ACYP2

Chr 2

acylphosphatase 2

Also known as: ACYM, ACYP

Acylphosphatase-2 hydrolyzes phosphoenzyme intermediates of membrane pumps, particularly the Ca2+/Mg2+-ATPase in skeletal muscle sarcoplasmic reticulum, with increased expression during muscle differentiation. This gene is extremely tolerant to loss-of-function variants in the general population, but specific disease associations and inheritance patterns have not been established in the provided data.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
4
Pubs (1 yr)
10
P/LP submissions
0%
P/LP missense
1.84
LOEUF
DN
Mechanism· predicted
Clinical SummaryACYP2
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
10 unique Pathogenic / Likely Pathogenic· 98 VUS of 137 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.84LOEUF
pLI 0.000
Z-score -0.21
OE 1.10 (0.591.84)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.51Z-score
OE missense 1.20 (0.981.48)
63 obs / 52.5 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE1.10 (0.591.84)
00.351.4
Missense OE1.20 (0.981.48)
00.61.4
Synonymous OE1.29
01.21.6
LoF obs/exp: 6 / 5.5Missense obs/exp: 63 / 52.5Syn Z: -0.95
DN
0.6550th %ile
GOF
0.4875th %ile
LOF
0.4135th %ile

The highest-scoring mechanism for this gene is dominant-negative.

DNprediction 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.

ClinVar Variant Classifications

137 submitted variants in ClinVar

Classification Summary

Pathogenic9
Likely Pathogenic1
VUS98
Likely Benign12
Benign13
9
Pathogenic
1
Likely Pathogenic
98
VUS
12
Likely Benign
13
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
9
0
9
Likely Pathogenic
0
0
1
0
1
VUS
0
91
7
0
98
Likely Benign
0
5
5
2
12
Benign
0
6
3
4
13
Total0102256133

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

View in ClinVar →

Protein Context — Lollipop Plot

ACYP2 · 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.

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Clinical Literature
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