NLRP3

Genetic findings for NLRP3 in Cryopyrin-Associated Periodic Syndromes

Variants (6)

VariantTypeFrequencySignificance
Multiple pathogenic variants (>250)germline~50-70% detectable by SangerGain-of-function mutations causing constitutive inflammasome activation. Most in exon 3 (NACHT domain).
R260WgermlineCommon MWS variantMost frequent MWS-associated variant in French population.
T348MgermlineCommon in severe CAPSAssociated with early onset, chronic course, hearing loss.
Somatic mosaicism variantssomatic40% of mutation-negative CAPSLow-level allele frequency (1.9–45%); can increase over time. Explains 'mutation-negative' CAPS.
De novo mutationsgermline~50-60% of NOMIDSpontaneous new mutations; no family history.
Y861 LRR domain variantsgermlineRareAtypical phenotype with minimal cold-triggered rash.

Clinical implications

Multiple pathogenic variants (>250)

Confirms CAPS diagnosis; genotype-phenotype correlation guides prognosis (certain variants → more severe disease).

Also found in: Schnitzler syndrome (somatic mosaicism, rare) (Rare), Gout (common variants) (Common)

R260W

Typical MWS phenotype with hearing loss risk.

T348M

Higher risk of neurological involvement; may require higher dose IL-1 blockade.

Somatic mosaicism variants

Deep sequencing required for detection; can cause late adult-onset disease.

Also found in: Schnitzler syndrome (Rare)

De novo mutations

Most NOMID cases are sporadic; genetic counseling important.

Y861 LRR domain variants

Non-classical presentation; may be missed by clinical criteria alone.

Related molecules

NLRP3 (Cryopyrin)established

Inflammasome sensor and scaffoldmutated

Related hypotheses

NLRP3 gain-of-function mutations cause constitutive inflammasome activation driving IL-1β-mediated autoinflammation in CAPS

98
leading·15 studies

Somatic NLRP3 mosaicism explains disease in a subset of 'mutation-negative' CAPS patients

72
competing·8 studies

Cold-induced cryo-sensitive aggregation of mutant NLRP3 explains FCAS triggering

38
emerging·2 studies

Modifier genes and epigenetic factors determine CAPS severity within shared genotypes

35
emerging·3 studies

Sources (11)

A1Hoffman HM et al. (2001) Mutation of a new gene encoding a putative pyrin-like protein causes familial cold autoinflammatory syndrome and Muckle-Wells syndrome · Nat GenetPubMed
C2Aksentijevich I et al. (2002) De novo CIAS1 mutations, cytokine activation, and evidence for genetic heterogeneity in patients with NOMID · Arthritis RheumPubMed
B4Booshehri LM et al. (2019) CAPS and NLRP3 · J Clin ImmunolPubMed
J7Cuisset L et al. (2011) Mutations in the autoinflammatory cryopyrin-associated periodic syndrome gene: epidemiological study and lessons from eight years of genetic analysis in France · Ann Rheum DisPubMed
A4Levy R et al. (2015) Phenotypic and genotypic characteristics of CAPS: a series of 136 patients from the Eurofever Registry · Ann Rheum DisPubMed
C3Tanaka N et al. (2011) High incidence of NLRP3 somatic mosaicism in patients with chronic infantile neurologic, cutaneous, articular syndrome · Arthritis RheumPubMed
C4Rowczenio DM et al. (2017) Late-onset cryopyrin-associated periodic syndromes caused by somatic NLRP3 mosaicism — UK single center experience · Front ImmunolPubMed
C5Izawa K et al. (2012) Detection of base substitution-type somatic mosaicism of the NLRP3 gene with >99.9% statistical confidence by massively parallel sequencing · J Mol DiagnPubMed
C7Melo Gomes S et al. (2025) Somatic NLRP3 mosaicism in patients with 'mutation-negative' CAPS: insights from a single centre UK cohort · Front Pediatr
C2Aksentijevich I et al. (2002) De novo CIAS1 mutations, cytokine activation, and evidence for genetic heterogeneity in patients with NOMID · Arthritis RheumPubMed
B4Booshehri LM et al. (2019) CAPS and NLRP3 · J Clin ImmunolPubMed