CLTRN

Chr X

collectrin, amino acid transport regulator

Also known as: NX-17, NX17, TMEM27

The encoded protein traffics amino acid transporters to the apical brush border of proximal tubules and regulates SNARE complex formation in pancreatic beta cells for insulin exocytosis. Mutations cause Hartnup disorder, an autosomal recessive condition affecting neutral amino acid transport in the kidneys and intestines. The gene shows low constraint to loss-of-function variation, consistent with the relatively mild phenotype of Hartnup disorder.

OMIMResearchSummary from RefSeq, UniProt
MultiplemechanismLOEUF 1.75
Clinical SummaryCLTRN
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
36 unique Pathogenic / Likely Pathogenic· 36 VUS of 100 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.75LOEUF
pLI 0.000
Z-score 0.09
OE 0.96 (0.521.75)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.28Z-score
OE missense 1.09 (0.911.31)
81 obs / 74.2 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.96 (0.521.75)
00.351.4
Missense OE1.09 (0.911.31)
00.61.4
Synonymous OE0.79
01.21.6
LoF obs/exp: 6 / 6.2Missense obs/exp: 81 / 74.2Syn Z: 0.82
DN
0.6840th %ile
GOF
0.6737th %ile
LOF
0.2970th %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

100 submitted variants in ClinVar

Classification Summary

Pathogenic36
VUS36
Likely Benign2
36
Pathogenic
36
VUS
2
Likely Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
36
0
36
Likely Pathogenic
0
0
0
0
0
VUS
0
22
14
0
36
Likely Benign
0
2
0
0
2
Benign
0
0
0
0
0
Total02450074

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

View in ClinVar →

Protein Context — Lollipop Plot

CLTRN · 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
Open Research Assistant →
Full-Text Mentions
NLP-detected gene mentions in article bodies · via PubTator3
PubTator3
Top 5 full-text resultsSearch PubTator3 ↗
Key Publications
Landmark & review papers · by relevance
PubMed
Top 2 results · since 2015Search PubMed ↗
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC