AGO2

Chr 8AD

argonaute RISC catalytic component 2

Also known as: CASC7, EIF2C2, LESKRES, LINC00980, PPD, Q10

This protein is essential for RNA-mediated gene silencing, functioning as the catalytic component of the RNA-induced silencing complex (RISC) that uses microRNAs and small interfering RNAs to regulate gene expression through mRNA cleavage or translational repression. Mutations cause Lessel-Kreienkamp syndrome, an autosomal dominant neurodevelopmental disorder. The gene is highly constrained against loss-of-function mutations (pLI >0.99, LOEUF 0.106), indicating that complete protein loss is likely incompatible with normal development.

Summary from RefSeq, OMIM, UniProt
Research Assistant →

Primary Disease Associations & Inheritance

Lessel-Kreienkamp syndromeMIM #619149
AD
0
Active trials
139
Pubs (1 yr)
85
P/LP submissions
20%
P/LP missense
0.11
LOEUF· LoF intol.
LOF
Mechanism· predicted
Clinical SummaryAGO2
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Gene-Disease Validity (ClinGen)
Lessel-Kreienkamp syndrome · ADDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 1.00). One damaged copy is likely sufficient to cause disease.
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ClinVar Variants
71 unique Pathogenic / Likely Pathogenic· 127 VUS of 265 total submissions
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GeneReview available — AGO2
Authoritative clinical overview · Recommended first read
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Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Dual constrained — LoF & missense intolerant
LoF Constraint
0.11LOEUF
pLI 1.000
Z-score 6.07
OE 0.02 (0.010.11)
Highly constrained

Among the most LoF-intolerant genes (~top 3%)

Missense Constraint
6.06Z-score
OE missense 0.29 (0.250.33)
162 obs / 568.4 exp
Constrained

Extremely missense-constrained (top ~0.01%)

Observed / Expected Ratios
LoF OE0.02 (0.010.11)
00.351.4
Missense OE0.29 (0.250.33)
00.61.4
Synonymous OE0.93
01.21.6
LoF obs/exp: 1 / 44.9Missense obs/exp: 162 / 568.4Syn Z: 0.90
DN
0.2997th %ile
GOF
0.2696th %ile
LOF
0.76top 10%

The highest-scoring mechanism for this gene is loss-of-function (haploinsufficiency).

LOFprediction above median · 1 literature citation · LOEUF 0.11

Literature Evidence

LOFThere was no evidence for a dominant-negative effect, indicating that the mutations most likely result in a loss of function.PMID:33199684

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

265 submitted variants in ClinVar

Classification Summary

Pathogenic56
Likely Pathogenic15
VUS127
Likely Benign29
Benign14
Conflicting1
56
Pathogenic
15
Likely Pathogenic
127
VUS
29
Likely Benign
14
Benign
1
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
4
52
0
56
Likely Pathogenic
0
10
5
0
15
VUS
5
105
17
0
127
Likely Benign
0
1
7
21
29
Benign
0
0
6
8
14
Conflicting
1
Total51208729242

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

View in ClinVar →

Protein Context — Lollipop Plot

AGO2 · 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.

Search ClinicalTrials.gov →
Clinical Literature
Open Research Assistant →
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC