DSG2

Chr 18ADAR

desmoglein 2

Also known as: CDHF5, HDGC

This gene encodes desmoglein-2, a calcium-binding transmembrane glycoprotein that forms desmosomes mediating cell-cell adhesion in cardiac, epithelial, and other tissues. Mutations cause arrhythmogenic right ventricular dysplasia and dilated cardiomyopathy, presenting as cardiac arrhythmias and heart failure primarily affecting the myocardium. The condition follows both autosomal dominant and autosomal recessive inheritance patterns.

Summary from RefSeq, OMIM, UniProt
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Primary Disease Associations & Inheritance

Arrhythmogenic right ventricular dysplasia 10MIM #610193
AD
Cardiomyopathy, dilated, 1BBMIM #612877
AR
1
Active trials
58
Pubs (1 yr)
54
P/LP submissions
2%
P/LP missense
0.79
LOEUF
Multiple*
Mechanism· predicted
Clinical SummaryDSG2
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Gene-Disease Validity (ClinGen)
arrhythmogenic right ventricular cardiomyopathy · ADDefinitive

Definitive — sufficient evidence for diagnostic panels

2 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
53 unique Pathogenic / Likely Pathogenic· 301 VUS of 500 total submissions
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Clinical Trials
1 active or recruiting trial — potential therapeutic options may be available
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GeneReview available — DSG2
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
0.79LOEUF
pLI 0.000
Z-score 2.61
OE 0.55 (0.390.79)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.30Z-score
OE missense 0.97 (0.901.03)
567 obs / 587.2 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.55 (0.390.79)
00.351.4
Missense OE0.97 (0.901.03)
00.61.4
Synonymous OE0.94
01.21.6
LoF obs/exp: 22 / 39.7Missense obs/exp: 567 / 587.2Syn Z: 0.67
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
limitedDSG2-related dilated cardiomyopathyOTHERAD
definitiveDSG2-related arrhythmogenic right ventricular cardiomyopathyOTHERAD
definitiveDSG2-related arrhythmogenic right ventricular cardiomyopathyOTHERAR
DN
0.7034th %ile
GOF
0.82top 10%
LOF
0.2968th %ile

This gene has evidence for multiple mechanisms of pathogenicity (dominant-negative, gain-of-function and loss-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 dominant-negative. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

DNprediction above median · 1 literature citation
GOFprediction above median
LOF1 literature citation · 57% of P/LP variants are LoF

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

DNThe results suggested a dominant-negative effect of the mutated DSG2 proteins because they were incorporated into the desmosomes.PMID:23381804
LOFBecause the mutation creates a premature termination codon, mutant transcripts were predicted to be rapidly degraded by the nonsense-mediated mRNA decay (NMD) pathway. This would then suggest that haploinsufficiency for desmoglein-2 is not the mechanism for disease.PMID:16773573

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

500 submitted variants in ClinVar

Classification Summary

Pathogenic29
Likely Pathogenic24
VUS301
Likely Benign128
Benign1
Conflicting6
29
Pathogenic
24
Likely Pathogenic
301
VUS
128
Likely Benign
1
Benign
6
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
11
0
18
0
29
Likely Pathogenic
19
1
4
0
24
VUS
10
269
19
3
301
Likely Benign
0
6
33
89
128
Benign
0
0
1
0
1
Conflicting
6
Total402767592489

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

View in ClinVar →

Protein Context — Lollipop Plot

DSG2 · protein map & ClinVar variants

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

Clinical Literature
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