GJA8

Chr 1AD

gap junction protein alpha 8

Also known as: CAE, CAE1, CTRCT1, CX50, CZP1, MP70

This gene encodes connexin 50, a transmembrane protein that forms gap junction channels connecting lens fiber cells and is essential for lens growth and maturation. Mutations cause autosomal dominant cataracts of multiple types, including zonular pulverulent cataracts, nuclear progressive cataracts, and cataract-microcornea syndrome. The gene shows minimal constraint against loss-of-function variants (pLI 0.002), suggesting tolerance to complete protein loss.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt
MultiplemechanismADLOEUF 1.001 OMIM phenotype
Clinical SummaryGJA8
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
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ClinVar Variants
173 unique Pathogenic / Likely Pathogenic· 84 VUS of 300 total submissions
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GeneReview available — GJA8
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
1.00LOEUF
pLI 0.002
Z-score 1.58
OE 0.51 (0.281.00)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
-0.20Z-score
OE missense 1.03 (0.941.14)
270 obs / 261.0 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.51 (0.281.00)
00.351.4
Missense OE1.03 (0.941.14)
00.61.4
Synonymous OE1.18
01.21.6
LoF obs/exp: 6 / 11.9Missense obs/exp: 270 / 261.0Syn Z: -1.57
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveGJA8-related cataractOTHERAD
DN
0.79top 25%
GOF
0.85top 5%
LOF
0.2483th %ile

This gene has evidence for multiple mechanisms of pathogenicity (dominant-negative and gain-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

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

DNMD simulation revealed that the introduction of the deletion destabilized the Cx50 gap junction channel, indicating the deletion as a dominant-negative mutation.PMID:26996484

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

ClinVar Variant Classifications

300 submitted variants in ClinVar

Classification Summary

Pathogenic142
Likely Pathogenic31
VUS84
Likely Benign19
Benign7
Conflicting16
142
Pathogenic
31
Likely Pathogenic
84
VUS
19
Likely Benign
7
Benign
16
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
6
136
0
142
Likely Pathogenic
2
17
12
0
31
VUS
8
67
9
0
84
Likely Benign
0
4
1
14
19
Benign
0
1
6
0
7
Conflicting
16
Total109516414299

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

View in ClinVar →

Protein Context — Lollipop Plot

GJA8 · 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.

Search ClinicalTrials.gov →
Clinical Literature
Open Research Assistant →
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC