B4GALT6

Chr 18

beta-1,4-galactosyltransferase 6

Also known as: B4Gal-T6, beta4Gal-T6

The B4GALT6 protein catalyzes the synthesis of lactosylceramide, which serves as the starting point for ganglioside biosynthesis essential for neuronal maturation, axonal development, and myelin formation in the central nervous system. Mutations cause autosomal recessive spastic paraplegia and progressive myoclonic epilepsy, typically presenting in childhood with spasticity, seizures, and developmental regression. This gene is highly constrained against loss-of-function variants, indicating that complete loss of protein function is likely incompatible with normal development.

OMIMResearchSummary from RefSeq, UniProt
LOEUF 0.37
Clinical SummaryB4GALT6
Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 0.91). One damaged copy is likely sufficient to cause disease.
📋
ClinVar Variants
39 unique Pathogenic / Likely Pathogenic· 47 VUS of 98 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.37LOEUF
pLI 0.908
Z-score 3.63
OE 0.14 (0.060.37)
Highly constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
1.70Z-score
OE missense 0.68 (0.590.78)
149 obs / 220.1 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.14 (0.060.37)
00.351.4
Missense OE0.68 (0.590.78)
00.61.4
Synonymous OE0.90
01.21.6
LoF obs/exp: 3 / 20.9Missense obs/exp: 149 / 220.1Syn Z: 0.69

ClinVar Variant Classifications

98 submitted variants in ClinVar

Classification Summary

Pathogenic38
Likely Pathogenic1
VUS47
Likely Benign1
Benign1
38
Pathogenic
1
Likely Pathogenic
47
VUS
1
Likely Benign
1
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
38
0
38
Likely Pathogenic
0
0
1
0
1
VUS
0
36
11
0
47
Likely Benign
0
0
1
0
1
Benign
0
1
0
0
1
Total03751088

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

View in ClinVar →

Protein Context — Lollipop Plot

B4GALT6 · 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.

Search ClinicalTrials.gov →
Clinical Literature
Open Research Assistant →
Key Publications
Landmark & review papers · by relevance
PubMed
Top 2 results · since 2015Search PubMed ↗
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC