TSPAN11

Chr 12

tetraspanin 11

Also known as: VSSW1971

TSPAN11 encodes a membrane protein that facilitates focal adhesion assembly to regulate osteoblast alignment and guide bone matrix formation orthogonally to cell orientation. Mutations cause autosomal recessive osteogenesis imperfecta, a bone fragility disorder characterized by fractures, bone deformity, and skeletal abnormalities. The gene shows low constraint against loss-of-function variants (pLI near 0), consistent with its recessive inheritance pattern.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
3
Pubs (1 yr)
32
P/LP submissions
0%
P/LP missense
1.29
LOEUF
Multiple*
Mechanism· predicted
Clinical SummaryTSPAN11
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
32 unique Pathogenic / Likely Pathogenic· 36 VUS of 85 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.29LOEUF
pLI 0.000
Z-score 0.76
OE 0.78 (0.491.29)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.06Z-score
OE missense 0.99 (0.861.13)
156 obs / 158.3 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.78 (0.491.29)
00.351.4
Missense OE0.99 (0.861.13)
00.61.4
Synonymous OE0.90
01.21.6
LoF obs/exp: 11 / 14.1Missense obs/exp: 156 / 158.3Syn Z: 0.68
DN
0.83top 10%
GOF
0.80top 10%
LOF
0.1499th %ile

This gene has evidence for multiple mechanisms of pathogenicity (dominant-negative and gain-of-function). Both the Badonyi & Marsh prediction and the broader genomic evidence point to dominant-negative as the predominant mechanism. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

DNprediction above median
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.

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

ClinVar Variant Classifications

85 submitted variants in ClinVar

Classification Summary

Pathogenic29
Likely Pathogenic3
VUS36
Likely Benign4
29
Pathogenic
3
Likely Pathogenic
36
VUS
4
Likely Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
29
0
29
Likely Pathogenic
0
0
3
0
3
VUS
0
34
2
0
36
Likely Benign
0
3
1
0
4
Benign
0
0
0
0
0
Total03735072

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

View in ClinVar →

Protein Context — Lollipop Plot

TSPAN11 · protein map & ClinVar variants

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

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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