SGPL1

Chr 10AR

sphingosine-1-phosphate lyase 1

Also known as: NPHS14, RENI, S1PL, SPL

The protein cleaves sphingosine-1-phosphate into fatty aldehydes and phosphoethanolamine, playing a critical role in sphingolipid metabolism, lipid homeostasis, and neuronal autophagy regulation. Mutations cause RENI syndrome, an autosomal recessive disorder characterized by renal involvement (nephrotic syndrome) and neurological manifestations. The gene shows high constraint against loss-of-function variants (LOEUF 0.554), reflecting its essential cellular functions.

OMIMResearchSummary from RefSeq, OMIM, UniProt
DNmechanismARLOEUF 0.551 OMIM phenotype
Clinical SummarySGPL1
🧬
Gene-Disease Validity (ClinGen)
nephrotic syndrome 14 · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.33) despite low pLI — interpret in context.
💊
Clinical Trials
1 active or recruiting trial — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.55LOEUF
pLI 0.004
Z-score 3.45
OE 0.33 (0.200.55)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
1.82Z-score
OE missense 0.71 (0.640.80)
227 obs / 318.5 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.33 (0.200.55)
00.351.4
Missense OE0.71 (0.640.80)
00.61.4
Synonymous OE0.97
01.21.6
LoF obs/exp: 10 / 30.6Missense obs/exp: 227 / 318.5Syn Z: 0.28
DN
0.7326th %ile
GOF
0.5562th %ile
LOF
0.3261th %ile

The highest-scoring mechanism for this gene is dominant-negative.

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

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

SGPL1 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
Clinical Literature
Open Research Assistant →