SLC12A1

Chr 15AR

solute carrier family 12 member 1

Also known as: BSC, BSC-1, BSC1, CCC2, NKCC2

This gene encodes a kidney-specific sodium-potassium-chloride cotransporter that is expressed on the luminal membrane of renal epithelial cells of the thick ascending limb of Henle's loop and the macula densa. It plays a key role in concentrating urine and accounts for most of the NaCl resorption. It is sensitive to such diuretics as furosemide and bumetanide. Some Bartter-like syndromes result from defects in this gene. Alternative splicing results in multiple transcript variants encoding distinct isoforms. Additional splice variants have been described but their biological validity in humans has not been experimentally proven.[provided by RefSeq, May 2010]

Primary Disease Associations & Inheritance

Bartter syndrome, type 1MIM #601678
AR
UniProtBartter syndrome 1, antenatal
1
Active trials
15
Pubs (1 yr)
66
P/LP submissions
10%
P/LP missense
0.82
LOEUF
Multiple*
Mechanism· predicted
Clinical SummarySLC12A1
🧬
Gene-Disease Validity (ClinGen)
obsolete antenatal Bartter syndrome · 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
61 unique Pathogenic / Likely Pathogenic· 171 VUS of 500 total submissions
💊
Clinical Trials
1 active or recruiting trial — potential therapeutic options may be available
📖
GeneReview available — SLC12A1
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
0.82LOEUF
pLI 0.000
Z-score 2.63
OE 0.62 (0.470.82)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
1.16Z-score
OE missense 0.86 (0.800.93)
502 obs / 580.7 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.62 (0.470.82)
00.351.4
Missense OE0.86 (0.800.93)
00.61.4
Synonymous OE1.05
01.21.6
LoF obs/exp: 35 / 56.3Missense obs/exp: 502 / 580.7Syn Z: -0.60
DN
0.77top 25%
GOF
0.79top 10%
LOF
0.2288th %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

500 submitted variants in ClinVar

Classification Summary

Pathogenic23
Likely Pathogenic38
VUS171
Likely Benign250
Benign4
Conflicting6
23
Pathogenic
38
Likely Pathogenic
171
VUS
250
Likely Benign
4
Benign
6
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
5
0
18
0
23
Likely Pathogenic
26
6
6
0
38
VUS
2
153
14
2
171
Likely Benign
0
1
119
130
250
Benign
0
0
4
0
4
Conflicting
6
Total33160161132492

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

View in ClinVar →

Protein Context — Lollipop Plot

SLC12A1 · protein map & ClinVar variants

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

Clinical Literature
Open Research Assistant →
Key Publications
Landmark & review papers · by relevance
PubMed
Top 5 results · since 2015Search PubMed ↗