AGRN

Chr 1AR

agrin

Also known as: AGRIN, CMS8, CMSPPD

The protein encoded by this gene is critical for neuromuscular junction development and contains multiple functional domains including laminin G, Kazal type serine protease inhibitor, and epidermal growth factor domains. Mutations cause congenital myasthenic syndrome type 8 with pre- and postsynaptic defects affecting limb-girdle muscles through an autosomal recessive inheritance pattern. The pathogenic mechanism involves gain-of-function effects of the mutant protein.

Summary from RefSeq, OMIM, UniProt, Mechanism
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Primary Disease Associations & Inheritance

Myasthenic syndrome, congenital, 8, with pre- and postsynaptic defectsMIM #615120
AR
0
Active trials
30
Pubs (1 yr)
21
P/LP submissions
5%
P/LP missense
0.43
LOEUF
Multiple*
Mechanism· predicted
Clinical SummaryAGRN
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Gene-Disease Validity (ClinGen)
congenital myasthenic syndrome 8 · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.32) despite low pLI — interpret in context.
📋
ClinVar Variants
21 unique Pathogenic / Likely Pathogenic· 149 VUS of 400 total submissions
📖
GeneReview available — AGRN
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.43LOEUF
pLI 0.000
Z-score 6.01
OE 0.32 (0.240.43)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
0.23Z-score
OE missense 0.98 (0.941.03)
1303 obs / 1326.1 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.32 (0.240.43)
00.351.4
Missense OE0.98 (0.941.03)
00.61.4
Synonymous OE1.20
01.21.6
LoF obs/exp: 29 / 90.9Missense obs/exp: 1303 / 1326.1Syn Z: -3.96
DN
0.6552th %ile
GOF
0.6834th %ile
LOF
0.2971th %ile

This gene has evidence for multiple mechanisms of pathogenicity (gain-of-function, dominant-negative and loss-of-function). Both the Badonyi & Marsh prediction and the broader genomic evidence point to gain-of-function as the predominant mechanism. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

GOFprediction above median
DNprediction above median
LOF52% of P/LP variants are LoF · LOEUF 0.43

Note: In-silico variant effect predictors (SIFT, PolyPhen, REVEL, CADD) may underestimate pathogenicity of missense variants in genes with GOF or DN mechanisms. Consider functional evidence and clinical context.

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

400 submitted variants in ClinVar

Classification Summary

Pathogenic13
Likely Pathogenic8
VUS149
Likely Benign188
Conflicting1
13
Pathogenic
8
Likely Pathogenic
149
VUS
188
Likely Benign
1
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
7
0
6
0
13
Likely Pathogenic
4
1
3
0
8
VUS
1
139
9
0
149
Likely Benign
0
4
75
109
188
Benign
0
0
0
0
0
Conflicting
1
Total1214493109359

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

View in ClinVar →

Protein Context — Lollipop Plot

AGRN · 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
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