PTRHD1

Chr 2AR

peptidyl-tRNA hydrolase domain containing 1

Also known as: C2orf79, NEDPBA

This gene encodes a putative peptidyl-tRNA hydrolase that may be involved in releasing tRNAs from ribosomes during protein synthesis, though its enzymatic activity remains uncertain. Mutations cause autosomal recessive neurodevelopmental disorder with early-onset parkinsonism and behavioral abnormalities. The gene shows very low constraint against loss-of-function variants (pLI near zero), consistent with its recessive inheritance pattern.

Summary from RefSeq, OMIM, UniProt
Research Assistant →

Primary Disease Associations & Inheritance

Neurodevelopmental disorder with early-onset parkinsonism and behavioral abnormalitiesMIM #620747
AR
0
Active trials
2
Pubs (1 yr)
26
P/LP submissions
13%
P/LP missense
1.94
LOEUF
Mechanism
Clinical SummaryPTRHD1
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
24 unique Pathogenic / Likely Pathogenic· 36 VUS of 73 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.94LOEUF
pLI 0.000
Z-score -1.22
OE 1.59 (0.861.94)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.19Z-score
OE missense 1.06 (0.891.26)
89 obs / 84.1 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE1.59 (0.861.94)
00.351.4
Missense OE1.06 (0.891.26)
00.61.4
Synonymous OE1.25
01.21.6
LoF obs/exp: 8 / 5.0Missense obs/exp: 89 / 84.1Syn Z: -1.19

ClinVar Variant Classifications

73 submitted variants in ClinVar

Classification Summary

Pathogenic22
Likely Pathogenic2
VUS36
Likely Benign5
Benign3
22
Pathogenic
2
Likely Pathogenic
36
VUS
5
Likely Benign
3
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
3
19
0
22
Likely Pathogenic
1
0
1
0
2
VUS
0
28
8
0
36
Likely Benign
0
1
0
4
5
Benign
0
1
2
0
3
Total13330468

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

View in ClinVar →

Protein Context — Lollipop Plot

PTRHD1 · 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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Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC