PIEZO2

Chr 18ADAR

piezo type mechanosensitive ion channel component 2

Also known as: C18orf30, C18orf58, DA3, DA5, DAIPT, FAM38B, FAM38B2, HsT748

The protein functions as a mechanically-activated cation channel that converts mechanical forces into biological signals and mediates touch and proprioceptive sensation in somatosensory neurons. Mutations cause distal arthrogryposis (types 3 and 5) and arthrogryposis with impaired proprioception and touch, following both autosomal dominant and autosomal recessive inheritance patterns. The pathogenic mechanism involves gain-of-function mutations that disrupt normal mechanosensation.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
LOFmechanismAD/ARLOEUF 0.374 OMIM phenotypes
Clinical SummaryPIEZO2
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.28) despite low pLI — interpret in context.
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Clinical Trials
2 active or recruiting trials — potential therapeutic options may be available
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GeneReview available — PIEZO2
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Missense constrained — critical functional residues
LoF Constraint
0.37LOEUF
pLI 0.000
Z-score 7.85
OE 0.28 (0.220.37)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
3.44Z-score
OE missense 0.75 (0.710.78)
1081 obs / 1450.1 exp
Constrained

Highly missense-constrained (top ~0.1%)

Observed / Expected Ratios
LoF OE0.28 (0.220.37)
00.351.4
Missense OE0.75 (0.710.78)
00.61.4
Synonymous OE0.87
01.21.6
LoF obs/exp: 40 / 140.4Missense obs/exp: 1081 / 1450.1Syn Z: 2.31
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitivePIEZO2-related arthrogryposis, distalLOFAD
definitivePIEZO2-related ataxia, dysmetria, contractures and scoliosis with normal cognition but loss of discriminative touch perceptionLOFAR
DN
0.7227th %ile
GOF
0.79top 25%
LOF
0.2971th %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 · 1 literature citation
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.

Literature Evidence

GOFGain-of-function mutations in the human PIEZO2 gene cause three clinical types of autosomal dominant distal arthrogryposis.PMID:28728825

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

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

PIEZO2 · protein map & ClinVar variants

Showing all ClinVar variants across the protein. Search a specific variant to highlight its position.

3D Protein StructureAlphaFold
Clinical Literature
Open Research Assistant →
Key Publications
Landmark & review papers · by relevance
PubMed
Top 5 results · since 2015Search PubMed ↗