HomeAboutRankingsData Sources
Β© 2026 GeneE
🧬
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.
CHKB
choline kinase beta
Chromosome 22 Β· 22q13.33
NCBI Gene: 1120Ensembl: ENSG00000100288.21HGNC: HGNC:1938UniProt: Q9Y259
45PubMed Papers
21Diseases
0Drugs
43Pathogenic Variants
FUNCTIONAL ROLE
Kinase
CLINICAL
OMIM Disease Gene
DATA QUALITY
βœ“ Experimental GO Evidenceβœ“ Swiss-Prot Reviewed
choline kinase activityethanolamine kinase activityphosphatidylethanolamine biosynthetic processCDP-choline pathwayCongenital muscular dystrophy due to phosphatidylcholine biosynthesis defectmegaconial type congenital muscular dystrophygenetic disorderneurodegenerative disease
✦AI Summary

CHKB (choline kinase beta) catalyzes the first committed step in phosphatidylcholine and phosphatidylethanolamine biosynthesis via the Kennedy pathway 1. As a key enzyme in phospholipid metabolism, CHKB maintains structural integrity of cell membranes and regulates mitochondrial homeostasis 2. Autosomal recessive CHKB mutations cause megaconial congenital muscular dystrophy (MCMD), characterized by early-onset muscle weakness, intellectual disability, and pathognomonic enlarged mitochondria 34. Loss-of-function mutations reduce choline kinase activity and phosphatidylcholine levels, triggering mitophagy and elimination of dysfunctional mitochondria through autophagy 4. Affected patients show reduced complex I respiratory chain activity and impaired mitochondrial fission factor (DRP1) expression 1. Clinical manifestations extend beyond muscle; brain MRI spectroscopy reveals decreased choline-based substrates, and patients develop gastrointestinal complications with acute deterioration during infection 5. CHKB is also essential for endochondral bone formation, with Chkb knockout mice displaying radius and ulna deformations due to impaired chondrocyte differentiation 6. Recently, the long noncoding RNA CHKB-DT was identified as downregulated in dilated cardiomyopathy, suggesting broader physiological roles in energy metabolism and cardiac function 7.

Sources cited
1
CHKB catalyzes first step in phosphatidylcholine and phosphatidylethanolamine biosynthesis; mutations cause megaconial congenital muscular dystrophy
PMID: 36175989
2
Choline involved in structural integrity of cell membranes and lipid-derived signaling; CHKB mutations cause inborn errors of metabolism
PMID: 30681159
3
CHKB mutations associated with megaconial congenital muscular dystrophy; decreased CHKB expression produces giant mitochondria
PMID: 36896673
4
Loss-of-function CHKB mutations cause decreased phosphatidylcholine levels and undetectable choline kinase activity; mitochondria eliminated by mitophagy
PMID: 24291895
5
CHKB deficiency causes brain choline-based substrate reduction and gastrointestinal features; reduced complex I activity in muscle
PMID: 23692895
6
CHKB required for normal endochondral bone formation; Chkb knockout impairs chondrocyte differentiation and radius/ulna development
PMID: 24637075
7
CHKB-DT long noncoding RNA downregulated in dilated cardiomyopathy; regulates energy metabolism and ATP production
PMID: 38299365
Disease Associationsβ“˜21
Congenital muscular dystrophy due to phosphatidylcholine biosynthesis defectOpen Targets
0.77Strong
megaconial type congenital muscular dystrophyOpen Targets
0.75Strong
genetic disorderOpen Targets
0.45Moderate
neurodegenerative diseaseOpen Targets
0.39Weak
muscular dystrophyOpen Targets
0.28Weak
chronic obstructive pulmonary diseaseOpen Targets
0.17Weak
SeizureOpen Targets
0.12Weak
Severe global developmental delayOpen Targets
0.12Weak
acute myeloid leukemiaOpen Targets
0.12Weak
neoplasmOpen Targets
0.12Weak
amyotrophic lateral sclerosisOpen Targets
0.11Weak
breast cancerOpen Targets
0.11Weak
acute kidney injuryOpen Targets
0.10Weak
hepatocellular carcinomaOpen Targets
0.10Weak
cancerOpen Targets
0.10Suggestive
infectionOpen Targets
0.10Suggestive
rhabdomyolysisOpen Targets
0.10Suggestive
myelodysplastic syndromeOpen Targets
0.09Suggestive
Duchenne muscular dystrophyOpen Targets
0.09Suggestive
gastric cancerOpen Targets
0.09Suggestive
Muscular dystrophy, congenital, megaconial typeUniProt
Pathogenic Variants43
NM_005198.5(CHKB):c.151C>T (p.Gln51Ter)Pathogenic
not provided|Megaconial type congenital muscular dystrophy
β˜…β˜…β˜†β˜†2025β†’ Residue 51
NM_005198.5(CHKB):c.598del (p.Gln200fs)Pathogenic
Megaconial type congenital muscular dystrophy|not provided
β˜…β˜…β˜†β˜†2023β†’ Residue 200
NM_005198.5(CHKB):c.331C>T (p.Gln111Ter)Pathogenic
Megaconial type congenital muscular dystrophy|not provided
β˜…β˜…β˜†β˜†2023β†’ Residue 111
NM_005198.5(CHKB):c.689_690del (p.Glu230fs)Pathogenic
Megaconial type congenital muscular dystrophy
β˜…β˜†β˜†β˜†2026β†’ Residue 230
NM_005198.5(CHKB):c.678-2A>GLikely pathogenic
Megaconial type congenital muscular dystrophy
β˜…β˜†β˜†β˜†2026
NM_005198.5(CHKB):c.722A>G (p.Asn241Ser)Pathogenic
Megaconial type congenital muscular dystrophy
β˜…β˜†β˜†β˜†2025β†’ Residue 241
NM_005198.5(CHKB):c.592_593del (p.Gln198fs)Pathogenic
Megaconial type congenital muscular dystrophy
β˜…β˜†β˜†β˜†2025β†’ Residue 198
NM_005198.5(CHKB):c.939del (p.Arg314fs)Pathogenic
Megaconial type congenital muscular dystrophy
β˜…β˜†β˜†β˜†2024β†’ Residue 314
NM_005198.5(CHKB):c.225-1_225delinsTTLikely pathogenic
Megaconial type congenital muscular dystrophy
β˜…β˜†β˜†β˜†2024
NM_005198.5(CHKB):c.1068G>A (p.Trp356Ter)Likely pathogenic
Megaconial type congenital muscular dystrophy
β˜…β˜†β˜†β˜†2024β†’ Residue 356
NM_005198.5(CHKB):c.683dup (p.Leu228fs)Pathogenic
Inborn genetic diseases
β˜…β˜†β˜†β˜†2024β†’ Residue 228
NM_005198.5(CHKB):c.565_568del (p.Phe189fs)Pathogenic
Megaconial type congenital muscular dystrophy
β˜…β˜†β˜†β˜†2024β†’ Residue 189
NM_005198.5(CHKB):c.678-2A>TLikely pathogenic
Megaconial type congenital muscular dystrophy
β˜…β˜†β˜†β˜†2024
NM_005198.5(CHKB):c.810T>A (p.Tyr270Ter)Pathogenic
Megaconial type congenital muscular dystrophy|CHKB-related disorder
β˜…β˜†β˜†β˜†2024β†’ Residue 270
NM_005198.5(CHKB):c.187C>T (p.Arg63Ter)Pathogenic
Megaconial type congenital muscular dystrophy
β˜…β˜†β˜†β˜†2023β†’ Residue 63
NM_005198.5(CHKB):c.556del (p.His186fs)Pathogenic
Megaconial type congenital muscular dystrophy
β˜…β˜†β˜†β˜†2023β†’ Residue 186
NM_005198.5(CHKB):c.536del (p.Met179fs)Pathogenic
Megaconial type congenital muscular dystrophy
β˜…β˜†β˜†β˜†2023β†’ Residue 179
NM_005198.5(CHKB):c.268del (p.His90fs)Pathogenic
Megaconial type congenital muscular dystrophy
β˜…β˜†β˜†β˜†2023β†’ Residue 90
NM_005198.5(CHKB):c.224+1G>APathogenic
Megaconial type congenital muscular dystrophy
β˜…β˜†β˜†β˜†2023
NM_005198.5(CHKB):c.446del (p.Pro149fs)Pathogenic
Megaconial type congenital muscular dystrophy
β˜…β˜†β˜†β˜†2023β†’ Residue 149
View on ClinVar β†—
Related Genes
TAC3Protein interaction100%GPCPD1Protein interaction95%TACR2Protein interaction93%ETNPPLProtein interaction91%CPT1BProtein interaction91%PHOSPHO1Protein interaction91%
Tissue Expression6 tissues
Liver
100%
Lung
94%
Ovary
75%
Bone Marrow
63%
Heart
32%
Brain
12%
Gene Interaction Network
Click a node to explore
CHKBTAC3GPCPD1TACR2ETNPPLCPT1BPHOSPHO1
PROTEIN STRUCTURE
Preparing viewer…
PDB3FEG Β· 1.30 Γ… Β· X-ray
View on RCSB β†—
Constraintβ“˜
LOEUFβ“˜
1.01LoF Tolerant
pLIβ“˜
0.00Tolerant
Observed/Expected LoF0.79 [0.63–1.01]
RankingsWhere CHKB stands among ~20K protein-coding genes
  • #9,426of 20,598
    Most Researched45
  • #1,475of 5,498
    Most Pathogenic Variants43
  • #9,917of 17,882
    Most Constrained (LOEUF)1.01
Genes detectedCHKB
Sources retrieved10 papers
Response timeβ€”
πŸ“„ Sources
10β–Ό
1
LncRNA CHKB-DT Downregulation Enhances Dilated Cardiomyopathy Through ALDH2.
PMID: 38299365
Circ Res Β· 2024
1.00
2
A 20-year Clinical and Genetic Neuromuscular Cohort Analysis in Lebanon: An International Effort.
PMID: 34602496
J Neuromuscul Dis Β· 2022
0.90
3
Large heterozygous deletion and uniparental disomy masquerading as homozygosity in CHKB gene.
PMID: 36896673
Mol Genet Genomic Med Β· 2023
0.80
4
Choline-related-inherited metabolic diseases-A mini review.
PMID: 30681159
J Inherit Metab Dis Β· 2019
0.70
5
Megaconial congenital muscular dystrophy due to novel CHKB variants: a case report and literature review.
PMID: 36175989
Skelet Muscle Β· 2022
0.60