SLC6A20

Chr 3

solute carrier family 6 member 20

Also known as: IMINO, SIT1, XT3, Xtrp3

The protein functions as a sodium- and chloride-dependent transporter that mediates uptake of imino acids (particularly proline) and glycine, primarily expressed in kidney, small intestine, and brain where it regulates amino acid homeostasis and modulates NMDAR currents. Mutations cause hyperglycinuria and iminoglycinuria, which are autosomal recessive aminoaciduria disorders characterized by excessive urinary excretion of glycine and imino acids. The gene shows high tolerance to loss-of-function variation (pLI nearly zero, LOEUF 1.26), consistent with the recessive inheritance pattern of associated disorders.

OMIMResearchSummary from RefSeq, UniProt
MultiplemechanismLOEUF 1.26
Clinical SummarySLC6A20
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
7 unique Pathogenic / Likely Pathogenic· 197 VUS of 275 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.26LOEUF
pLI 0.000
Z-score 0.44
OE 0.91 (0.671.26)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.50Z-score
OE missense 0.93 (0.851.01)
333 obs / 359.5 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.91 (0.671.26)
00.351.4
Missense OE0.93 (0.851.01)
00.61.4
Synonymous OE1.09
01.21.6
LoF obs/exp: 27 / 29.6Missense obs/exp: 333 / 359.5Syn Z: -0.88
DN
0.7133th %ile
GOF
0.80top 10%
LOF
0.2287th %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

275 submitted variants in ClinVar

Classification Summary

Pathogenic6
Likely Pathogenic1
VUS197
Likely Benign29
Benign36
Conflicting2
6
Pathogenic
1
Likely Pathogenic
197
VUS
29
Likely Benign
36
Benign
2
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
6
0
6
Likely Pathogenic
0
0
1
0
1
VUS
4
109
74
10
197
Likely Benign
0
6
12
11
29
Benign
0
3
29
4
36
Conflicting
2
Total411812225271

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

View in ClinVar →

Protein Context — Lollipop Plot

SLC6A20 · 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 →