MXD4

Chr 4

MAX dimerization protein 4

Also known as: MAD4, MST149, MSTP149, bHLHc12

The MXD4 protein is a transcriptional repressor that heterodimerizes with MAX protein to form DNA-binding complexes, antagonizing MYC transcriptional activity and suppressing cell transformation. Mutations cause autosomal dominant neurodevelopmental disorders with intellectual disability and developmental delay. This gene shows high constraint against loss-of-function variants, suggesting that even single functional copies may be insufficient for normal development.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
7
Pubs (1 yr)
134
P/LP submissions
0%
P/LP missense
0.51
LOEUF
DN
Mechanism· predicted
Clinical SummaryMXD4
Population Constraint (gnomAD)
Moderately constrained gene (pLI 0.78) — some intolerance to loss-of-function variants.
📋
ClinVar Variants
134 unique Pathogenic / Likely Pathogenic· 41 VUS of 187 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.51LOEUF
pLI 0.776
Z-score 2.52
OE 0.11 (0.040.51)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
0.74Z-score
OE missense 0.81 (0.690.96)
97 obs / 119.7 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.11 (0.040.51)
00.351.4
Missense OE0.81 (0.690.96)
00.61.4
Synonymous OE1.28
01.21.6
LoF obs/exp: 1 / 9.3Missense obs/exp: 97 / 119.7Syn Z: -1.61
DN
0.6649th %ile
GOF
0.4381th %ile
LOF
0.61top 25%

The highest-scoring mechanism for this gene is dominant-negative.

DNprediction 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

187 submitted variants in ClinVar

Classification Summary

Pathogenic128
Likely Pathogenic6
VUS41
Likely Benign1
128
Pathogenic
6
Likely Pathogenic
41
VUS
1
Likely Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
128
0
128
Likely Pathogenic
0
0
6
0
6
VUS
0
28
13
0
41
Likely Benign
0
0
0
1
1
Benign
0
0
0
0
0
Total0281471176

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

View in ClinVar →

Protein Context — Lollipop Plot

MXD4 · 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.

Search ClinicalTrials.gov →
Clinical Literature
Open Research Assistant →
Key Publications
Landmark & review papers · by relevance
PubMed
Top 5 results · since 2015Search PubMed ↗