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GeneE
10 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.
DOLK
dolichol kinase
Chromosome 9 Β· 9q34.11
NCBI Gene: 22845Ensembl: ENSG00000175283.8HGNC: HGNC:23406UniProt: A0A0S2Z597
25PubMed Papers
21Diseases
0Drugs
14Pathogenic Variants
FUNCTIONAL ROLE
Kinase
CLINICAL
OMIM Disease Gene
DATA QUALITY
βœ“ Experimental GO Evidenceβœ“ Swiss-Prot Reviewed
dolichol kinase activityprotein bindingdolichyl monophosphate biosynthetic processendoplasmic reticulum membraneDK1-congenital disorder of glycosylationcongenital disorder of glycosylationAbnormality of the cardiovascular systemcongenital disorder of glycosylation type I
✦AI Summary

DOLK encodes dolichol kinase, an endoplasmic reticulum membrane protein that catalyzes the final, CTP-dependent phosphorylation step in dolichyl monophosphate (Dol-P) biosynthesis 1. Dol-P functions as an essential oligosaccharide carrier required for N-linked and O-linked glycosylation and GPI anchor synthesis 1. The ER N-glycosylation pathway involving DOLK is evolutionarily conserved across multiple organisms 1. DOLK deficiency causes congenital disorder of glycosylation type 1M (DOLK-CDG), a severe autosomal recessive condition with early infantile onset and poor prognosis 2. DOLK-CDG manifests as a multi-system disorder affecting cardiac, cutaneous, neurological, and skeletal tissues 2. Clinical presentations include congenital heart disease, ichthyosis-like skin manifestations, developmental delay, hypotonia, and seizures, with affected individuals typically experiencing early mortality from multiple organ failure 2. DOLK is one of 29 genes associated with congenital disorders of glycosylation presenting cardiac complications, highlighting the critical importance of glycosylation in cardiac development and function 3. Novel compound heterozygous mutations (c.1268C>G and c.1581_1583del) have been identified, expanding the known DOLK mutation spectrum 2.

Sources cited
1
DOLK catalyzes CTP-dependent phosphorylation in dolichol phosphate biosynthesis, with Dol-P serving as an oligosaccharide carrier for N-glycosylation; the ER N-glycosylation pathway is evolutionarily conserved
PMID: 37250845
2
DOLK-CDG is a severe autosomal recessive disease with early onset affecting multiple organs including heart, skin, nerves, and bones; novel compound heterozygous mutations identified
PMID: 35674301
3
DOLK is one of 29 genes associated with congenital disorders of glycosylation that present with cardiac manifestations
PMID: 37239976
⚠Limited data available β€” This gene has 3 indexed publications. Summary and analysis may be incomplete.
Disease Associationsβ“˜21
DK1-congenital disorder of glycosylationOpen Targets
0.81Strong
congenital disorder of glycosylationOpen Targets
0.60Moderate
Abnormality of the cardiovascular systemOpen Targets
0.52Moderate
congenital disorder of glycosylation type IOpen Targets
0.37Weak
familial isolated dilated cardiomyopathyOpen Targets
0.37Weak
SRD5A3-congenital disorder of glycosylationOpen Targets
0.37Weak
dilated cardiomyopathyOpen Targets
0.12Weak
hypertrophic cardiomyopathyOpen Targets
0.12Weak
hypertrophic cardiomyopathy 26Open Targets
0.12Weak
major depressive disorderOpen Targets
0.05Suggestive
attention deficit hyperactivity disorderOpen Targets
0.05Suggestive
male reproductive organ cancerOpen Targets
0.05Suggestive
osteosarcomaOpen Targets
0.02Suggestive
hepatocellular carcinomaOpen Targets
0.02Suggestive
preeclampsiaOpen Targets
0.02Suggestive
hyperinsulinemic hypoglycemia, familial, 4Open Targets
0.02Suggestive
colon carcinomaOpen Targets
0.01Suggestive
infectionOpen Targets
0.01Suggestive
cancerOpen Targets
0.01Suggestive
autosomal recessive dilated cardiomyopathyOpen Targets
0.01Suggestive
Congenital disorder of glycosylation 1MUniProt
Pathogenic Variants14
NM_014908.4(DOLK):c.2T>C (p.Met1Thr)Pathogenic
DK1-congenital disorder of glycosylation|not provided|Cardiovascular phenotype
β˜…β˜…β˜†β˜†2024β†’ Residue 1
NM_014908.4(DOLK):c.1342G>C (p.Gly448Arg)Pathogenic
DK1-congenital disorder of glycosylation
β˜…β˜†β˜†β˜†2025β†’ Residue 448
NM_014908.4(DOLK):c.1222C>G (p.His408Asp)Likely pathogenic
DK1-congenital disorder of glycosylation
β˜…β˜†β˜†β˜†2024β†’ Residue 408
NM_014908.4(DOLK):c.132G>A (p.Trp44Ter)Likely pathogenic
Cardiovascular phenotype
β˜…β˜†β˜†β˜†2024β†’ Residue 44
NM_014908.4(DOLK):c.1110dup (p.Ile371fs)Likely pathogenic
DK1-congenital disorder of glycosylation
β˜…β˜†β˜†β˜†2024β†’ Residue 371
NM_014908.4(DOLK):c.734_737del (p.Phe245fs)Pathogenic
Cardiovascular phenotype
β˜…β˜†β˜†β˜†2024β†’ Residue 245
NM_014908.4(DOLK):c.3G>A (p.Met1Ile)Pathogenic
DK1-congenital disorder of glycosylation
β˜…β˜†β˜†β˜†2023β†’ Residue 1
NM_014908.4(DOLK):c.857G>A (p.Trp286Ter)Likely pathogenic
Cardiovascular phenotype
β˜…β˜†β˜†β˜†2023β†’ Residue 286
NM_014908.4(DOLK):c.991C>T (p.Gln331Ter)Likely pathogenic
Cardiovascular phenotype
β˜…β˜†β˜†β˜†2020β†’ Residue 331
NM_014908.4(DOLK):c.1112T>A (p.Ile371Asn)Likely pathogenic
not provided
β˜…β˜†β˜†β˜†2018β†’ Residue 371
NC_000009.12:g.128945857G>TPathogenic
not provided
β˜…β˜†β˜†β˜†2013
NM_014908.4(DOLK):c.912G>T (p.Trp304Cys)Pathogenic
DK1-congenital disorder of glycosylation
β˜†β˜†β˜†β˜†2011β†’ Residue 304
NM_014908.4(DOLK):c.1322A>C (p.Tyr441Ser)Pathogenic
DK1-congenital disorder of glycosylation
β˜†β˜†β˜†β˜†2007β†’ Residue 441
NM_014908.4(DOLK):c.295T>A (p.Cys99Ser)Pathogenic
DK1-congenital disorder of glycosylation
β˜†β˜†β˜†β˜†2007β†’ Residue 99
View on ClinVar β†—
Related Genes
FKRPProtein interaction100%POMGNT1Protein interaction99%POMT2Protein interaction99%POMKProtein interaction98%DPAGT1Protein interaction95%ALG5Protein interaction95%
Tissue Expression6 tissues
Heart
100%
Liver
88%
Lung
74%
Brain
59%
Ovary
55%
Bone Marrow
21%
Gene Interaction Network
Click a node to explore
DOLKFKRPPOMGNT1POMT2POMKDPAGT1ALG5
PROTEIN STRUCTURE
Preparing viewer…
AlphaFoldAI-predicted Β· UniProt Q9UPQ8
View on AlphaFold β†—
Constraintβ“˜
LOEUFβ“˜
0.86LoF Tolerant
pLIβ“˜
0.00Tolerant
Observed/Expected LoF0.59 [0.41–0.86]
RankingsWhere DOLK stands among ~20K protein-coding genes
  • #12,967of 20,598
    Most Researched25
  • #2,543of 5,498
    Most Pathogenic Variants14
  • #7,509of 17,882
    Most Constrained (LOEUF)0.86
Genes detectedDOLK
Sources retrieved10 papers
Response timeβ€”
πŸ“„ Sources
10β–Ό
1
Metabolic Cardiomyopathies and Cardiac Defects in Inherited Disorders of Carbohydrate Metabolism: A Systematic Review.
PMID: 37239976
Int J Mol Sci Β· 2023
1.00
2
The prevalence of congenital anomalies in Europe.
PMID: 20824455
Adv Exp Med Biol Β· 2010
0.90
3
International standards for newborn weight, length, and head circumference by gestational age and sex: the Newborn Cross-Sectional Study of the INTERGROWTH-21st Project.
PMID: 25209487
Lancet Β· 2014
0.80
4
Disability-adjusted life years (DALYs) for 291 diseases and injuries in 21 regions, 1990-2010: a systematic analysis for the Global Burden of Disease Study 2010.
PMID: 23245608
Lancet Β· 2012
0.70
5
Years lived with disability (YLDs) for 1160 sequelae of 289 diseases and injuries 1990-2010: a systematic analysis for the Global Burden of Disease Study 2010.
PMID: 23245607
Lancet Β· 2012
0.60