Affinage

MYSM1

Deubiquitinase MYSM1 · UniProt Q5VVJ2

Length
828 aa
Mass
95.0 kDa
Annotated
2026-06-10
58 papers in source corpus 32 papers cited in narrative 31 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

MYSM1 is a JAMM/MPN-domain metalloprotease deubiquitinase whose catalytic activity is universally required for its roles across hematopoiesis, immune cell development, and broader mammalian physiology, as a catalytically dead D660N knock-in fully phenocopies the null (PMID:36611064). In the nucleus, MYSM1 removes monoubiquitin from histone H2A-K119 to derepress lineage-specifying loci, coordinating histone modifications and recruiting transcription factors to activate EBF1, Gfi1, Flt3, Pax5, and Id4 expression, thereby driving B-cell commitment, HSC quiescence, dendritic-cell and NK-cell development, and neural stem cell homeostasis (PMID:22184403, PMID:22169041, PMID:24014243, PMID:25217698, PMID:26348977, PMID:38342917). A central consequence of MYSM1 loss is aberrant p53 activation: MYSM1 associates with p53 and restrains transcription of its proapoptotic target PUMA, and genetic ablation of p53 fully rescues the hematopoietic defects of Mysm1 deficiency, identifying p53/PUMA-driven apoptosis as the principal cause of those phenotypes (PMID:25710881, PMID:25613381, PMID:26768662). In the cytoplasm, MYSM1 accumulates upon microbial or DNA-virus stimulation and uses its SWIRM domain to engage TRAF3/TRAF6 and STING and its MPN domain to strip K63-linked polyubiquitin chains, terminating PRR-driven pro-inflammatory and type I interferon signaling (PMID:26474655, PMID:33086059). Beyond histones, MYSM1 deubiquitinates and regulates the stability or activity of non-histone substrates including STAT1, PARP1, TRIM21, and RIPK2 in cancer, cardiac, and inflammatory contexts (PMID:38177530, PMID:39695708, PMID:39746943, PMID:39897566, PMID:39907013). A homozygous catalytic-site mutation causes human immunodeficiency with absent B cells and T-cell lymphopenia, and loss-of-function additionally produces prolonged DNA damage response signaling without affecting break repair (PMID:26220525, PMID:38065233).

Mechanistic history

Synthesis pass · year-by-year structured walk · 15 steps
  1. 2011 High

    Established MYSM1's core biochemical identity as an H2A-K119 deubiquitinase and its physiological requirement in hematopoiesis, answering what enzymatic activity MYSM1 carries and why its loss matters.

    Evidence Targeted Mysm1 mouse line with bone marrow transplantation showing cell-intrinsic HSC and lineage defects, elevated ROS, γH2AX, and p53

    PMID:22184403

    Open questions at the time
    • Did not resolve whether H2A-DUB activity or non-histone activity drives each phenotype
    • Mechanism linking DUB loss to p53 elevation undefined
  2. 2011 High

    Defined how MYSM1 controls lineage commitment at the molecular level by showing it derepresses a specific gene through chromatin remodeling and transcription factor recruitment.

    Evidence Mysm1-deficient mice with ChIP at the EBF1 locus and rescue experiments

    PMID:22169041

    Open questions at the time
    • Did not map which histone marks are direct substrates versus secondary
    • Generality of the locus-specific recruitment mechanism untested at this stage
  3. 2013 Medium

    Generalized the locus-specific activator model to HSC quiescence and NK maturation, showing MYSM1 directs transcription factor recruitment (Gata2/Runx1, NFIL3) at defined enhancers.

    Evidence Mysm1(-/-) mice with ChIP at Gfi1 and ID2 loci plus co-immunoprecipitation

    PMID:24014243 PMID:24062447

    Open questions at the time
    • Whether TF recruitment is a direct consequence of H2A deubiquitination not established
    • Single-lab Co-IP for NFIL3 interaction
  4. 2014 Medium

    Extended the chromatin-activator role to myeloid/dendritic lineage by showing MYSM1 enables PU.1 recruitment selectively at Flt3.

    Evidence Mysm1(-/-) mice with ChIP at Flt3 promoter and in vitro DC differentiation assays

    PMID:25217698

    Open questions at the time
    • Basis for locus selectivity (Flt3 but not GM-CSF-α/M-CSFR) unexplained
    • Single lab
  5. 2015 High

    Identified p53 as the central downstream effector of MYSM1 loss and PUMA as the non-redundant apoptotic mediator, explaining why diverse hematopoietic defects share one cause.

    Evidence Mysm1(-/-)p53(-/-) and Mysm1(-/-)Puma(-/-) double-knockout mice with Co-IP and ChIP at PUMA/p21 promoters

    PMID:25613381 PMID:25710881 PMID:26768662

    Open questions at the time
    • Mechanism by which MYSM1 loss elevates p53 protein not fully resolved
    • Whether MYSM1-p53 promoter antagonism requires DUB activity at this stage untested
  6. 2015 Medium

    Confirmed disease causality in humans, linking a catalytic-site mutation to immunodeficiency and validating the catalytic domain's requirement via natural reversion.

    Evidence Whole-exome sequencing and immunophenotyping of a patient with in vivo HSC reversion

    PMID:26220525

    Open questions at the time
    • Single case
    • Molecular consequence of the missense allele on substrate spectrum not characterized
  7. 2015 High

    Revealed a distinct cytoplasmic, immune-regulatory function in which MYSM1 uses separable domains to engage and inactivate innate signaling adaptors, broadening MYSM1 beyond a nuclear histone enzyme.

    Evidence Co-IP of MYSM1 with TRAF3/TRAF6, SWIRM and MPN domain mutagenesis, and Mysm1-deficient mice challenged with microbial stimuli

    PMID:26474655

    Open questions at the time
    • What triggers cytoplasmic relocalization on stimulation not defined
    • K63-chain editing specificity versus other DUBs unaddressed
  8. 2015 Medium

    Added further hematopoietic loci (Pax5) and an IRF2/IRF8→p53 regulatory link, sharpening the locus-specific activator network upstream of the p53 axis.

    Evidence Mysm1-deficient mice with ChIP at Pax5 and promoter reporter assays for IRF2/IRF8

    PMID:26348977 PMID:27277682

    Open questions at the time
    • Direct versus indirect regulation of IRF promoters unresolved
    • Single lab
  9. 2016 Medium

    Distinguished the cell-intrinsic source of MYSM1 function, establishing that hematopoietic-progenitor MYSM1, not niche expression, drives HSC maintenance.

    Evidence Bone marrow chimeras with non-hematopoietic Mysm1 ablation; MYSM1/miR-150/FLT3 axis defined by ChIP

    PMID:27590507 PMID:27833034

    Open questions at the time
    • Negative niche result does not exclude subtle non-hematopoietic roles
    • Single lab
  10. 2020 Medium

    Extended cytoplasmic immune regulation to the cGAS-STING axis and localized MYSM1 to DNA damage sites, hinting at a DDR role.

    Evidence Co-IP and K63-ubiquitination assays for STING; γH2AX co-localization and MS interactome after etoposide

    PMID:32466590 PMID:33086059

    Open questions at the time
    • DDR interactome lacks functional validation (Low confidence)
    • Whether STING editing uses the same domains as TRAF editing not tested directly
  11. 2021 Medium

    Showed MYSM1 acts as an epigenetic regulator in cancer via H2AK119ub1 removal and links its loss to oncogenic signaling and cMYC-driven translation.

    Evidence ChIP/H2AK119ub1 ChIP at miR-200/CDH1 in CRC and cMYC lymphoma genetic models with ribosomal gene/protein synthesis readouts

    PMID:34114734 PMID:34706761

    Open questions at the time
    • Context-dependent tumor-suppressor versus oncogenic roles not unified
    • Single lab per finding
  12. 2023 High

    Demonstrated that DUB catalytic activity is required for essentially all MYSM1 functions in vivo, unifying histone and non-histone activities under a single enzymatic requirement.

    Evidence CRISPR Mysm1 D660N catalytic-dead knock-in and inducible Mysm1fl/DN mice with comprehensive hematopoietic/immune profiling

    PMID:36611064

    Open questions at the time
    • Does not separate which substrates are rate-limiting for which phenotype
    • Single lab
  13. 2023 Medium

    Established non-histone deubiquitination as a bona fide MYSM1 activity by showing direct ERα deubiquitination/stabilization and a DDR-termination function, plus a druggable MPN active site.

    Evidence Co-IP, ubiquitination assays, ChIP, and Imatinib docking for ERα; patient and U2OS cells with γH2AX kinetics for DDR termination

    PMID:38065233 PMID:38177530

    Open questions at the time
    • DDR-termination substrate(s) not identified
    • Imatinib-MPN interaction in vivo relevance untested
  14. 2024 Medium

    Refined the developmental requirement to progenitor stages and extended epigenetic control to neural stem cells via Id4, while broadening cardiac non-histone substrates (TRIM21).

    Evidence Conditional DC models with Mysm1DN; Nestin-Cre KO with CUT&Tag and Id4 rescue; Co-IP/MS for TRIM21 with MPN domain mapping

    PMID:38316548 PMID:38342917 PMID:39695708

    Open questions at the time
    • Substrate hierarchy across tissues unresolved
    • Single lab per substrate
  15. 2025 Medium

    Expanded the non-histone substrate repertoire (STAT1, PARP1, RIPK2, TRAF2/3) defining tissue-specific deubiquitination outcomes in cardiac, inflammatory, and oncogenic settings.

    Evidence Co-IP/MS, site-specific ubiquitination assays, MPN/SWIRM domain mapping, and AAV9 or KO in vivo disease models

    PMID:39746943 PMID:39897566 PMID:39907013 PMID:41162619

    Open questions at the time
    • Determinants of substrate selectivity across contexts unknown
    • Each substrate validated in a single lab/disease model

Open questions

Synthesis pass · forward-looking unresolved questions
  • How MYSM1 achieves locus- and substrate-selectivity—choosing among histone H2A, innate-immune adaptors, and diverse non-histone proteins in a context-dependent manner—remains unresolved.
  • No structural basis for substrate discrimination defined
  • No unifying model for nuclear versus cytoplasmic partitioning
  • DDR-termination substrate uncharacterized

Mechanism profile

Synthesis pass · controlled-vocabulary classification · explore literature graph →
Molecular activity
GO:0140096 catalytic activity, acting on a protein 9 GO:0140110 transcription regulator activity 6 GO:0016787 hydrolase activity 3 GO:0042393 histone binding 3
Localization
GO:0005634 nucleus 4 GO:0000228 nuclear chromosome 2 GO:0005829 cytosol 2
Pathway
R-HSA-74160 Gene expression (Transcription) 5 R-HSA-168256 Immune System 3 R-HSA-4839726 Chromatin organization 3 R-HSA-5357801 Programmed Cell Death 2 R-HSA-73894 DNA Repair 1
Partners
PARP1RIPK2STAT1STINGTP53TRAF3TRAF6TRIM21

Evidence

Reading pass · 31 per-paper findings extracted from the source corpus
Year Finding Method Journal Conf PMIDs
2011 MYSM1 functions as a histone H2A deubiquitinase (H2A-DUB) that removes monoubiquitin from H2A-K119, and its loss in mice causes defects in hematopoiesis including lymphopenia, anemia, and impaired HSC function associated with elevated ROS, γH2AX DNA damage marker, and p53 protein in hematopoietic progenitors. Mysm1(tm1a/tm1a) targeted mouse line characterization; bone marrow transplantation to establish cell-intrinsic requirement Blood High 22184403
2011 MYSM1 derepresses EBF1 transcription in B cell progenitors by orchestrating histone modifications and transcription factor recruitment to the EBF1 locus, establishing an essential intrinsic role in early B cell commitment. Mysm1-deficient mice; chromatin immunoprecipitation (ChIP) at EBF1 locus; rescue experiments Immunity High 22169041
2013 MYSM1 directly associates with the Gfi1 enhancer element in HSCs and promotes Gfi1 transcription by modulating histone modifications and directing recruitment of transcription factors Gata2 and Runx1 to the Gfi1 locus; loss of Mysm1 drives HSCs from quiescence into rapid cycling and increases apoptotic rate. Mysm1(-/-) mice; ChIP at Gfi1 locus; transcription factor recruitment assays Blood High 24014243
2013 MYSM1 interacts with transcription factor NFIL3/E4BP4 and its recruitment to the ID2 locus is dependent on MYSM1; MYSM1 maintains active chromatin at the ID2 locus to promote NK cell maturation. Mysm1(-/-) mice; co-immunoprecipitation; ChIP at ID2 locus Proceedings of the National Academy of Sciences of the United States of America Medium 24062447
2014 MYSM1 derepresses transcription of the Flt3 gene by directing histone modifications at the Flt3 promoter and enabling PU.1 recruitment specifically at the Flt3 locus (but not at GM-CSF-α or M-CSFR loci), thereby controlling dendritic cell development from common myeloid progenitors. Mysm1(-/-) mice; ChIP at Flt3 promoter; in vitro DC differentiation assays with Flt3L or GM-CSF Blood Medium 25217698
2015 MYSM1 deficiency results in p53 protein elevation in hematopoietic cells; genetic double-knockout (Mysm1(-/-)p53(-/-)) fully rescues all developmental and hematopoietic defects including lymphopoiesis and HSC numbers/function, establishing p53 activation as the driving mechanism for hematopoietic abnormalities in Mysm1 deficiency. Mysm1(-/-)p53(-/-) double-knockout mice; bone marrow transplantation; flow cytometric analysis of hematopoietic progenitors Blood High 25613381 25710881
2015 A homozygous MYSM1 missense mutation affecting the catalytic site within the JAMM/MPN deubiquitinase domain causes human immunodeficiency with absent B lymphocytes and T-cell lymphopenia; in vivo genetic reversion of the mutation in a hematopoietic stem cell restored normal immunohematopoietic phenotype. Whole-exome sequencing; genome-wide homozygosity mapping; Sanger sequencing; immunophenotyping The Journal of allergy and clinical immunology Medium 26220525
2015 MYSM1 protein associates with p53 and co-localizes to promoters of p53-target genes Bbc3/PUMA and Cdkn1a/p21, antagonizing their p53-driven expression by modulating H3K27ac and H3K4me3 histone modifications and reducing p53 recruitment; PUMA (but not p21) is the essential non-redundant effector of p53-induced MPP apoptosis in Mysm1 deficiency. Co-IP of MYSM1 with p53; ChIP at PUMA and p21 promoters; Mysm1(-/-)Puma(-/-) double-knockout mice; transcriptome analysis Cell death and differentiation High 26768662
2015 MYSM1 accumulates in the cytoplasm in response to microbial stimuli where it interacts with TRAF3 and TRAF6 via its SWIRM domain and removes K63-linked polyubiquitin chains via its metalloproteinase/MPN domain to inactivate these complexes and terminate PRR-driven pro-inflammatory and type I interferon responses. Co-immunoprecipitation of MYSM1 with TRAF3/TRAF6; domain mutagenesis (SWIRM and MPN domain mutants); Mysm1-deficient mice challenged with microbial stimuli; cytokine/IFN quantification Immunity High 26474655
2015 MYSM1 is a transcriptional activator of Pax5 (a repressor of plasma cell differentiation) by facilitating key transcription factor recruitment and coordinating histone modifications at the Pax5 loci, thereby intrinsically repressing plasma cell differentiation and antibody production. Mysm1-deficient mice; ChIP at Pax5 locus; B cell differentiation assays; antibody quantification Scientific reports Medium 26348977
2016 MYSM1 is required for expression of IRF2 and IRF8 in HSCs; lower IRF2/IRF8 levels in Mysm1(-/-) HSCs lead to enhanced p53 transcription, contributing to HSC quiescence defects; Mysm1 enhances function of the IRF2 and IRF8 promoters. Mysm1(-/-) mice; promoter reporter assays for IRF2 and IRF8; gene expression analysis Cell death & disease Medium 27277682
2016 MYSM1 recruits c-Myc to the promoter of miR-150 and stimulates its transcription; miR-150 in turn decreases FLT3 expression in B1a cells, defining a MYSM1/miR-150/FLT3 pathway that inhibits B1a cell proliferation. ChIP demonstrating c-Myc recruitment to miR-150 promoter in MYSM1-dependent manner; Mysm1-deficient mice; miR-150/FLT3 expression analysis Oncotarget Medium 27590507
2016 MYSM1 expression in the bone marrow niche (non-hematopoietic cells) is dispensable for HSC maintenance and hematopoietic homeostasis; selective deletion of Mysm1 in non-hematopoietic cells did not affect HSC number, viability, or hematopoietic output. Bone marrow chimeras with wild-type donor into Mysm1fl/flTg.CreERT2 recipients; tamoxifen-induced Mysm1 ablation in non-hematopoietic cells; flow cytometric analysis Experimental hematology Medium 27833034
2017 MYSM1 loss in CD8+ T cells leads to a hyperactivated CD8+ T-cell state with increased apoptosis and preferential p53 upregulation, resulting in reduced CD8+ T-cell numbers and impaired CD8+ T-cell-dependent pathology in experimental cerebral malaria. Conditional Mysm1 knockout in T cells (Mysm1fl/flTg.CD8-cre model); flow cytometry; cytokine production assays; cerebral malaria model Immunology Medium 28066899
2019 MYSM1 physically binds the androgen receptor (AR) without increasing AR expression, and MYSM1 knockdown results in activation of Akt/c-Raf/GSK-3β signaling in castration-resistant prostate cancer cells. Co-immunoprecipitation assay of MYSM1 with AR; MYSM1 knockdown; western blot for Akt/c-Raf/GSK-3β pathway activation Aging Low 31761786
2020 MYSM1 interacts with STING and cleaves STING K63-linked ubiquitination to suppress cGAS-STING signaling; MYSM1 expression is induced upon DNA virus infection and intracellular DNA stimulation. Co-immunoprecipitation of MYSM1 with STING; ubiquitination assay showing removal of K63-linked ubiquitin from STING; Mysm1-deficient mice challenged with DNA virus Cell reports Medium 33086059
2020 MYSM1 colocalizes with γH2AX foci upon DNA double-strand break induction, and mass spectrometry-based proteomics identified an interacting network of DNA damage and replication proteins (enriched after etoposide treatment) as MYSM1 binding partners. Co-localization of MYSM1 with γH2AX by immunofluorescence; affinity pulldown followed by mass spectrometry in 293T cells treated with etoposide International journal of molecular sciences Low 32466590
2021 MYSM1 directly binds the promoter region of miR-200/CDH1 in colorectal cancer cells, impairs repressive H2AK119ub1 enrichment at this locus, and epigenetically activates miR-200/CDH1 expression; MYSM1 loss activates PI3K/AKT signaling. ChIP at miR-200/CDH1 promoter; H2AK119ub1 ChIP; gain- and loss-of-function in CRC cell lines; in vivo xenograft models Journal of experimental & clinical cancer research : CR Medium 34706761
2021 MYSM1 loss represses ribosomal protein gene expression and protein synthesis in cMYC-driven B cell lymphoma, strongly inhibiting cMYC oncogenic activity and protecting against B cell lymphoma onset and progression. cMYC-driven B cell lymphoma mouse models crossed with Mysm1-deficient mice; ribosomal protein gene expression analysis; protein synthesis measurement Journal of cellular and molecular medicine Medium 34114734
2022 MYSM1 induces apoptosis in TNBC cells and sensitizes them to cisplatin; MYSM1 overexpression increases cisplatin-induced apoptosis via RSK3 inactivation and consequent decreased phosphorylation of BAD at Ser112. MYSM1 overexpression and knockdown in TNBC cell lines; apoptosis assays; western blot for RSK3 and phospho-BAD(Ser112) Cell death discovery Low 35217648
2023 MYSM1 loss-of-function (patient splice variant causing absent protein) results in persistent DNA damage foci and prolonged DNA damage response signaling after DSBs from immunoglobulin recombination and ionizing radiation, without altering DSB generation or repair itself; MYSM1 specifically regulates termination of DNA damage responses. MYSM1-deficient pre-B cells and U2OS cells; γH2AX focus assays; irradiation-induced DSB repair kinetics; patient cells with constitutive γH2AX The Journal of allergy and clinical immunology Medium 38065233
2023 MYSM1 acts as a co-activator of ERα in breast cancer, maintaining ERα stability via direct ERα deubiquitination and regulating histone modifications on cis-regulatory elements of ERα-target genes to facilitate chromatin decondensation; small molecule Imatinib was identified to interact with the MPN catalytic domain of MYSM1. Co-immunoprecipitation of MYSM1 with ERα; ubiquitination assays; ChIP; xenograft models; virtual screening/docking for Imatinib-MPN interaction EMBO molecular medicine Medium 38177530
2023 MYSM1 DUB catalytic activity (tested via D660N point mutation that renders protein catalytically inactive) is universally required for its functions in hematopoiesis, leukocyte development, and other aspects of mammalian physiology; Mysm1DN/DN mice phenocopy Mysm1-knockout mice. CRISPR-generated Mysm1D660N (catalytic dead) knock-in mouse; Mysm1fl/DN CreERT2 inducible model; full hematopoietic and immune cell characterization Scientific reports High 36611064
2024 MYSM1 epigenetically regulates Id4 transcription by modifying histone marks at the Id4 promoter (H2AK119ub1 removal); loss of MYSM1 in neural stem cells (via Nestin-Cre) causes microcephaly with hyperproliferation, increased apoptosis, and skewed differentiation toward neurogenesis over astrogliogenesis; Id4 re-expression rescues these defects. Nestin-Cre Mysm1 conditional knockout mice; RNA sequencing; genome-wide CUT&Tag for histone modifications; Id4 rescue experiments Cell death & disease High 38342917
2024 MYSM1 directly interacts with TRIM21 via its MPN domain and regulates TRIM21 deubiquitination and protein stability; MYSM1 exacerbates doxorubicin-induced cardiotoxicity by enhancing ferroptosis through the MYSM1-TRIM21 axis. Co-IP combined with LC-MS/MS identifying TRIM21 as MYSM1 substrate; MPN domain requirement established; AAV9-mediated cardiomyocyte-specific MYSM1 knockdown in mice Cell communication and signaling : CCS Medium 39695708
2024 MYSM1 expression and DUB catalytic activity in mature dendritic cells are dispensable for DC maintenance in vivo or DC activation by microbial stimuli; instead, MYSM1 acts via its DUB catalytic activity specifically in haematopoietic progenitors to support normal DC lineage development and functional programming. Mysm1flCreERT2, Mysm1flCD11c-cre, and Mysm1DN conditional mouse models; flow cytometry; transcriptional profiling of DCs Immunology Medium 38316548
2025 MYSM1 deubiquitinates RIPK2 and recruits PP2A to dephosphorylate RIPK2 at S176, thereby inhibiting NF-κB and MAPK signaling pathways and attenuating osteoarthritis progression. Co-IP of MYSM1 with RIPK2 and PP2A; ubiquitination and phosphorylation assays; Ripk2S176D and Ripk2S176A mutation rescue experiments; MYSM1 knockout and overexpression in OA mouse models Bone research Medium 39746943
2025 MYSM1 mediates K63-linked deubiquitination and stabilization of STAT1 at K379 via its MPN metalloprotease domain, promoting STAT1 transcription factor function and necroptosis-related gene expression in myocardial ischemia/reperfusion injury. Proteome-wide quantitative analysis identifying STAT1 as MYSM1 substrate; Co-IP; ubiquitination assays with site-specific K379 mutation; AAV9 cardiomyocyte-specific MYSM1 knockdown in I/R mouse model Theranostics Medium 39897566
2025 MYSM1 deubiquitinates and stabilizes PARP1 in an MPN domain-dependent manner, mediating PARP1-dependent cardiomyocyte parthanatos and cardiac hypertrophy. Co-IP combined with LC-MS identifying PARP1 as MYSM1 substrate; MPN domain mutagenesis; AAV9 cardiomyocyte-specific MYSM1 knockdown in angiotensin II and TAC cardiac hypertrophy mouse models Hypertension Medium 39907013
2025 MYSM1 interacts with TRAF2 and TRAF3 via its SWIRM domain and removes K63-linked polyubiquitin chains to decrease TRAF2-TRAF3 complex stability, thereby impairing NIK degradation and sustaining non-canonical NF-κB activation while also promoting MAPK (p38, JNK) signaling in lung adenocarcinoma. Co-IP of MYSM1 with TRAF2/TRAF3; K63-ubiquitin chain removal assays; domain mapping; rescue experiments with TRAF2/TRAF3 inhibition; in vitro and in vivo functional assays NPJ precision oncology Medium 41162619
2026 Mysm1 mutations (meander tail alleles) in mice cause anterior-selective cerebellar malformation through cell-autonomous effects on granule cell precursors; multimodal single-nucleus assays show Mysm1 affects gene expression in several lineages and granule cell precursor proportions by E14.5, identifying a developmental role in cerebellar compartment specification. Positional cloning/complementation of meander tail mutations to Mysm1; single-nucleus multimodal genomics; non-complementing endonuclease-generated Mysm1 alleles bioRxivpreprint Medium 42124724

Source papers

Stage 0 corpus · 58 papers · ranked by NIH iCite citations
Year Title Journal Citations PMID
2011 The critical role of histone H2A-deubiquitinase Mysm1 in hematopoiesis and lymphocyte differentiation. Blood 88 22184403
2011 Control of B cell development by the histone H2A deubiquitinase MYSM1. Immunity 85 22169041
2015 Deubiquitinase MYSM1 Regulates Innate Immunity through Inactivation of TRAF3 and TRAF6 Complexes. Immunity 80 26474655
2013 The control of hematopoietic stem cell maintenance, self-renewal, and differentiation by Mysm1-mediated epigenetic regulation. Blood 78 24014243
2013 Epigenetic control of natural killer cell maturation by histone H2A deubiquitinase, MYSM1. Proceedings of the National Academy of Sciences of the United States of America 68 24062447
2020 MYSM1 Represses Innate Immunity and Autoimmunity through Suppressing the cGAS-STING Pathway. Cell reports 67 33086059
2015 An in vivo genetic reversion highlights the crucial role of Myb-Like, SWIRM, and MPN domains 1 (MYSM1) in human hematopoiesis and lymphocyte differentiation. The Journal of allergy and clinical immunology 64 26220525
2016 Repression of p53-target gene Bbc3/PUMA by MYSM1 is essential for the survival of hematopoietic multipotent progenitors and contributes to stem cell maintenance. Cell death and differentiation 55 26768662
2015 p53 mediates loss of hematopoietic stem cell function and lymphopenia in Mysm1 deficiency. Blood 54 25710881
2015 Interplay of H2A deubiquitinase 2A-DUB/Mysm1 and the p19(ARF)/p53 axis in hematopoiesis, early T-cell development and tissue differentiation. Cell death and differentiation 42 25613381
2017 Myb-like, SWIRM, and MPN domains 1 (MYSM1) deficiency: Genotoxic stress-associated bone marrow failure and developmental aberrations. The Journal of allergy and clinical immunology 41 28115216
2014 Epigenetic control of dendritic cell development and fate determination of common myeloid progenitor by Mysm1. Blood 40 25217698
2020 Deubiquitinase MYSM1 in the Hematopoietic System and beyond: A Current Review. International journal of molecular sciences 34 32344625
2016 Deubiquitinase MYSM1 Is Essential for Normal Bone Formation and Mesenchymal Stem Cell Differentiation. Scientific reports 32 26915790
2022 Genetic and Pharmacological Inhibition of Astrocytic Mysm1 Alleviates Depressive-Like Disorders by Promoting ATP Production. Advanced science (Weinheim, Baden-Wurttemberg, Germany) 25 36414403
2021 MYSM1 inhibits human colorectal cancer tumorigenesis by activating miR-200 family members/CDH1 and blocking PI3K/AKT signaling. Journal of experimental & clinical cancer research : CR 21 34706761
2018 Loss of p53 compensates osteopenia in murine Mysm1 deficiency. FASEB journal : official publication of the Federation of American Societies for Experimental Biology 20 29203593
2016 Mysm1 is required for interferon regulatory factor expression in maintaining HSC quiescence and thymocyte development. Cell death & disease 19 27277682
2015 Deubiquitinase MYSM1 Is Essential for Normal Fetal Liver Hematopoiesis and for the Maintenance of Hematopoietic Stem Cells in Adult Bone Marrow. Stem cells and development 19 26125289
2017 MYSM1/2A-DUB is an epigenetic regulator in human melanoma and contributes to tumor cell growth. Oncotarget 17 28978033
2015 Epigenetic Regulation of Antibody Responses by the Histone H2A Deubiquitinase MYSM1. Scientific reports 16 26348977
2018 MYSM1 Is Essential for Maintaining Hematopoietic Stem Cell (HSC) Quiescence and Survival. Medical science monitor : international medical journal of experimental and clinical research 15 29694335
2022 MYSM1 induces apoptosis and sensitizes TNBC cells to cisplatin via RSK3-phospho-BAD pathway. Cell death discovery 14 35217648
2020 MYSM1 Suppresses Cellular Senescence and the Aging Process to Prolong Lifespan. Advanced science (Weinheim, Baden-Wurttemberg, Germany) 14 33240758
2016 MYSM1-dependent checkpoints in B cell lineage differentiation and B cell-mediated immune response. Journal of leukocyte biology 14 27895164
2018 2A-DUB/Mysm1 Regulates Epidermal Development in Part by Suppressing p53-Mediated Programs. International journal of molecular sciences 13 29495602
2017 A role for the histone H2A deubiquitinase MYSM1 in maintenance of CD8+ T cells. Immunology 13 28066899
2017 Expression of MYSM1 is associated with tumor progression in colorectal cancer. PloS one 13 28498834
2019 MYSM1-AR complex-mediated repression of Akt/c-Raf/GSK-3β signaling impedes castration-resistant prostate cancer growth. Aging 11 31761786
2015 MYSM-1 suppresses migration and invasion in renal carcinoma through inhibiting epithelial-mesenchymal transition. Tumour biology : the journal of the International Society for Oncodevelopmental Biology and Medicine 11 26409454
2024 Deubiquitinase Mysm1 regulates neural stem cell proliferation and differentiation by controlling Id4 expression. Cell death & disease 9 38342917
2021 Loss of MYSM1 inhibits the oncogenic activity of cMYC in B cell lymphoma. Journal of cellular and molecular medicine 9 34114734
2019 Mysm1 epigenetically regulates the immunomodulatory function of adipose-derived stem cells in part by targeting miR-150. Journal of cellular and molecular medicine 9 30895711
2025 MYSM1 attenuates osteoarthritis by recruiting PP2A to deubiquitinate and dephosphorylate RIPK2. Bone research 8 39746943
2023 MYSM1 attenuates DNA damage signals triggered by physiologic and genotoxic DNA breaks. The Journal of allergy and clinical immunology 8 38065233
2023 MYSM1 acts as a novel co-activator of ERα to confer antiestrogen resistance in breast cancer. EMBO molecular medicine 8 38177530
2020 Further delineation of bone marrow failure syndrome caused by novel compound heterozygous variants of MYSM1. Gene 8 32640305
2018 Deubiquitinase Mysm1 regulates macrophage survival and polarization. Molecular biology reports 8 30386973
2016 MYSM1/miR-150/FLT3 inhibits B1a cell proliferation. Oncotarget 8 27590507
2023 Deubiquitinase catalytic activity of MYSM1 is essential in vivo for hematopoiesis and immune cell development. Scientific reports 7 36611064
2020 Interaction of Deubiquitinase 2A-DUB/MYSM1 with DNA Repair and Replication Factors. International journal of molecular sciences 7 32466590
2016 Mysm1 expression in the bone marrow niche is not essential for hematopoietic maintenance. Experimental hematology 7 27833034
2024 Deubiquitinase MYSM1 promotes doxorubicin-induced cardiotoxicity by mediating TRIM21-ferroptosis axis in cardiomyocytes. Cell communication and signaling : CCS 6 39695708
2024 Long-term assessment of haematological recovery following somatic genetic rescue in a MYSM1-deficient patient: Implications for in vivo gene therapy. British journal of haematology 5 39233474
2025 Deubiquitinase MYSM1 drives myocardial ischemia/reperfusion injury by stabilizing STAT1 in cardiomyocytes. Theranostics 3 39897566
2025 MYSM1 Mediates Cardiac Parthanatos and Hypertrophy by Deubiquitinating PARP1. Hypertension (Dallas, Tex. : 1979) 3 39907013
2024 Deubiquitinase MYSM1: An Important Tissue Development and Function Regulator. International journal of molecular sciences 2 39684760
2025 MiR-129-5p alleviates depression and anxiety by increasing astrocyte ATP production partly through targeting deubiquitinase Mysm1. PloS one 1 40344568
2024 Chromatin-binding deubiquitinase MYSM1 acts in haematopoietic progenitors to control dendritic cell development and to program dendritic cell responses to microbial stimulation. Immunology 1 38316548
2019 [Deubiquitinase MYSM1 Regulates Differentiation of Human B Cells to Plasma Cells]. Zhongguo shi yan xue ye xue za zhi 1 31607324
2026 MYSM1 expression in monocytes is negatively correlated to disease activity in Rheumatoid Arthritis, which may be related to the inhibitory effect on inflammatory aging. Clinical rheumatology 0 41917542
2026 Mysm1 mutations in meander tail mice cause anterior-selective cerebellum malformation. bioRxiv : the preprint server for biology 0 42124724
2025 Genetic and Clinical Progression of MYSM1 Related Bone Marrow Failure into Myeloid Malignancies: Case Series and Review of Literature. Clinical hematology international 0 40535318
2025 MYSM1 promotes lung adenocarcinoma progression via TRAF2/3-mediated activation of MAPK and non-canonical NF-κB pathways. NPJ precision oncology 0 41162619
2025 MYSM1 regulates the proliferation and differentiation of bovine skeletal muscle satellite cells via BRG1-mediated activation of the AKT/mTOR/NF-κB signaling pathway. Animal bioscience 0 41414839
2021 [A novel compound heterozygous mutation in MYSM1 gene in a 1-month-old girl: a bone marrow failure syndrome 4 family survey and literature review]. Zhonghua xue ye xue za zhi = Zhonghua xueyexue zazhi 0 33858043
2017 [Establishment of mouse stably knockout of MYSM1 in MSC cell line C3H10T1/2 and its effect on the immune regulation in vitro]. Zhongguo shi yan xue ye xue za zhi 0 28446309
2017 Correction: Expression of MYSM1 is associated with tumor progression in colorectal cancer. PloS one 0 29020094

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