RBM44

Chr 2

RNA binding motif protein 44

RBM44 encodes a protein that functions as a component of intercellular bridges during meiosis, structures that connect differentiating germ cells, and is predicted to be involved in mRNA splicing via the spliceosome. Pathogenic variants in RBM44 cause neurodevelopmental disorder with seizures and nonepileptic paroxysms, which follows an autosomal dominant inheritance pattern. This gene is highly constrained against loss-of-function variants, indicating intolerance to protein-truncating mutations.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
0
Pubs (1 yr)
81
P/LP submissions
0%
P/LP missense
0.51
LOEUF
Multiple*
Mechanism· predicted
Clinical SummaryRBM44
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.32) despite low pLI — interpret in context.
📋
ClinVar Variants
79 unique Pathogenic / Likely Pathogenic· 131 VUS of 231 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.51LOEUF
pLI 0.001
Z-score 4.01
OE 0.32 (0.210.51)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
0.26Z-score
OE missense 0.97 (0.901.04)
467 obs / 483.1 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.32 (0.210.51)
00.351.4
Missense OE0.97 (0.901.04)
00.61.4
Synonymous OE0.90
01.21.6
LoF obs/exp: 13 / 40.6Missense obs/exp: 467 / 483.1Syn Z: 1.01
DN
0.6938th %ile
GOF
0.6540th %ile
LOF
0.3552th %ile

This gene has evidence for multiple mechanisms of pathogenicity (dominant-negative and gain-of-function). Both the Badonyi & Marsh prediction and the broader genomic evidence point to dominant-negative as the predominant mechanism. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

DNprediction above median
GOFprediction 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

231 submitted variants in ClinVar

Classification Summary

Pathogenic78
Likely Pathogenic1
VUS131
Likely Benign16
78
Pathogenic
1
Likely Pathogenic
131
VUS
16
Likely Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
78
0
78
Likely Pathogenic
0
0
1
0
1
VUS
0
121
10
0
131
Likely Benign
0
15
0
1
16
Benign
0
0
0
0
0
Total0136891226

LoF = frameshift, stop gained/lost, canonical splice · Counts from ClinVar esearch · Updated hourly

View in ClinVar →

Protein Context — Lollipop Plot

RBM44 · protein map & ClinVar variants

Showing all ClinVar variants across the protein. Search a specific variant to highlight its position.

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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Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC