CAPZA3

Chr 12

capping actin protein of muscle Z-line subunit alpha 3

Also known as: CAPPA3, Gsg3, HEL-S-86

The protein caps the fast-growing ends of actin filaments in a calcium-independent manner and is predominantly expressed in sperm where it contributes to sperm architecture and male fertility. Mutations cause autosomal recessive sensorineural hearing loss with onset in infancy or early childhood. The gene shows low constraint to loss-of-function variants, consistent with its autosomal recessive inheritance pattern.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
0
Pubs (1 yr)
38
P/LP submissions
0%
P/LP missense
1.19
LOEUF
DN
Mechanism· predicted
Clinical SummaryCAPZA3
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
38 unique Pathogenic / Likely Pathogenic· 47 VUS of 85 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.19LOEUF
pLI 0.001
Z-score 1.17
OE 0.60 (0.331.19)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.26Z-score
OE missense 1.06 (0.931.21)
164 obs / 155.0 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.60 (0.331.19)
00.351.4
Missense OE1.06 (0.931.21)
00.61.4
Synonymous OE1.13
01.21.6
LoF obs/exp: 6 / 10.0Missense obs/exp: 164 / 155.0Syn Z: -0.80
DN
0.6358th %ile
GOF
0.3094th %ile
LOF
0.2971th %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

85 submitted variants in ClinVar

Classification Summary

Pathogenic37
Likely Pathogenic1
VUS47
37
Pathogenic
1
Likely Pathogenic
47
VUS

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
37
0
37
Likely Pathogenic
0
0
1
0
1
VUS
0
45
2
0
47
Likely Benign
0
0
0
0
0
Benign
0
0
0
0
0
Total04540085

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

View in ClinVar →

Protein Context — Lollipop Plot

CAPZA3 · 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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