INO80E

Chr 16

INO80 complex subunit E

Also known as: CCDC95

The INO80E protein is a regulatory component of the INO80 chromatin remodeling complex, which controls gene transcription, DNA replication, and DNA repair. Mutations cause autosomal recessive developmental delay with hypotonia and dysmorphic features, typically presenting in early childhood with motor delays and characteristic facial features. The gene shows minimal constraint against loss-of-function variants based on population data.

ResearchSummary from RefSeq, UniProt
DNmechanismLOEUF 0.95
Clinical SummaryINO80E
Population Constraint (gnomAD)
Low constraint (pLI 0.01) — loss-of-function variants are relatively tolerated in the population.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.95LOEUF
pLI 0.009
Z-score 1.69
OE 0.45 (0.230.95)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
-0.25Z-score
OE missense 1.06 (0.931.22)
138 obs / 129.8 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.45 (0.230.95)
00.351.4
Missense OE1.06 (0.931.22)
00.61.4
Synonymous OE1.18
01.21.6
LoF obs/exp: 5 / 11.1Missense obs/exp: 138 / 129.8Syn Z: -1.07
DN
0.6549th %ile
GOF
0.3690th %ile
LOF
0.57top 25%

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

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

INO80E · 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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