TRPC5

Chr X

transient receptor potential cation channel subfamily C member 5

Also known as: PPP1R159, TRP5

The protein forms a receptor-activated non-selective calcium permeant cation channel that regulates neuronal calcium influx and controls hypothalamic-mediated behaviors including feeding, anxiety, socialization, and maternal care. Loss-of-function mutations cause autosomal dominant neurodevelopmental disorders with extremely high constraint scores indicating severe intolerance to protein-truncating variants. The pathogenic mechanism involves disrupted calcium signaling leading to impaired neuronal function and altered innate behaviors.

Summary from RefSeq, UniProt
0
Active trials
40
Pubs (1 yr)
12
P/LP submissions
0%
P/LP missense
0.17
LOEUF· LoF intol.
GOF
Mechanism· predicted
Clinical SummaryTRPC5
Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 1.00). One damaged copy is likely sufficient to cause disease.
📋
ClinVar Variants
12 unique Pathogenic / Likely Pathogenic· 81 VUS of 300 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Dual constrained — LoF & missense intolerant
LoF Constraint
0.17LOEUF
pLI 1.000
Z-score 4.72
OE 0.04 (0.010.17)
Highly constrained

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

Missense Constraint
3.27Z-score
OE missense 0.52 (0.470.59)
197 obs / 375.4 exp
Constrained

Highly missense-constrained (top ~0.1%)

Observed / Expected Ratios
LoF OE0.04 (0.010.17)
00.351.4
Missense OE0.52 (0.470.59)
00.61.4
Synonymous OE1.04
01.21.6
LoF obs/exp: 1 / 27.9Missense obs/exp: 197 / 375.4Syn Z: -0.41
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
limitedTRPC5-related neurodevelopmental disorderOTHERXLR
DN
0.5081th %ile
GOF
0.7030th %ile
LOF
0.60top 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 0.17
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

300 submitted variants in ClinVar

Classification Summary

Pathogenic12
VUS81
Likely Benign65
Benign2
Conflicting1
12
Pathogenic
81
VUS
65
Likely Benign
2
Benign
1
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
12
0
12
Likely Pathogenic
0
0
0
0
0
VUS
2
75
4
0
81
Likely Benign
0
5
8
52
65
Benign
0
0
0
2
2
Conflicting
1
Total2802454161

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

View in ClinVar →

Protein Context — Lollipop Plot

TRPC5 · 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
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