COLGALT1

Chr 19AR

collagen beta(1-O)galactosyltransferase 1

Also known as: BSVD3, ColGalT 1, GLT25D1

This gene encodes a beta-galactosyltransferase that transfers galactose to hydroxylysine residues in type I and type IV collagen, facilitating collagen triple helix formation. Mutations cause autosomal recessive osteogenesis imperfecta, a connective tissue disorder affecting primarily bone and other collagen-rich tissues. The gene is not highly constrained against loss-of-function variants.

OMIMResearchSummary from RefSeq, UniProt
ARLOEUF 0.881 OMIM phenotype
Clinical SummaryCOLGALT1
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Gene-Disease Validity (ClinGen)
brain small vessel disease 3 · ARModerate

Moderate evidence — consider for supplementary testing

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
17 unique Pathogenic / Likely Pathogenic· 203 VUS of 352 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.88LOEUF
pLI 0.000
Z-score 2.06
OE 0.58 (0.390.88)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.52Z-score
OE missense 0.92 (0.841.01)
337 obs / 365.1 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.58 (0.390.88)
00.351.4
Missense OE0.92 (0.841.01)
00.61.4
Synonymous OE0.98
01.21.6
LoF obs/exp: 16 / 27.7Missense obs/exp: 337 / 365.1Syn Z: 0.22

ClinVar Variant Classifications

352 submitted variants in ClinVar

Classification Summary

Pathogenic15
Likely Pathogenic2
VUS203
Likely Benign91
Benign15
Conflicting5
15
Pathogenic
2
Likely Pathogenic
203
VUS
91
Likely Benign
15
Benign
5
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
3
3
9
0
15
Likely Pathogenic
0
1
1
0
2
VUS
3
178
22
0
203
Likely Benign
0
3
33
55
91
Benign
0
0
2
13
15
Conflicting
5
Total61856768331

LoF = frameshift, stop gained/lost, canonical splice · Counts from ClinVar esearch · Updated hourly

View in ClinVar →

Protein Context — Lollipop Plot

COLGALT1 · protein map & ClinVar variants

Showing all ClinVar variants across the protein. Search a specific variant to highlight its position.

3D Protein StructureAlphaFold

Clinical Trials

Active and recruiting trials from ClinicalTrials.gov

No active trials found for this gene.

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Clinical Literature
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