INTU

Chr 4AR

inturned planar cell polarity protein

Also known as: CPLANE4, INT, OFD17, PDZD6, PDZK6, SRTD20

The INTU protein regulates cilia formation by controlling basal body positioning and apical actin cytoskeleton organization, functioning as a core component of the CPLANE complex essential for normal ciliogenesis. Biallelic mutations cause orofaciodigital syndrome XVII and short-rib thoracic dysplasia with polydactyly, both congenital ciliopathies affecting craniofacial structures, digits, and the thoracic cage with autosomal recessive inheritance. This gene shows extremely low tolerance to loss-of-function variants (pLI near 1), indicating strong evolutionary constraint.

OMIMResearchSummary from RefSeq, OMIM, UniProt
ARLOEUF 0.742 OMIM phenotypes
Clinical SummaryINTU
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Gene-Disease Validity (ClinGen)
INTU-related skeletal ciliopathy · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

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
0.74LOEUF
pLI 0.000
Z-score 2.91
OE 0.52 (0.370.74)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.38Z-score
OE missense 0.95 (0.881.03)
463 obs / 486.8 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.52 (0.370.74)
00.351.4
Missense OE0.95 (0.881.03)
00.61.4
Synonymous OE0.89
01.21.6
LoF obs/exp: 22 / 42.5Missense obs/exp: 463 / 486.8Syn Z: 1.17

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

INTU · 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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Full-Text Mentions
NLP-detected gene mentions in article bodies · via PubTator3
PubTator3
Top 5 full-text resultsSearch PubTator3 ↗