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GeneE
26 sources retrieved Β· Most recent: April 2026 Β· Index updated 14 days ago
β“˜GeneE is for informational purposes only. It is not a substitute for professional medical advice, diagnosis, or treatment.
DKC1
dyskerin pseudouridine synthase 1
Chromosome X Β· Xq28
NCBI Gene: 1736Ensembl: ENSG00000130826.19HGNC: HGNC:2890UniProt: A0A8Q3SIY6
301PubMed Papers
23Diseases
0Drugs
31Pathogenic Variants
FUNCTIONAL ROLE
Highly ConstrainedHub Gene
RESEARCH IMPACT
Trending
CLINICAL
OMIM Disease Gene
DATA QUALITY
βœ“ Experimental GO Evidenceβœ“ Swiss-Prot Reviewed
positive regulation of telomerase RNA localization to Cajal bodytelomerase activityfibrillar centernucleusdyskeratosis congenita, X-linkeddyskeratosis congenitaHoyeraal-Hreidarsson syndromeneurodegenerative disease
✦AI Summary

DKC1 encodes dyskerin, a pseudouridine synthase that plays critical roles in telomere maintenance and RNA modification. The protein functions as a component of both the telomerase complex and box H/ACA small nucleolar ribonucleoprotein (snoRNP) particles 1. DKC1 catalyzes pseudouridine formation in ribosomal RNA and contributes to telomerase activity by stabilizing telomerase RNA components 2. Beyond its enzymatic functions, DKC1 promotes cell proliferation by binding to and stabilizing mRNA of ribosomal proteins including RPL10A, RPL22L1, RPL34, and RPS3 3. The protein's catalytic activity is essential for its oncogenic effects, as catalytically inactive mutants fail to accelerate cell growth 3. Clinically, DKC1 mutations cause X-linked dyskeratosis congenita, a multisystem disorder characterized by mucocutaneous abnormalities, bone marrow failure, and cancer predisposition 4. The disease results from defective telomere maintenance, with patients typically presenting very short telomeres 4. DKC1 mutations are also associated with related telomeropathies including Hoyeraal-Hreidarsson syndrome and contribute to familial interstitial lung disease 5. In cancer, DKC1 is upregulated in colorectal tumors and correlates with poor patient outcomes 3.

Sources cited
1
DKC1 encodes dyskerin, a component of snoRNP particles and telomerase complex
PMID: 12737310
2
DKC1 catalyzes pseudouridine formation and contributes to telomerase activity
PMID: 25219674
3
DKC1 promotes cell proliferation by stabilizing ribosomal protein mRNAs and requires catalytic activity for oncogenic effects
PMID: 34026451
4
DKC1 mutations cause X-linked dyskeratosis congenita with defective telomere maintenance and very short telomeres
PMID: 22160078
5
DKC1 mutations contribute to familial interstitial lung disease
PMID: 35715316
Disease Associationsβ“˜23
dyskeratosis congenita, X-linkedOpen Targets
0.85Strong
dyskeratosis congenitaOpen Targets
0.75Strong
Hoyeraal-Hreidarsson syndromeOpen Targets
0.72Strong
neurodegenerative diseaseOpen Targets
0.52Moderate
DKC1-related disorderOpen Targets
0.49Moderate
minimally differentiated acute myeloblastic leukemiaOpen Targets
0.46Moderate
cataracts, hearing impairment, nephrotic syndrome, and enterocolitis 1Open Targets
0.46Moderate
isolated cerebellar hypoplasia/agenesisOpen Targets
0.26Weak
pulmonary fibrosisOpen Targets
0.14Weak
immunodeficiency diseaseOpen Targets
0.12Weak
neoplasmOpen Targets
0.11Weak
cancerOpen Targets
0.10Suggestive
colorectal carcinomaOpen Targets
0.09Suggestive
neuroblastomaOpen Targets
0.09Suggestive
hepatocellular carcinomaOpen Targets
0.09Suggestive
gastric cancerOpen Targets
0.09Suggestive
lung adenocarcinomaOpen Targets
0.08Suggestive
nonpapillary renal cell carcinomaOpen Targets
0.08Suggestive
breast cancerOpen Targets
0.08Suggestive
prostate cancerOpen Targets
0.07Suggestive
Cataracts, hearing impairment, nephrotic syndrome, and enterocolitis 1UniProt
Dyskeratosis congenita, X-linkedUniProt
Hoyeraal-Hreidarsson syndromeUniProt
Pathogenic Variants31
NM_001363.5(DKC1):c.1058C>T (p.Ala353Val)Pathogenic
Dyskeratosis congenita, X-linked|Dyskeratosis congenita|not provided
β˜…β˜…β˜†β˜†2025β†’ Residue 353
NM_001363.5(DKC1):c.196A>G (p.Thr66Ala)Pathogenic
Dyskeratosis congenita, X-linked|not provided|Dyskeratosis congenita
β˜…β˜…β˜†β˜†2025β†’ Residue 66
NM_001363.5(DKC1):c.114C>G (p.Ile38Met)Likely pathogenic
Dyskeratosis congenita, X-linked
β˜…β˜…β˜†β˜†2024β†’ Residue 38
NM_001363.5(DKC1):c.146C>T (p.Thr49Met)Pathogenic
Dyskeratosis congenita, X-linked|not provided|Dyskeratosis congenita
β˜…β˜…β˜†β˜†2024β†’ Residue 49
NM_001363.5(DKC1):c.969T>A (p.Tyr323Ter)Pathogenic
Dyskeratosis congenita
β˜…β˜†β˜†β˜†2025β†’ Residue 323
NM_001363.5(DKC1):c.949C>T (p.Leu317Phe)Likely pathogenic
Dyskeratosis congenita, X-linked|Dyskeratosis congenita
β˜…β˜†β˜†β˜†2025β†’ Residue 317
NM_001363.5(DKC1):c.5_7del (p.Ala2del)Pathogenic
Dyskeratosis congenita
β˜…β˜†β˜†β˜†2023β†’ Residue 2
NM_001363.5(DKC1):c.197C>T (p.Thr66Ile)Likely pathogenic
not provided
β˜…β˜†β˜†β˜†2023β†’ Residue 66
NM_001363.5(DKC1):c.964C>T (p.Arg322Ter)Likely pathogenic
Dyskeratosis congenita, X-linked
β˜…β˜†β˜†β˜†2022β†’ Residue 322
NM_001363.5(DKC1):c.1226C>T (p.Pro409Leu)Likely pathogenic
Dyskeratosis congenita, X-linked|Dyskeratosis congenita
β˜…β˜†β˜†β˜†2022β†’ Residue 409
NM_001363.5(DKC1):c.204C>G (p.His68Gln)Likely pathogenic
Dyskeratosis congenita, X-linked
β˜…β˜†β˜†β˜†2022β†’ Residue 68
NM_001363.5(DKC1):c.149C>A (p.Ser50Tyr)Likely pathogenic
Dyskeratosis congenita
β˜…β˜†β˜†β˜†2022β†’ Residue 50
NM_001363.5(DKC1):c.189T>G (p.Asn63Lys)Pathogenic
Dyskeratosis congenita
β˜…β˜†β˜†β˜†2021β†’ Residue 63
NM_001363.5(DKC1):c.1195G>C (p.Asp399His)Likely pathogenic
Dyskeratosis congenita, X-linked
β˜…β˜†β˜†β˜†2020β†’ Residue 399
NM_001363.5(DKC1):c.109_111del (p.Leu37del)Likely pathogenic
Dyskeratosis congenita, X-linked|Dyskeratosis congenita
β˜…β˜†β˜†β˜†2020β†’ Residue 37
NM_001363.5(DKC1):c.203A>G (p.His68Arg)Pathogenic
Dyskeratosis congenita, X-linked
β˜…β˜†β˜†β˜†2017β†’ Residue 68
NM_001363.5(DKC1):c.1255T>A (p.Tyr419Asn)Likely pathogenic
Dyskeratosis congenita, X-linked
β˜…β˜†β˜†β˜†2016β†’ Residue 419
NM_001363.5(DKC1):c.133G>A (p.Ala45Thr)Likely pathogenic
Dyskeratosis congenita, X-linked
β˜…β˜†β˜†β˜†2016β†’ Residue 45
NM_001363.5(DKC1):c.1345C>G (p.Arg449Gly)Pathogenic
not provided
β˜…β˜†β˜†β˜†2015β†’ Residue 449
NM_001363.5(DKC1):c.1156G>A (p.Ala386Thr)Pathogenic
Dyskeratosis congenita, X-linked
β˜…β˜†β˜†β˜†β†’ Residue 386
View on ClinVar β†—
Related Genes
SNU13Protein interaction100%RRP9Protein interaction100%NOP56Protein interaction100%NOP58Protein interaction100%TERF1Protein interaction100%FBLProtein interaction100%
Tissue Expression6 tissues
Bone Marrow
100%
Brain
45%
Ovary
37%
Heart
28%
Lung
28%
Liver
25%
Gene Interaction Network
Click a node to explore
DKC1SNU13RRP9NOP56NOP58TERF1FBL
PROTEIN STRUCTURE
Preparing viewer…
PDB8OUE Β· 2.70 Γ… Β· EM
View on RCSB β†—
Constraintβ“˜
LOEUFβ“˜
0.20Highly Constrained
pLIβ“˜
1.00Intolerant
Observed/Expected LoF0.09 [0.04–0.20]
RankingsWhere DKC1 stands among ~20K protein-coding genes
  • #1,157of 20,598
    Most Researched301 Β· top 10%
  • #1,775of 5,498
    Most Pathogenic Variants31
  • #430of 17,882
    Most Constrained (LOEUF)0.20 Β· top 5%
Genes detectedDKC1
Sources retrieved26 papers
Response timeβ€”
πŸ“„ Sources
26β–Ό
1
Telomeres and telomerase: three decades of progress.
PMID: 30760854
Nat Rev Genet Β· 2019
1.00
2
Dual Inhibition of DKC1 and MEK1/2 Synergistically Restrains the Growth of Colorectal Cancer Cells.
PMID: 34026451
Adv Sci (Weinh) Β· 2021
0.90
3
Quantitative profiling of pseudouridylation landscape in the human transcriptome.
PMID: 36997645
Nat Chem Biol Β· 2023
0.80
4
Transcriptome-wide mapping reveals widespread dynamic-regulated pseudouridylation of ncRNA and mRNA.
PMID: 25219674
Cell Β· 2014
0.70
5
Dyskeratosis Congenita.
PMID: 31486376
Mayo Clin Proc Β· 2019
0.68