COL5A2

Chr 2AD

collagen type V alpha 2 chain

Also known as: EDSC, EDSCL2

The COL5A2 protein forms part of type V collagen, which regulates the assembly of connective tissue fibers containing both type I and type V collagen and is found throughout nearly all tissues. Mutations cause classic Ehlers-Danlos syndrome type 2 with autosomal dominant inheritance, affecting connective tissues and typically presenting in childhood. This gene is extremely intolerant to loss-of-function variants (pLI >0.99, LOEUF 0.097), indicating that most protein-disrupting variants are likely pathogenic.

OMIMResearchSummary from RefSeq, OMIM, UniProt
DNmechanismADLOEUF 0.101 OMIM phenotype
Clinical SummaryCOL5A2
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Gene-Disease Validity (ClinGen)
Ehlers-Danlos syndrome, classic type · ADDefinitive

Definitive — sufficient evidence for diagnostic panels

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
0.10LOEUF
pLI 1.000
Z-score 8.63
OE 0.04 (0.020.10)
Highly constrained

Among the most LoF-intolerant genes (~top 3%)

Missense Constraint
2.44Z-score
OE missense 0.77 (0.720.82)
679 obs / 883.0 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.04 (0.020.10)
00.351.4
Missense OE0.77 (0.720.82)
00.61.4
Synonymous OE0.99
01.21.6
LoF obs/exp: 4 / 94.5Missense obs/exp: 679 / 883.0Syn Z: 0.16
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveCOL5A2-related Ehlers-Danlos syndrome, classic typeDNAD
DN
0.4983th %ile
GOF
0.3293th %ile
LOF
0.71top 10%

The highest-scoring mechanism for this gene is loss-of-function (haploinsufficiency).

LOFprediction above median · 1 literature citation · LOEUF 0.10

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

LOFHemizygous deletion of COL3A1, COL5A2, and MSTN causes a complex phenotype with aortic dissection: a lesson for and from true haploinsufficiency.PMID:20648054

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

COL5A2 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold

Clinical Trials

Active and recruiting trials from ClinicalTrials.gov

No active trials found for this gene.

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Clinical Literature
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