TMEM67

Chr 8AR

transmembrane protein 67

Also known as: JBTS6, MECKELIN, MKS3, NPHP11, TNEM67

The protein localizes to the primary cilium and plasma membrane where it regulates ciliary structure, function, and length, controls centrosome migration and duplication, and participates in Wnt signaling pathways. Autosomal recessive mutations cause a spectrum of ciliopathies including Meckel syndrome type 3, Joubert syndrome type 6, COACH syndrome, and nephronophthisis 11, likely through loss of normal ciliary function. The protein is also associated with RHYNS syndrome and acts as a modifier in Bardet-Biedl syndrome.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
LOFmechanismARLOEUF 0.946 OMIM phenotypes
Clinical SummaryTMEM67
🧬
Gene-Disease Validity (ClinGen)
ciliopathy · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
37 unique Pathogenic / Likely Pathogenic· 81 VUS of 200 total submissions
💊
Clinical Trials
1 active or recruiting trial — potential therapeutic options may be available
📖
GeneReview available — TMEM67
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.94LOEUF
pLI 0.000
Z-score 1.92
OE 0.74 (0.580.94)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.49Z-score
OE missense 0.94 (0.871.01)
483 obs / 514.4 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.74 (0.580.94)
00.351.4
Missense OE0.94 (0.871.01)
00.61.4
Synonymous OE0.88
01.21.6
LoF obs/exp: 46 / 62.3Missense obs/exp: 483 / 514.4Syn Z: 1.27
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveTMEM67-related nephronophthisisOTHERAR
definitiveTMEM67-related COACH syndromeLOFAR

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

DN
0.6552th %ile
GOF
0.5856th %ile
LOF
0.3259th %ile

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.

ClinVar Variant Classifications

200 submitted variants in ClinVar

Classification Summary

Pathogenic18
Likely Pathogenic19
VUS81
Likely Benign43
Benign3
Conflicting1
18
Pathogenic
19
Likely Pathogenic
81
VUS
43
Likely Benign
3
Benign
1
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
12
1
5
0
18
Likely Pathogenic
14
5
0
0
19
VUS
2
72
6
1
81
Likely Benign
0
1
19
23
43
Benign
0
0
3
0
3
Conflicting
1
Total28793324165

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

View in ClinVar →

Protein Context — Lollipop Plot

TMEM67 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
Clinical Literature
Open Research Assistant →