SCRIB

Chr 8

scribble planar cell polarity protein

Also known as: CRIB1, SCRB1, SCRIB1, Vartul, oSCRIB

The SCRIB protein functions as a scaffold protein involved in cell polarization processes and tumor suppression pathways, binding to multiple other proteins through its PDZ domain. Mutations cause autosomal dominant neural tube defects including spina bifida and anencephaly. This gene is highly constrained against loss-of-function variants (pLI = 0.93, LOEUF = 0.31), indicating that complete loss of function is likely not tolerated.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
25
Pubs (1 yr)
3
P/LP submissions
0%
P/LP missense
0.31
LOEUF· LoF intol.
LOF*
Mechanism· G2P
Clinical SummarySCRIB
Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 0.93). One damaged copy is likely sufficient to cause disease.
📋
ClinVar Variants
3 unique Pathogenic / Likely Pathogenic· 110 VUS of 200 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.31LOEUF
pLI 0.929
Z-score 6.21
OE 0.20 (0.130.31)
Highly constrained

Highly LoF-intolerant (top ~10% of genes)

Missense Constraint
0.38Z-score
OE missense 0.97 (0.921.02)
982 obs / 1016.2 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.20 (0.130.31)
00.351.4
Missense OE0.97 (0.921.02)
00.61.4
Synonymous OE1.36
01.21.6
LoF obs/exp: 14 / 70.2Missense obs/exp: 982 / 1016.2Syn Z: -6.03
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
limitedSCRIB-related 8q24.3 deletion-like syndromeLOFAD
DN
0.5280th %ile
GOF
0.74top 25%
LOF
0.59top 25%

This gene has evidence for multiple mechanisms of pathogenicity (loss-of-function and gain-of-function). The Badonyi & Marsh model scores gain-of-function highest among its predictions, but genomic evidence (constraint, ClinVar variant spectrum, and literature) most strongly supports loss-of-function (haploinsufficiency). Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

LOFLOEUF 0.31
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. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

200 submitted variants in ClinVar

Classification Summary

Pathogenic3
VUS110
Likely Benign5
3
Pathogenic
110
VUS
5
Likely Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
3
0
3
Likely Pathogenic
0
0
0
0
0
VUS
0
109
1
0
110
Likely Benign
0
5
0
0
5
Benign
0
0
0
0
0
Total011440118

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

View in ClinVar →

Protein Context — Lollipop Plot

SCRIB · 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 ↗