OBSL1

Chr 2AR

obscurin like cytoskeletal adaptor 1

This gene encodes a cytoskeletal adaptor protein that functions as a core component of the 3M complex, which regulates microtubule dynamics and acts as a regulator of ubiquitin ligase pathways controlling Golgi morphogenesis and dendrite patterning in the brain. Biallelic mutations cause 3-M syndrome type 2, a growth disorder characterized by severe pre- and postnatal growth restriction with distinctive facial features, inherited in an autosomal recessive pattern. The gene shows very low constraint against loss-of-function variants in the general population.

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismARLOEUF 0.881 OMIM phenotype
Clinical SummaryOBSL1
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
0.88LOEUF
pLI 0.000
Z-score 2.42
OE 0.70 (0.550.88)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.01Z-score
OE missense 1.00 (0.951.05)
1170 obs / 1170.7 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.70 (0.550.88)
00.351.4
Missense OE1.00 (0.951.05)
00.61.4
Synonymous OE1.01
01.21.6
LoF obs/exp: 51 / 73.4Missense obs/exp: 1170 / 1170.7Syn Z: -0.19
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveOBSL1-related 3-M syndromeLOFAR

Predictions shown for reference only — model trained on dominant genes, not applicable to AR conditions.

DN
0.6744th %ile
GOF
0.6736th %ile
LOF
0.3940th %ile

The Badonyi & Marsh prediction model was trained exclusively on dominant disease genes. Predictions are not reliable for genes with autosomal recessive inheritance and are shown at reduced opacity for reference only.

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

OBSL1 · 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 →