knockdown specifically increased polyQ aggregation, while overexpressed DNAJC7 colocalized with both polyQ and polyG aggregates and reduced their aggregation. Direct knockdown of DNAJC7 did not affect polyG aggregation in the baseline model (negative result for polyG). FRET-based aggregation reporter, CRISPRi screen (all chaperones), KD validation, overexpression colocalization in human cells The Journal of biological chemistry Medium 41708002

Source papers

Stage 0 corpus · 31 papers · ranked by NIH iCite citations
Year Title Journal Citations PMID
2019 Exome sequencing in amyotrophic lateral sclerosis implicates a novel gene, DNAJC7, encoding a heat-shock protein. Nature neuroscience 110 31768050
2003 Cofactor Tpr2 combines two TPR domains and a J domain to regulate the Hsp70/Hsp90 chaperone system. The EMBO journal 107 12853476
2018 ABI5-BINDING PROTEIN2 Coordinates CONSTANS to Delay Flowering by Recruiting the Transcriptional Corepressor TPR2. Plant physiology 42 30514725
2008 Role of the cochaperone Tpr2 in Hsp90 chaperoning. Biochemistry 40 18620420
2021 DnaJC7 binds natively folded structural elements in tau to inhibit amyloid formation. Nature communications 37 34504072
2014 Coordinated regulation of nuclear receptor CAR by CCRP/DNAJC7, HSP70 and the ubiquitin-proteasome system. PloS one 27 24789201
2022 DnaJC7 in Amyotrophic Lateral Sclerosis. International journal of molecular sciences 24 35456894
2014 The roles of co-chaperone CCRP/DNAJC7 in Cyp2b10 gene activation and steatosis development in mouse livers. PloS one 21 25542016
2023 DnaJC7 specifically regulates tau seeding. eLife 18 37387473
2012 Co-chaperon DnaJC7/TPR2 enhances p53 stability and activity through blocking the complex formation between p53 and MDM2. Biochemical and biophysical research communications 16 23261415
2001 The J domain of Tpr2 regulates its interaction with the proapoptotic and cell-cycle checkpoint protein, Rad9. Biochemical and biophysical research communications 16 11573955
2022 Hsp70 Inhibits the Replication of Fowl Adenovirus Serotype 4 by Suppressing Viral Hexon with the Assistance of DnaJC7. Journal of virology 15 35852354
2020 Mutations of DNAJC7 are rare in Chinese amyotrophic lateral sclerosis patients. Amyotrophic lateral sclerosis & frontotemporal degeneration 13 32897108
2024 Activation of stress-response genes by retrograde signaling-mediated destabilization of nuclear importin IMPα-9 and its interactor TPR2. Molecular plant 11 38693693
2020 A Novel Potentially Pathogenic Rare Variant in the DNAJC7 Gene Identified in Amyotrophic Lateral Sclerosis Patients From Mainland China. Frontiers in genetics 11 33193563
2021 Validation of the pathogenic role of rare DNAJC7 variants in Chinese patients with amyotrophic lateral sclerosis. Neurobiology of aging 10 34233860
2021 Mutation screening of the DNAJC7 gene in Japanese patients with sporadic amyotrophic lateral sclerosis. Neurobiology of aging 10 35039179
2016 Reduced cytosolic carboxypeptidase 6 (CCP6) level leads to accumulation of serum polyglutamylated DNAJC7 protein: A potential biomarker for renal cell carcinoma early detection. Oncotarget 8 26993597
2008 Involvement of the TPR2 subdomain movement in the activities of phi29 DNA polymerase. Nucleic acids research 8 19033368
2020 Publisher Correction: Exome sequencing in amyotrophic lateral sclerosis implicates a novel gene, DNAJC7, encoding a heat-shock protein. Nature neuroscience 4 31857710
2025 Biallelic variants in DNAJC7 cause familial amyotrophic lateral sclerosis with the TDP-43 pathology. Acta neuropathologica 3 40802071
2025 Increased expression of DNAJC7 promotes the progression of hepatocellular carcinoma by influencing the cell cycle and immune microenvironment. Journal of cancer research and clinical oncology 2 40312488
2023 Rare DNAJC7 Variants May Play a Minor Role in Chinese Patients with ALS. Molecular neurobiology 2 37870677
2026 The ALS-associated E425K mutation uncouples DNAJC7 from the Hsp70 chaperone cycle. The FEBS journal 1 41531269
2024 In Vivo-Matured Oocyte Resists Post-Ovulatory Aging through the Hub Genes DDX18 and DNAJC7 in Pigs. Antioxidants (Basel, Switzerland) 1 39061935
2026 The Hsp40 cochaperone DNAJC7 regulates polyglutamine aggregation and exhibits context-dependent effects on polyglycine aggregation. The Journal of biological chemistry 0 41708002
2026 Holding but not folding: How a single charge flip uncouples the DNAJC7-Hsp70 relay in amyotrophic lateral sclerosis. The FEBS journal 0 41728998
2026 The USP19-DnaJC7 Axis Stabilizes p53 in Cisplatin-Treated Epithelial Ovarian Cancer. Cells 0 42193933
2025 The Hsp40 co-chaperone DNAJC7 modifies polyglutamine but not polyglycine aggregation. bioRxiv : the preprint server for biology 0 40832276
2024 The ALS-associated co-chaperone DNAJC7 mediates neuroprotection against proteotoxic stress by modulating HSF1 activity. bioRxiv : the preprint server for biology 0 39651147
2023 DnaJC7 specifically regulates tau seeding. bioRxiv : the preprint server for biology 0 36993367

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