TMEM97

Chr 17

transmembrane protein 97

Also known as: MAC30, S2R, sigma2R

TMEM97 encodes a sigma-2 receptor that regulates cellular cholesterol homeostasis by controlling cholesterol biosynthesis, trafficking, and lysosomal transport, and also functions in membrane trafficking and autophagy. The gene has high tolerance to loss-of-function variants (pLI 0.0003, LOEUF 1.495), and no established human disease phenotype has been reported in the literature. Pathogenic variants would likely follow a dominant-negative mechanism based on predictions.

Summary from RefSeq, UniProt, Mechanism
1
Active trials
12
Pubs (1 yr)
6
P/LP submissions
0%
P/LP missense
1.50
LOEUF
DN
Mechanism· predicted
Clinical SummaryTMEM97
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
6 unique Pathogenic / Likely Pathogenic· 21 VUS of 35 total submissions
💊
Clinical Trials
1 active or recruiting trial — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.50LOEUF
pLI 0.000
Z-score 0.60
OE 0.77 (0.421.50)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.17Z-score
OE missense 0.95 (0.801.13)
89 obs / 93.6 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.77 (0.421.50)
00.351.4
Missense OE0.95 (0.801.13)
00.61.4
Synonymous OE0.94
01.21.6
LoF obs/exp: 6 / 7.8Missense obs/exp: 89 / 93.6Syn Z: 0.29
DN
0.76top 25%
GOF
0.6247th %ile
LOF
0.2680th %ile

The highest-scoring mechanism for this gene is dominant-negative.

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

35 submitted variants in ClinVar

Classification Summary

Pathogenic5
Likely Pathogenic1
VUS21
Likely Benign1
Benign1
5
Pathogenic
1
Likely Pathogenic
21
VUS
1
Likely Benign
1
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
5
0
5
Likely Pathogenic
0
0
1
0
1
VUS
0
18
3
0
21
Likely Benign
0
1
0
0
1
Benign
0
0
1
0
1
Total01910029

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

View in ClinVar →

Protein Context — Lollipop Plot

TMEM97 · protein map & ClinVar variants

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

Clinical Literature
Open Research Assistant →
Full-Text Mentions
NLP-detected gene mentions in article bodies · via PubTator3
PubTator3
Top 5 full-text resultsSearch PubTator3 ↗