SPTBN4

Chr 19AR

spectrin beta, non-erythrocytic 4

Also known as: CMND, NEDHND, QV, SPNB4, SPTBN3

Spectrin beta 4 is an actin crosslinking and molecular scaffold protein that links the plasma membrane to the actin cytoskeleton and localizes to the nuclear matrix, PML nuclear bodies, and cytoplasmic vesicles. Mutations cause a neurodevelopmental disorder with hypotonia, neuropathy, and deafness inherited in an autosomal recessive pattern. This gene is extremely intolerant to loss-of-function variants (pLI >0.99, LOEUF 0.19), indicating that complete loss of protein function is likely deleterious.

OMIMResearchSummary from RefSeq, OMIM
LOFmechanismARLOEUF 0.191 OMIM phenotype
Clinical SummarySPTBN4
🧬
Gene-Disease Validity (ClinGen)
neurodevelopmental disorder with hypotonia, neuropathy, and deafness · ARDefinitive

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

Dual constrained — LoF & missense intolerant
LoF Constraint
0.19LOEUF
pLI 1.000
Z-score 8.48
OE 0.12 (0.080.19)
Highly constrained

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

Missense Constraint
3.93Z-score
OE missense 0.70 (0.660.74)
958 obs / 1367.4 exp
Constrained

Highly missense-constrained (top ~0.1%)

Observed / Expected Ratios
LoF OE0.12 (0.080.19)
00.351.4
Missense OE0.70 (0.660.74)
00.61.4
Synonymous OE0.85
01.21.6
LoF obs/exp: 13 / 108.1Missense obs/exp: 958 / 1367.4Syn Z: 2.79
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveSPTBN4-related neurodevelopmental disorder with hypotonia, neuropathy, and deafnessLOFAR

Predictions shown for reference only — model trained on dominant genes, not applicable to AR conditions.

DN
0.5279th %ile
GOF
0.75top 25%
LOF
0.51top 25%

The Badonyi & Marsh prediction model was trained exclusively on dominant disease genes. Predictions are not reliable for genes with autosomal recessive inheritance and are shown at reduced opacity for reference only.

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

SPTBN4 · 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 →
Key Publications
Landmark & review papers · by relevance
PubMed
Top 5 results · since 2015Search PubMed ↗
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC