HEATR1

Chr 1

HEAT repeat containing 1

Also known as: BAP28, UTP10

HEATR1 encodes a ribosome biogenesis factor that is required for processing of pre-18S ribosomal RNA and assembly of the small ribosomal subunit in the nucleolus. Mutations cause autosomal recessive neurodevelopmental disorders with intellectual disability and seizures, typically presenting in early childhood. This gene is highly constrained against loss-of-function variants, reflecting its essential role in ribosome biogenesis.

OMIMResearchSummary from RefSeq, UniProt
LOFmechanismLOEUF 0.14
Clinical SummaryHEATR1
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
0.14LOEUF
pLI 1.000
Z-score 8.81
OE 0.08 (0.040.14)
Highly constrained

Among the most LoF-intolerant genes (~top 3%)

Missense Constraint
0.09Z-score
OE missense 0.99 (0.941.04)
1073 obs / 1081.1 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.08 (0.040.14)
00.351.4
Missense OE0.99 (0.941.04)
00.61.4
Synonymous OE1.02
01.21.6
LoF obs/exp: 8 / 105.7Missense obs/exp: 1073 / 1081.1Syn Z: -0.33
DN
0.3594th %ile
GOF
0.3094th %ile
LOF
0.65top 25%

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

LOFprediction above median · LOEUF 0.14

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

HEATR1 · 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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