MID2

Chr XXLR

midline 2

Also known as: FXY2, MRX101, RNF60, TRIM1, XLID101

MID2 encodes an E3 ubiquitin ligase that mediates microtubule stabilization and regulates LRRK2 protein degradation in neurons through polyubiquitination. Mutations cause X-linked intellectual developmental disorder with males being affected and females typically being carriers. The gene shows moderate constraint against loss-of-function variants (LOEUF 0.496), suggesting some tolerance to protein-disrupting changes.

Summary from RefSeq, OMIM, UniProt
Research Assistant →

Primary Disease Associations & Inheritance

?Intellectual developmental disorder, X-linked 101MIM #300928
XLR
0
Active trials
8
Pubs (1 yr)
0
P/LP submissions
P/LP missense
0.50
LOEUF
Multiple*
Mechanism· predicted
Clinical SummaryMID2
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.25) despite low pLI — interpret in context.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.50LOEUF
pLI 0.175
Z-score 3.39
OE 0.25 (0.140.50)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
1.58Z-score
OE missense 0.74 (0.660.83)
217 obs / 293.3 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.25 (0.140.50)
00.351.4
Missense OE0.74 (0.660.83)
00.61.4
Synonymous OE0.86
01.21.6
LoF obs/exp: 6 / 23.9Missense obs/exp: 217 / 293.3Syn Z: 1.12
DN
0.7034th %ile
GOF
0.7028th %ile
LOF
0.3551th %ile

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

GOFprediction above median
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

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

MID2 · 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.

Search ClinicalTrials.gov →
Clinical Literature
Open Research Assistant →