SNX19

Chr 11

sorting nexin 19

Also known as: CHET8

SNX19 encodes a sorting nexin protein that regulates intracellular vesicle trafficking and exocytosis, particularly maintaining insulin-containing dense core vesicles in pancreatic beta-cells and facilitating insulin secretion. Biallelic mutations cause autosomal recessive early-onset diabetes mellitus with additional features including microcephaly and intellectual disability. The gene is not highly constrained against loss-of-function variants, consistent with the recessive inheritance pattern observed in affected individuals.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
2
Pubs (1 yr)
81
P/LP submissions
0%
P/LP missense
0.86
LOEUF
Multiple*
Mechanism· predicted
Clinical SummarySNX19
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
81 unique Pathogenic / Likely Pathogenic· 147 VUS of 264 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.86LOEUF
pLI 0.000
Z-score 2.27
OE 0.61 (0.440.86)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
-0.58Z-score
OE missense 1.07 (1.001.14)
600 obs / 561.4 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.61 (0.440.86)
00.351.4
Missense OE1.07 (1.001.14)
00.61.4
Synonymous OE1.10
01.21.6
LoF obs/exp: 24 / 39.4Missense obs/exp: 600 / 561.4Syn Z: -1.22
DN
0.6454th %ile
GOF
0.6637th %ile
LOF
0.2969th %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

264 submitted variants in ClinVar

Classification Summary

Pathogenic74
Likely Pathogenic7
VUS147
Likely Benign12
Benign3
74
Pathogenic
7
Likely Pathogenic
147
VUS
12
Likely Benign
3
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
74
0
74
Likely Pathogenic
0
0
7
0
7
VUS
1
142
4
0
147
Likely Benign
0
8
0
4
12
Benign
0
3
0
0
3
Total1153854243

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

View in ClinVar →

Protein Context — Lollipop Plot

SNX19 · protein map & ClinVar variants

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

Clinical Trials

Active and recruiting trials from ClinicalTrials.gov

No active trials found for this gene.

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Clinical Literature
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