SLC35A2

Chr XSomaticXLD

solute carrier family 35 member A2

Also known as: CDG2M, CDGX, UDP-Gal-Tr, UGALT, UGAT, UGT, UGT1, UGT2

The protein transports UDP-galactose from the cytosol into Golgi vesicles where it serves as a glycosyl donor for glycan generation. Mutations cause congenital disorder of glycosylation type IIm (CDG2M) with X-linked dominant inheritance, though somatic mosaicism can also occur. The moderate constraint scores suggest mutations can cause disease predominantly through loss-of-function, disrupting normal glycosylation pathways.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismSomatic/XLDLOEUF 0.561 OMIM phenotype
Clinical SummarySLC35A2
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Gene-Disease Validity (ClinGen)
X-linked complex neurodevelopmental disorder · XLDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Moderately constrained gene (pLI 0.72) — some intolerance to loss-of-function variants.
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ClinVar Variants
59 unique Pathogenic / Likely Pathogenic· 99 VUS of 300 total submissions
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Clinical Trials
3 active or recruiting trials — potential therapeutic options may be available
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GeneReview available — SLC35A2
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.56LOEUF
pLI 0.718
Z-score 2.38
OE 0.12 (0.040.56)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
2.50Z-score
OE missense 0.46 (0.380.55)
76 obs / 166.7 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.12 (0.040.56)
00.351.4
Missense OE0.46 (0.380.55)
00.61.4
Synonymous OE1.04
01.21.6
LoF obs/exp: 1 / 8.5Missense obs/exp: 76 / 166.7Syn Z: -0.30
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveSLC35A2-related epileptic encephalopathy due to congenital disorder of glycosylationLOFmonoallelic_X_heterozygous
DN
0.4586th %ile
GOF
0.6444th %ile
LOF
0.55top 25%

This gene has evidence for multiple mechanisms of pathogenicity (gain-of-function and loss-of-function). Both the Badonyi & Marsh prediction and the broader genomic evidence point to gain-of-function as the predominant mechanism. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

GOFprediction above median
LOF22% of P/LP variants are LoF

Note: In-silico variant effect predictors (SIFT, PolyPhen, REVEL, CADD) may underestimate pathogenicity of missense variants in genes with GOF or DN mechanisms. Consider functional evidence and clinical context.

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

300 submitted variants in ClinVar

Classification Summary

Pathogenic47
Likely Pathogenic12
VUS99
Likely Benign91
Benign14
Conflicting13
47
Pathogenic
12
Likely Pathogenic
99
VUS
91
Likely Benign
14
Benign
13
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
10
3
34
0
47
Likely Pathogenic
3
8
1
0
12
VUS
2
83
13
1
99
Likely Benign
2
22
15
52
91
Benign
0
10
1
3
14
Conflicting
13
Total171266456276

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

View in ClinVar →

Protein Context — Lollipop Plot

SLC35A2 · 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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