R3HDM1

Chr 2

R3H domain containing 1

Also known as: R3HDM

R3HDM1 encodes a protein that binds RNA and is highly constrained against loss-of-function variants (pLI 0.86, LOEUF 0.33). Mutations cause autosomal recessive intellectual disability with microcephaly and seizures, typically presenting in early infancy. The gene appears to be critical for normal brain development and function.

OMIMResearchSummary from RefSeq
LOFmechanismLOEUF 0.33
Clinical SummaryR3HDM1
Population Constraint (gnomAD)
Moderately constrained gene (pLI 0.86) — some intolerance to loss-of-function variants.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.33LOEUF
pLI 0.861
Z-score 5.69
OE 0.20 (0.130.33)
Highly constrained

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

Missense Constraint
1.85Z-score
OE missense 0.79 (0.730.85)
463 obs / 589.1 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.20 (0.130.33)
00.351.4
Missense OE0.79 (0.730.85)
00.61.4
Synonymous OE1.10
01.21.6
LoF obs/exp: 12 / 59.3Missense obs/exp: 463 / 589.1Syn Z: -1.14
DN
0.4190th %ile
GOF
0.2895th %ile
LOF
0.75top 10%

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

LOFprediction above median · LOEUF 0.33

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

R3HDM1 · 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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Full-Text Mentions
NLP-detected gene mentions in article bodies · via PubTator3
PubTator3
Top 2 full-text resultsSearch PubTator3 ↗