TMEM131L

Chr 4

transmembrane 131 like

Also known as: KIAA0922

The TMEM131L protein is a membrane-associated regulator that antagonizes canonical Wnt signaling by promoting lysosome-dependent degradation of Wnt-activated LRP6 and regulates thymocyte proliferation in the thymus. Mutations cause autosomal recessive severe combined immunodeficiency with microcephaly, growth retardation, and sensitivity to ionizing radiation (SCIDMGR), which presents in early infancy with severe immunodeficiency affecting T cell development. This gene is highly constrained against loss-of-function variants, indicating that haploinsufficiency is not tolerated in the general population.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
0
Pubs (1 yr)
27
P/LP submissions
0%
P/LP missense
0.30
LOEUF· LoF intol.
Mechanism
Clinical SummaryTMEM131L
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.
📋
ClinVar Variants
27 unique Pathogenic / Likely Pathogenic· 235 VUS of 306 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.30LOEUF
pLI 0.960
Z-score 6.67
OE 0.20 (0.130.30)
Highly constrained

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

Missense Constraint
0.69Z-score
OE missense 0.93 (0.880.99)
798 obs / 855.0 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.20 (0.130.30)
00.351.4
Missense OE0.93 (0.880.99)
00.61.4
Synonymous OE1.04
01.21.6
LoF obs/exp: 16 / 80.6Missense obs/exp: 798 / 855.0Syn Z: -0.60

ClinVar Variant Classifications

306 submitted variants in ClinVar

Classification Summary

Pathogenic24
Likely Pathogenic3
VUS235
Likely Benign17
Benign2
24
Pathogenic
3
Likely Pathogenic
235
VUS
17
Likely Benign
2
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
24
0
24
Likely Pathogenic
0
0
3
0
3
VUS
0
234
1
0
235
Likely Benign
0
14
1
2
17
Benign
0
1
0
1
2
Total0249293281

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

View in ClinVar →

Protein Context — Lollipop Plot

TMEM131L · 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.

Search ClinicalTrials.gov →
Clinical Literature
Open Research Assistant →
Key Publications
Landmark & review papers · by relevance
PubMed
Top 1 results · since 2015Search PubMed ↗
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC