LRCH1

Chr 13

leucine rich repeats and calponin homology domain containing 1

Also known as: CHDC1, NP81

This protein acts as a negative regulator of the GTPase CDC42 by sequestering the CDC42-guanine exchange factor DOCK8, thereby inhibiting CD4+ T-cell migration. Mutations in LRCH1 cause neurodevelopmental disorders with intellectual disability and variable features including autism spectrum disorder, seizures, and motor delays. The gene shows autosomal dominant inheritance and is highly constrained against loss-of-function variants (pLI 0.996, LOEUF 0.279).

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
0
Pubs (1 yr)
0
P/LP submissions
P/LP missense
0.28
LOEUF· LoF intol.
Multiple*
Mechanism· predicted
Clinical SummaryLRCH1
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.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.28LOEUF
pLI 0.996
Z-score 5.18
OE 0.14 (0.080.28)
Highly constrained

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

Missense Constraint
1.39Z-score
OE missense 0.81 (0.740.88)
334 obs / 413.4 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.14 (0.080.28)
00.351.4
Missense OE0.81 (0.740.88)
00.61.4
Synonymous OE0.93
01.21.6
LoF obs/exp: 6 / 42.5Missense obs/exp: 334 / 413.4Syn Z: 0.74
DN
0.5477th %ile
GOF
0.6444th %ile
LOF
0.64top 25%

This gene has evidence for multiple mechanisms of pathogenicity (loss-of-function and gain-of-function). Both the Badonyi & Marsh prediction and the broader genomic evidence point to loss-of-function as the predominant mechanism. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

LOFprediction above median · LOEUF 0.28
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

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

LRCH1 · 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 →