CHST14

Chr 15AR

carbohydrate sulfotransferase 14

Also known as: ATCS, D4ST1, EDSMC1, HNK1ST

This gene encodes a sulfotransferase that catalyzes the transfer of sulfate to N-acetylgalactosamine residues in dermatan sulfate, playing a crucial role in dermatan sulfate biosynthesis and cerebellar neural network formation during postnatal brain development. Biallelic mutations cause Ehlers-Danlos syndrome, musculocontractural type 1, an autosomal recessive connective tissue disorder characterized by joint contractures, skin hyperextensibility, and skeletal abnormalities. The gene shows moderate constraint against loss-of-function variants (LOEUF 0.64), suggesting some tolerance to haploinsufficiency.

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismARLOEUF 0.641 OMIM phenotype
Clinical SummaryCHST14
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Gene-Disease Validity (ClinGen)
Ehlers-Danlos syndrome, musculocontractural type 1 · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.20) despite low pLI — interpret in context.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.64LOEUF
pLI 0.447
Z-score 2.31
OE 0.20 (0.080.64)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.68Z-score
OE missense 0.87 (0.770.98)
182 obs / 209.8 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.20 (0.080.64)
00.351.4
Missense OE0.87 (0.770.98)
00.61.4
Synonymous OE0.91
01.21.6
LoF obs/exp: 2 / 9.8Missense obs/exp: 182 / 209.8Syn Z: 0.70
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveCHST14-related Ehlers-Danlos syndrome, musculocontractural typeLOFAR

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

DN
0.5378th %ile
GOF
0.6639th %ile
LOF
0.4134th %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

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

CHST14 · 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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