PHYHD1

Chr 9

phytanoyl-CoA dioxygenase domain containing 1

The protein functions as a 2-oxoglutarate-dependent dioxygenase that catalyzes the iron-dependent conversion of 2-oxoglutarate to succinate and CO2, though it does not directly participate in phytanoyl coenzyme-A metabolism or show activity toward fatty acid CoA thioesters. Based on the provided information, no specific diseases associated with PHYHD1 mutations have been established. The extremely low pLI score suggests this gene is highly tolerant to loss-of-function mutations.

OMIMResearchSummary from RefSeq, UniProt
LOEUF 1.47
Clinical SummaryPHYHD1
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
32 unique Pathogenic / Likely Pathogenic· 68 VUS of 125 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.47LOEUF
pLI 0.000
Z-score -0.02
OE 1.01 (0.701.47)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.23Z-score
OE missense 0.95 (0.841.08)
171 obs / 179.7 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE1.01 (0.701.47)
00.351.4
Missense OE0.95 (0.841.08)
00.61.4
Synonymous OE1.08
01.21.6
LoF obs/exp: 19 / 18.9Missense obs/exp: 171 / 179.7Syn Z: -0.51

ClinVar Variant Classifications

125 submitted variants in ClinVar

Classification Summary

Pathogenic30
Likely Pathogenic2
VUS68
Likely Benign14
30
Pathogenic
2
Likely Pathogenic
68
VUS
14
Likely Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
30
0
30
Likely Pathogenic
0
0
2
0
2
VUS
1
55
12
0
68
Likely Benign
0
14
0
0
14
Benign
0
0
0
0
0
Total169440114

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

View in ClinVar →

Protein Context — Lollipop Plot

PHYHD1 · 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 →
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC