The protein catalyzes the final step of the urea cycle, converting L-arginine to urea and L-ornithine, with ornithine serving as a precursor for proline and polyamines essential for collagen synthesis and cell proliferation. Mutations cause argininemia, an autosomal recessive urea cycle disorder characterized by hyperargininemia and progressive neurological deterioration. The pathogenic mechanism involves loss of enzyme function leading to toxic accumulation of arginine and impaired urea production.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
LOFmechanismARLOEUF 0.921 OMIM phenotype
VCEP Guidelines: Urea Cycle DisordersReleased
ClinGen Panel
Clinical SummaryARG1
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Gene-Disease Validity (ClinGen)
hyperargininemia · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
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ClinVar Variants
130 unique Pathogenic / Likely Pathogenic· 144 VUS of 499 total submissions
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Clinical Trials
4 active or recruiting trials — potential therapeutic options may be available
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GeneReview available — ARG1
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.92LOEUF
pLI 0.000
Z-score 1.81
OE 0.51 (0.300.92)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.06Z-score
OE missense 0.99 (0.871.12)
176 obs / 178.1 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.51 (0.300.92)
00.351.4
Missense OE0.99 (0.871.12)
00.61.4
Synonymous OE1.00
01.21.6
LoF obs/exp: 8 / 15.7Missense obs/exp: 176 / 178.1Syn Z: 0.01
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveARG1-related argininemiaLOFAR

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

DN
0.75top 25%
GOF
0.4874th %ile
LOF
0.3260th %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

499 submitted variants in ClinVar

Classification Summary

Pathogenic64
Likely Pathogenic66
VUS144
Likely Benign200
Benign14
Conflicting9
64
Pathogenic
66
Likely Pathogenic
144
VUS
200
Likely Benign
14
Benign
9
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
39
11
14
0
64
Likely Pathogenic
48
11
7
0
66
VUS
6
109
29
0
144
Likely Benign
2
4
82
112
200
Benign
0
0
13
1
14
Conflicting
9
Total95135145113497

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

View in ClinVar →

Protein Context — Lollipop Plot

ARG1 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
Clinical Literature
Open Research Assistant →
Full-Text Mentions
NLP-detected gene mentions in article bodies · via PubTator3
PubTator3
Top 5 full-text resultsSearch PubTator3 ↗