B4GALT1

Chr 9AR

beta-1,4-galactosyltransferase 1

Also known as: B4GAL-T1, CDG2D, CLDLFIB, GGTB2, GT1, GTB, beta4Gal-T1

The protein is a galactosyltransferase that transfers galactose residues to N-acetylglucosamine on glycoproteins and glycolipids in the Golgi apparatus, and also forms part of the lactose synthase complex in mammary tissue. Biallelic mutations cause congenital disorder of glycosylation type IId and a condition with combined low LDL and fibrinogen, inherited in an autosomal recessive pattern. The gene shows tolerance to loss-of-function variants (pLI 0.04, LOEUF 0.70), suggesting that complete loss of function may be compatible with survival but causes clinically significant glycosylation defects.

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismARLOEUF 0.702 OMIM phenotypes
Clinical SummaryB4GALT1
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.33) despite low pLI — interpret in context.
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ClinVar Variants
7 unique Pathogenic / Likely Pathogenic· 59 VUS of 100 total submissions
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Clinical Trials
1 active or recruiting trial — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.70LOEUF
pLI 0.037
Z-score 2.38
OE 0.33 (0.170.70)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.98Z-score
OE missense 0.82 (0.720.92)
184 obs / 225.2 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.33 (0.170.70)
00.351.4
Missense OE0.82 (0.720.92)
00.61.4
Synonymous OE1.06
01.21.6
LoF obs/exp: 5 / 14.9Missense obs/exp: 184 / 225.2Syn Z: -0.47
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
strongB4GALT1-related congenital disorder of glycosylationLOFAR

Predictions shown for reference only — model trained on dominant genes, not applicable to AR conditions.

DN
0.6648th %ile
GOF
0.6444th %ile
LOF
0.3550th %ile

The Badonyi & Marsh prediction model was trained exclusively on dominant disease genes. Predictions are not reliable for genes with autosomal recessive inheritance and are shown at reduced opacity for reference only.

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312.

ClinVar Variant Classifications

100 submitted variants in ClinVar

Classification Summary

Pathogenic5
Likely Pathogenic2
VUS59
Likely Benign21
Benign1
Conflicting1
5
Pathogenic
2
Likely Pathogenic
59
VUS
21
Likely Benign
1
Benign
1
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
1
4
0
5
Likely Pathogenic
0
0
2
0
2
VUS
1
52
6
0
59
Likely Benign
0
3
10
8
21
Benign
0
0
0
1
1
Conflicting
1
Total15622989

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

View in ClinVar →

Protein Context — Lollipop Plot

B4GALT1 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
Clinical Literature
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