LDB3

Chr 10ADAR

LIM domain binding 3

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

This gene encodes a PDZ domain-containing protein. PDZ motifs are modular protein-protein interaction domains consisting of 80-120 amino acid residues. PDZ domain-containing proteins interact with each other in cytoskeletal assembly or with other proteins involved in targeting and clustering of membrane proteins. The protein encoded by this gene interacts with alpha-actinin-2 through its N-terminal PDZ domain and with protein kinase C via its C-terminal LIM domains. The LIM domain is a cysteine-rich motif defined by 50-60 amino acids containing two zinc-binding modules. This protein also interacts with all three members of the myozenin family. Mutations in this gene have been associated with myofibrillar myopathy and dilated cardiomyopathy. Alternatively spliced transcript variants encoding different isoforms have been identified; all isoforms have N-terminal PDZ domains while only longer isoforms (1, 2 and 5) have C-terminal LIM domains. [provided by RefSeq, Jan 2010]

GeneReviewsOMIMResearchGenerating clinical summary…
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
41 unique Pathogenic / Likely Pathogenic· 884 VUS of 1605 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 OE?0.43 (0.280.68)
00.351.4
Missense OE?0.96 (0.891.04)
00.61.4
Synonymous OE?1.07
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

This gene — mechanism propensity

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

1605 submitted variants in ClinVar

Classification Summary

Pathogenic31
Likely Pathogenic10
VUS884
Likely Benign451
Benign84
Conflicting140
31
Pathogenic
10
Likely Pathogenic
884
VUS
451
Likely Benign
84
Benign
140
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
26
2
3
0
31
Likely Pathogenic
6
4
0
0
10
VUS
34
707
102
41
884
Likely Benign
0
38
144
269
451
Benign
0
5
68
11
84
Conflicting
140
Total667563173211,600

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

View in ClinVar →

38 pathogenic / likely-pathogenic (of 49) ClinVar copy-number / structural variants overlap LDB3 — these span large chromosomal regions, not the gene specifically, and are excluded from the counts above. Explore in CNV tools →

Protein Context — Lollipop Plot

LDB3 · protein map & ClinVar variants

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