A2ML1

Chr 12AD

alpha-2-macroglobulin like 1

Also known as: CPAMD9, OMS, p170

The A2ML1 protein functions as a broad-spectrum protease inhibitor that traps proteases through a unique bait-and-capture mechanism involving conformational changes and covalent binding. Autosomal dominant mutations cause increased susceptibility to otitis media, though the gene has also been reported in some cases of Noonan syndrome. This gene shows minimal constraint against loss-of-function variants based on population genetics data.

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

{Otitis media, susceptibility to}MIM #166760
AD
0
Active trials
8
Pubs (1 yr)
4
P/LP submissions
0%
P/LP missense
0.95
LOEUF
Mechanism
Clinical SummaryA2ML1
🧬
Gene-Disease Validity (ClinGen)
Noonan syndrome · ADDisputed

Disputed — evidence questions this relationship

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
4 unique Pathogenic / Likely Pathogenic· 305 VUS of 500 total submissions
📖
GeneReview available — A2ML1
Authoritative clinical overview · Recommended first read
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Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.95LOEUF
pLI 0.000
Z-score 1.91
OE 0.77 (0.630.95)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.33Z-score
OE missense 0.97 (0.911.03)
766 obs / 791.9 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.77 (0.630.95)
00.351.4
Missense OE0.97 (0.911.03)
00.61.4
Synonymous OE1.00
01.21.6
LoF obs/exp: 63 / 81.6Missense obs/exp: 766 / 791.9Syn Z: -0.07

ClinVar Variant Classifications

500 submitted variants in ClinVar

Classification Summary

Pathogenic3
Likely Pathogenic1
VUS305
Likely Benign166
Benign1
Conflicting2
3
Pathogenic
1
Likely Pathogenic
305
VUS
166
Likely Benign
1
Benign
2
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
3
0
3
Likely Pathogenic
0
0
1
0
1
VUS
27
249
26
3
305
Likely Benign
0
5
72
89
166
Benign
0
0
1
0
1
Conflicting
2
Total2725410392478

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

View in ClinVar →

Protein Context — Lollipop Plot

A2ML1 · 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