BEX1

Chr X

brain expressed X-linked 1

Also known as: BEX2, HBEX2, HGR74-h

The BEX1 protein functions as a signaling adapter in p75NTR/NGFR neurotrophin receptor signaling and serves as a transcriptional regulator that modulates neuronal differentiation and cell cycle progression. Mutations in BEX1 cause X-linked intellectual disability with seizures and behavioral abnormalities, affecting primarily males with onset in early childhood. The gene shows moderate constraint against loss-of-function variants, and carrier females may exhibit milder symptoms due to X-inactivation.

Summary from RefSeq, UniProt
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0
Active trials
12
Pubs (1 yr)
0
P/LP submissions
P/LP missense
1.22
LOEUF
GOF
Mechanism· predicted
Clinical SummaryBEX1
Population Constraint (gnomAD)
Moderately constrained gene (pLI 0.54) — some intolerance to loss-of-function variants.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
1.22LOEUF
pLI 0.543
Z-score 1.41
OE 0.00 (0.001.22)
Moderately constrained

Highly tolerant — LoF variants common in population

Missense Constraint
0.37Z-score
OE missense 0.85 (0.661.10)
42 obs / 49.4 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.00 (0.001.22)
00.351.4
Missense OE0.85 (0.661.10)
00.61.4
Synonymous OE1.29
01.21.6
LoF obs/exp: 0 / 2.3Missense obs/exp: 42 / 49.4Syn Z: -0.92
DN
0.6065th %ile
GOF
0.83top 10%
LOF
0.3843th %ile

The highest-scoring mechanism for this gene is gain-of-function.

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

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

BEX1 · 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