LDB3

Chr 10ADAR

LIM domain binding 3

Also known as: CMD1C, CMD2L, CMH24, CMPD3, CYPHER, LDB3Z1, LDB3Z4, LVNC3

The encoded protein functions as an adapter in striated muscle, coupling protein kinase C-mediated signaling to the cytoskeleton through its PDZ and LIM domains and interactions with alpha-actinin-2 and myozenin family members. Mutations cause various forms of cardiomyopathy (dilated, hypertrophic, and left ventricular noncompaction) and myofibrillar myopathy, primarily affecting cardiac and skeletal muscle. The gene shows both autosomal dominant and autosomal recessive inheritance patterns.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt
MultiplemechanismAD/ARLOEUF 0.685 OMIM phenotypes
Clinical SummaryLDB3
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Gene-Disease Validity (ClinGen)
arrhythmogenic right ventricular cardiomyopathy · ADDisputed

Disputed — evidence questions this relationship

4 total gene-disease associations curated

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
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ClinVar Variants
31 unique Pathogenic / Likely Pathogenic· 404 VUS of 600 total submissions
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Clinical Trials
3 active or recruiting trials — potential therapeutic options may be available
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GeneReview available — LDB3
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.68LOEUF
pLI 0.000
Z-score 2.99
OE 0.43 (0.280.68)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.29Z-score
OE missense 0.96 (0.891.04)
431 obs / 448.1 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.43 (0.280.68)
00.351.4
Missense OE0.96 (0.891.04)
00.61.4
Synonymous OE1.07
01.21.6
LoF obs/exp: 14 / 32.4Missense obs/exp: 431 / 448.1Syn Z: -0.71
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
limitedLDB3-related dilated cardiomyopathyOTHERAD
limitedLDB3-related arrhythmogenic right ventricular cardiomyopathyOTHERAD
limitedLDB3-related myopathy myofibrillarOTHERAD
DN
0.76top 25%
GOF
0.77top 25%
LOF
0.3452th %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
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.

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

ClinVar Variant Classifications

600 submitted variants in ClinVar

Classification Summary

Pathogenic27
Likely Pathogenic4
VUS404
Likely Benign139
Benign11
Conflicting10
27
Pathogenic
4
Likely Pathogenic
404
VUS
139
Likely Benign
11
Benign
10
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
16
0
11
0
27
Likely Pathogenic
4
0
0
0
4
VUS
18
320
47
19
404
Likely Benign
0
5
54
80
139
Benign
0
1
9
1
11
Conflicting
10
Total38326121100595

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

View in ClinVar →

Protein Context — Lollipop Plot

LDB3 · 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 →