ADCY9

Chr 16

adenylate cyclase 9

Also known as: AC9, ACIX

Adenylate cyclase 9 is a membrane-bound enzyme that catalyzes the formation of cyclic AMP from ATP in response to G protein-coupled receptor activation, contributing to signaling cascades involving corticotropin-releasing factor, corticosteroids, and beta-adrenergic receptors. Mutations in this highly constrained gene (pLI 0.98, LOEUF 0.31) cause autosomal recessive intellectual disability with hypotonia and behavioral abnormalities. The gene shows extreme intolerance to loss-of-function variants, suggesting that even heterozygous loss may have clinical consequences.

OMIMResearchSummary from RefSeq, UniProt
MultiplemechanismLOEUF 0.31
Clinical SummaryADCY9
Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 0.98). One damaged copy is likely sufficient to cause disease.
📋
ClinVar Variants
44 unique Pathogenic / Likely Pathogenic· 174 VUS of 269 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.31LOEUF
pLI 0.979
Z-score 5.28
OE 0.17 (0.100.31)
Highly constrained

Highly LoF-intolerant (top ~10% of genes)

Missense Constraint
1.25Z-score
OE missense 0.88 (0.830.93)
730 obs / 831.1 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.17 (0.100.31)
00.351.4
Missense OE0.88 (0.830.93)
00.61.4
Synonymous OE1.19
01.21.6
LoF obs/exp: 8 / 47.2Missense obs/exp: 730 / 831.1Syn Z: -2.93
DN
0.5280th %ile
GOF
0.6638th %ile
LOF
0.58top 25%

This gene has evidence for multiple mechanisms of pathogenicity (loss-of-function and gain-of-function). The Badonyi & Marsh model scores gain-of-function highest among its predictions, but genomic evidence (constraint, ClinVar variant spectrum, and literature) most strongly supports loss-of-function (haploinsufficiency). Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

LOFLOEUF 0.31
GOFprediction above median

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

269 submitted variants in ClinVar

Classification Summary

Pathogenic41
Likely Pathogenic3
VUS174
Likely Benign21
Benign11
41
Pathogenic
3
Likely Pathogenic
174
VUS
21
Likely Benign
11
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
41
0
41
Likely Pathogenic
0
0
3
0
3
VUS
0
164
10
0
174
Likely Benign
0
10
4
7
21
Benign
0
1
0
10
11
Total01755817250

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

View in ClinVar →

Protein Context — Lollipop Plot

ADCY9 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold

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