CMTM6

Chr 3

CKLF like MARVEL transmembrane domain containing 6

Also known as: CKLFSF6, PRO2219

The CMTM6 protein regulates PD-L1 expression on immune cells by protecting it from degradation and maintaining its presence at the cell surface, thereby controlling immune tolerance and T-cell responses. Mutations in CMTM6 cause autosomal recessive combined immunodeficiency affecting adaptive immunity. The gene shows moderate constraint against loss-of-function variants, and patients typically present with recurrent infections and immune dysregulation in early childhood.

OMIMResearchSummary from RefSeq, UniProt
MultiplemechanismLOEUF 0.88
Clinical SummaryCMTM6
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.19) despite low pLI — interpret in context.
📋
ClinVar Variants
17 unique Pathogenic / Likely Pathogenic· 19 VUS of 40 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.88LOEUF
pLI 0.433
Z-score 1.75
OE 0.19 (0.070.88)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.50Z-score
OE missense 0.85 (0.701.03)
74 obs / 87.3 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.19 (0.070.88)
00.351.4
Missense OE0.85 (0.701.03)
00.61.4
Synonymous OE1.07
01.21.6
LoF obs/exp: 1 / 5.4Missense obs/exp: 74 / 87.3Syn Z: -0.29
DN
0.6453th %ile
GOF
0.79top 10%
LOF
0.2582th %ile

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

GOFprediction above median
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

40 submitted variants in ClinVar

Classification Summary

Pathogenic16
Likely Pathogenic1
VUS19
16
Pathogenic
1
Likely Pathogenic
19
VUS

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
16
0
16
Likely Pathogenic
0
0
1
0
1
VUS
0
17
2
0
19
Likely Benign
0
0
0
0
0
Benign
0
0
0
0
0
Total01719036

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

View in ClinVar →

Protein Context — Lollipop Plot

CMTM6 · 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
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