RS1

Chr XXLR

retinoschisin 1

Also known as: RS, XLRS1

The RS1 protein is secreted by retinal photoreceptors and bipolar cells as oligomeric complexes that bind membrane lipids and mediate cell-cell adhesion to maintain normal retinal structure. Mutations cause X-linked retinoschisis, an early-onset macular degeneration in males characterized by splitting of the inner retinal layers and severe vision loss. This gene is highly constrained against loss-of-function variants (pLI 0.96, LOEUF 0.30) and follows X-linked recessive inheritance.

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismXLRLOEUF 0.302 OMIM phenotypes
Clinical SummaryRS1
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Gene-Disease Validity (ClinGen)
X-linked retinoschisis · XLDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 0.96). One damaged copy is likely sufficient to cause disease.
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Clinical Trials
4 active or recruiting trials — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.30LOEUF
pLI 0.957
Z-score 2.91
OE 0.00 (0.000.30)
Highly constrained

Highly LoF-intolerant (top ~10% of genes)

Missense Constraint
0.97Z-score
OE missense 0.73 (0.610.89)
75 obs / 102.6 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.00 (0.000.30)
00.351.4
Missense OE0.73 (0.610.89)
00.61.4
Synonymous OE0.82
01.21.6
LoF obs/exp: 0 / 9.9Missense obs/exp: 75 / 102.6Syn Z: 0.92

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

RS1 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
Clinical Literature
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