MEFV

Genetic findings for MEFV in Familial Mediterranean Fever

Variants (5)

VariantTypeFrequencySignificance
M694V (c.2080A>G, p.Met694Val)germline~40% of disease alleles (most common)The most common and most severe FMF mutation. Located in exon 10 (B30.2/SPRY domain). Homozygosity associated with earliest onset, highest attack frequency, most arthritis, and greatest risk of AA amyloidosis.
M680I (c.2040G>A/C, p.Met680Ile)germline~11% of disease allelesThird most common FMF mutation. Located in exon 10. Homozygosity or compound heterozygosity with M694V associated with severe disease. M680I/M694V combination produces severe phenotype.
V726A (c.2177T>C, p.Val726Ala)germline~14% of disease allelesSecond most common FMF mutation. Located in exon 10. Heterozygosity generally associated with milder phenotype. However, V726A-E148Q complex allele can be severe.
E148Q (c.442G>C, p.Glu148Gln)germline~3% of disease alleles (controversial pathogenicity)Located in exon 2. Highly controversial — debated whether pathogenic variant or benign polymorphism. Extremely high frequency in general population. When part of a complex allele with V726A, significantly increases disease severity.
M694I (c.2082G>A/C, p.Met694Ile)germline~3% of disease allelesLocated in exon 10. Fifth founder mutation. Homozygosity associated with severe disease. M694I/M694I genotype identified as a specific risk factor for AA amyloidosis in Algerian patients.

Clinical implications

M694V (c.2080A>G, p.Met694Val)

M694V homozygotes require early aggressive treatment. Higher risk of colchicine resistance. Close monitoring for amyloidosis essential.

M680I (c.2040G>A/C, p.Met680Ile)

Severe disease when homozygous or compound with M694V. More frequent in patients with skin eruptions (erysipelas-like erythema).

V726A (c.2177T>C, p.Val726Ala)

Usually milder disease when heterozygous. Complex alleles with E148Q can cause severe phenotype including amyloidosis.

E148Q (c.442G>C, p.Glu148Gln)

Clinical significance uncertain when found alone. Should not be used as sole basis for FMF diagnosis. Complex alleles containing E148Q merit attention.

M694I (c.2082G>A/C, p.Met694Ile)

Severe phenotype when homozygous, especially regarding amyloidosis risk.

Related molecules

Pyrin (MEFV)established

Inflammasome sensor protein (mutated in FMF)Gain-of-function mutations

Related hypotheses

High carrier frequency of MEFV mutations reflects heterozygote advantage against intracellular pathogens, particularly Yersinia pestis

65
leading·15 studies

FMF heterozygotes can manifest clinical disease through a pseudo-dominant or dose-dependent mechanism involving gene modifiers and environmental triggers

50
emerging·25 studies