MIB2

Chr 1

MIB E3 ubiquitin protein ligase 2

Also known as: ZZANK1, ZZZ5

The encoded protein is an E3 ubiquitin-protein ligase that mediates ubiquitination of Delta receptors to positively regulate Notch signaling through receptor endocytosis. Mutations cause autosomal recessive developmental delay, intellectual disability, and distinctive facial features. This gene is highly constrained against loss-of-function variants (LOEUF 0.62), suggesting that complete protein loss is likely pathogenic.

Summary from RefSeq, UniProt
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0
Active trials
13
Pubs (1 yr)
0
P/LP submissions
P/LP missense
0.62
LOEUF
GOF
Mechanism· predicted
Clinical SummaryMIB2
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.62LOEUF
pLI 0.000
Z-score 3.59
OE 0.42 (0.290.62)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.62Z-score
OE missense 0.93 (0.871.00)
644 obs / 690.1 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.42 (0.290.62)
00.351.4
Missense OE0.93 (0.871.00)
00.61.4
Synonymous OE1.24
01.21.6
LoF obs/exp: 19 / 45.0Missense obs/exp: 644 / 690.1Syn Z: -3.36
DN
0.6064th %ile
GOF
0.6834th %ile
LOF
0.4234th %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

MIB2 · 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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