HSD17B4

Chr 5AR

hydroxysteroid 17-beta dehydrogenase 4

Also known as: DBP, MFE-2, MFP-2, MPF-2, PRLTS1, SDR8C1

The protein encoded by this gene is a bifunctional enzyme that is involved in the peroxisomal beta-oxidation pathway for fatty acids. It also acts as a catalyst for the formation of 3-ketoacyl-CoA intermediates from both straight-chain and 2-methyl-branched-chain fatty acids. Defects in this gene that affect the peroxisomal fatty acid beta-oxidation activity are a cause of D-bifunctional protein deficiency (DBPD). An apparent pseudogene of this gene is present on chromosome 8. Multiple alternatively spliced transcript variants encoding distinct isoforms have been found for this gene. [provided by RefSeq, May 2014]

GeneReviewsOMIMResearchGenerating clinical summary…

Primary Disease Associations & Inheritance

D-bifunctional protein deficiencyMIM #261515
AR
Perrault syndrome 1MIM #233400
AR

Clinical highlights

Gene-disease validity (ClinGen)
d-bifunctional protein deficiency · ARDefinitivesufficient evidence for diagnostic panels2 gene-disease associations curated in total
Interpreting a novel variant
Loss of function is the curated mechanism (Gene2Phenotype), though the gene is not strongly LoF-constrained in the population — weigh truncating variants against that tolerance.Curated gene-level mechanism — a prior for triage, not a per-variant call.
0
Active trials
27
Pubs (1 yr)
P/LP submissions
P/LP missense
0.74
LOEUF
LOF
Mechanism· G2P
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GeneReview available — HSD17B4
Authoritative clinical overview · Recommended first read
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Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint?
0.74LOEUF
pLI 0.000
Z-score 2.91
OE 0.53 (0.380.74)
Tolerant

Typical tolerance to LoF variation

Missense Constraint?
-0.28Z-score
OE missense 1.04 (0.961.13)
426 obs / 410.3 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios?
LoF OE?0.53 (0.380.74)
00.351.4
Missense OE?1.04 (0.961.13)
00.61.4
Synonymous OE?1.11
01.21.6
LoF obs/exp: 23 / 43.8Missense obs/exp: 426 / 410.3Syn Z: -0.98

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

HSD17B4 · 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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