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Behcet-Like Familial Autoinflammatory Syndrome

OMIM ID:

autosomal dominant

Behcet-Like Familial Autoinflammatory Syndrome

Alternate Names

AISBL
Behcet-like syndrome

Defective Genes

TNFAIP3

Clinical Characteristics

Ocular Features

A minority of reported patients (3 of 14) have had Behcet-like uveitis with retinal vasculitis and chorioretinal lesions.  In two of the 3 patients with uveitis the inflammation was anterior but the authors reported significant visual loss.

Systemic Features

Fourteen patients in 6 unrelated families have been reported.  Onset usually occurs during the first or second decades of life.  Patients usually developed oral and genital ulcers.  Other features variably present were polyarthritis, a skin rash and inflammatory disease or ulcerations in the GI tract.  Several patients had periodic fevers and hemolytic anemia was present in one.  The majority of individuals have been female.

Three patients in a single family had lupus anticoagulant and three others had antinuclear antibodies.

Genetics

Inheritance

This syndrome results from heterozygous mutations in the TNFAIP3 gene (6q23.3) with loss of function leading to A20 haploinsufficiency.  Changes in the TNFAIP3 gene have also been identified in rheumatoid arthritis, systemic lupus erythematosis, and idiopathic juvenile arthritis.

Pedigree

Autosomal dominant

Autosomal dominant disorders require only one mutation for the disease to be expressed.  Since an affected parent has two chromosomes, only one of which has the mutant gene, parents can expect that half (50%) of their children will receive that one and inherit the disease.  It is common for individuals that inherit the mutation, however, to not have evidence of the disease (nonpenetrance).

Image
Sample pedigree of autosomal dominant inheritance

Autosomal dominant inheritance leads to a vertical pattern of transmission

Treatment & Management

Several patients have responded positively to treatment with tumor necrosis factor inhibitors or colchicine.

Selected Resources

Web Resources

Publications

Displaying 1 - 1 of 1

Loss-of-function mutations in TNFAIP3 leading to A20 haploinsufficiency cause an early-onset autoinflammatory disease

PubMedID: 266422433