KDSR

Chr 18AR

3-ketodihydrosphingosine reductase

Also known as: DHSR, EKVP4, FVT1, SDR35C1

The protein encoded by this gene catalyzes the reduction of 3-ketodihydrosphingosine to dihydrosphingosine. The putative active site residues of the encoded protein are found on the cytosolic side of the endoplasmic reticulum membrane. A chromosomal rearrangement involving this gene is a cause of follicular lymphoma, also known as type II chronic lymphatic leukemia. The mutation of a conserved residue in the bovine ortholog causes spinal muscular atrophy. [provided by RefSeq, Jul 2008]

Primary Disease Associations & Inheritance

Erythrokeratodermia variabilis et progressiva 4MIM #617526
AR
0
Active trials
3
Pubs (1 yr)
0
P/LP submissions
P/LP missense
0.58
LOEUF
DN
Mechanism· predicted
Clinical SummaryKDSR
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Gene-Disease Validity (ClinGen)
erythrokeratodermia variabilis et progressiva 4 · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.27) despite low pLI — interpret in context.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.58LOEUF
pLI 0.117
Z-score 2.87
OE 0.27 (0.140.58)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
0.94Z-score
OE missense 0.80 (0.700.92)
146 obs / 181.6 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.27 (0.140.58)
00.351.4
Missense OE0.80 (0.700.92)
00.61.4
Synonymous OE0.91
01.21.6
LoF obs/exp: 5 / 18.2Missense obs/exp: 146 / 181.6Syn Z: 0.58
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
limitedKDSR-related erythrokeratodermiavariabilis et progressivaOTHERAR

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

DN
0.7131th %ile
GOF
0.5856th %ile
LOF
0.2580th %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

KDSR · protein map & ClinVar variants

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

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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