SPRED1

Chr 15AD

sprouty related EVH1 domain containing 1

Also known as: LGSS, NFLS, PPP1R147, hSpred1, spred-1

The SPRED1 protein regulates activation of the MAP kinase cascade by forming homodimers or heterodimers with SPRED2, and is phosphorylated by tyrosine kinases in response to growth factors. Loss-of-function mutations cause Legius syndrome (also known as neurofibromatosis type 1-like syndrome), which follows autosomal dominant inheritance. The pathogenic mechanism involves loss of normal MAP kinase regulation due to reduced SPRED1 function.

OMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
LOFmechanismADLOEUF 0.321 OMIM phenotype
VCEP Guidelines: NeurofibromatosesIn Progress
ClinGen Panel
Clinical SummarySPRED1
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Gene-Disease Validity (ClinGen)
Legius syndrome · 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 0.97). 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
0.32LOEUF
pLI 0.972
Z-score 4.00
OE 0.12 (0.060.32)
Highly constrained

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

Missense Constraint
1.17Z-score
OE missense 0.79 (0.700.89)
194 obs / 245.6 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.12 (0.060.32)
00.351.4
Missense OE0.79 (0.700.89)
00.61.4
Synonymous OE0.80
01.21.6
LoF obs/exp: 3 / 24.3Missense obs/exp: 194 / 245.6Syn Z: 1.42
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveSPRED1-related Legius syndromeLOFAD
DN
0.4091th %ile
GOF
0.4381th %ile
LOF
0.64top 25%

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

LOFprediction above median · 1 literature citation · LOEUF 0.32

Literature Evidence

LOFGermline loss-of-function mutations in SPRED1 cause a neurofibromatosis 1-like phenotypePMID:17704776

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

SPRED1 · 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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Full-Text Mentions
NLP-detected gene mentions in article bodies · via PubTator3
PubTator3
Top 5 full-text resultsSearch PubTator3 ↗