ATXN7L1

Chr 7

ataxin 7 like 1

Also known as: ATXN7L4

The protein is a component of the SAGA (Spt-Ada-Gcn5-acetyltransferase) transcriptional coactivator complex, functioning as a histone deubiquitinase that regulates gene expression through chromatin modification. Mutations cause cone-rod dystrophy with hearing loss, an autosomal recessive disorder affecting retinal photoreceptors and auditory function. This gene is highly constrained against loss-of-function variants, indicating that even single functional copies are insufficient for normal development.

LOFmechanismLOEUF 0.22
Clinical SummaryATXN7L1
Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 1.00). One damaged copy is likely sufficient to cause disease.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.22LOEUF
pLI 0.999
Z-score 4.59
OE 0.07 (0.030.22)
Highly constrained

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

Missense Constraint
2.30Z-score
OE missense 0.70 (0.640.77)
336 obs / 477.3 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.07 (0.030.22)
00.351.4
Missense OE0.70 (0.640.77)
00.61.4
Synonymous OE0.87
01.21.6
LoF obs/exp: 2 / 28.4Missense obs/exp: 336 / 477.3Syn Z: 1.44
DN
0.3395th %ile
GOF
0.2099th %ile
LOF
0.87top 5%

The highest-scoring mechanism for this gene is loss-of-function (haploinsufficiency).

LOFprediction above median · LOEUF 0.22

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

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

ATXN7L1 · 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.

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Clinical Literature
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