ERI1

Chr 8AR

exoribonuclease 1

Also known as: 3'HEXO, HEXO, HXAL, SEMDGC, SEMDGS, THEX1

The protein is a 3'-5' exonuclease that degrades histone mRNAs after DNA replication and processes 5.8S ribosomal RNA, playing a key role in chondrogenesis. Biallelic mutations cause Hoxha-Aliu syndrome and spondyloepimetaphyseal dysplasia (Guo-Campeau type), both skeletal dysplasias with autosomal recessive inheritance. The gene shows tolerance to loss-of-function variants (pLI near 0), consistent with the recessive inheritance pattern.

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismARLOEUF 1.072 OMIM phenotypes
Clinical SummaryERI1
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — 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
1.07LOEUF
pLI 0.000
Z-score 1.35
OE 0.63 (0.391.07)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-1.38Z-score
OE missense 1.30 (1.161.46)
217 obs / 167.0 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.63 (0.391.07)
00.351.4
Missense OE1.30 (1.161.46)
00.61.4
Synonymous OE1.53
01.21.6
LoF obs/exp: 10 / 15.8Missense obs/exp: 217 / 167.0Syn Z: -3.23
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
moderateERI1-related severe growth restriction and skeletal dysplasiaLOFAR
moderateERI1-related brachydactyly and mild neurodevelopmental delayLOFAR

Predictions shown for reference only — model trained on dominant genes, not applicable to AR conditions.

DN
0.6259th %ile
GOF
0.5465th %ile
LOF
0.3164th %ile

The Badonyi & Marsh prediction model was trained exclusively on dominant disease genes. Predictions are not reliable for genes with autosomal recessive inheritance and are shown at reduced opacity for reference only.

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

ERI1 · 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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