Colchicine-responsive protracted gouty arthritis with systemic inflammatory reactions
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CASE REPORT
Colchicine-responsive protracted gouty arthritis with systemic inflammatory reactions Fumiaki Nonaka • Kiyoshi Migita • Tomoko Haramura • Remi Sumiyoshi Atsushi Kawakami • Katsumi Eguchi
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Received: 21 August 2012 / Accepted: 12 November 2012 Ó Japan College of Rheumatology 2013
Abstract Acute gouty arthritis is a severe but self-limiting arthritis caused by inflammatory responses to urate crystals. Oral colchicines are effective for initial stages or prophylaxis, but generally, colchicines are ineffective for established gouty arthritis. We describe an unusual case of gouty arthritis with systemic inflammatory reactions, including high fever and polymyalgia. Refractory polyarthritis and high fever were eradicated by colchicine treatment. Genetic analysis revealed a heterozygous mutation in exon 2 of the MEFV gene (E148Q). This case underscores the possibility that MEFV gene mutations may modify the phenotype of gouty arthritis. Keywords
Colchicine Gouty arthritis MEFV gene
Introduction Gout is the most common inflammatory arthritis and is characterized by recurrent arthritic attacks of intra-articular monosodium urate deposition [1]. Multiple lines of evidence support the hypothesis that inflammasome-related genes are disease-susceptible candidate genes for gout [2, 3].
F. Nonaka T. Haramura R. Sumiyoshi K. Eguchi (&) Department of Rheumatology, Sasebo City General Hospital, 9-3 Hirase, Sasebo 857-8511, Japan e-mail: [email protected] K. Migita Clinical Research Center, NHO National Nagasaki Medical ˜ mura 856-8652, Japan Center, Kubara 2-1001-1, O A. Kawakami Department of Rheumatology, Nagasaki University Hospital, Nagasaki, Japan
Familial Mediterranean fever (FMF) is a hereditary autoinflammatory disease, caused by mutations in MEFV, the gene encoding the protein pyrin, which can act as a regulator of inflammasome function [4]. Recent studies indicated that the MEFV gene is associated with other conditions apart from typical FMF and is linked to additional clinical presentations within the family of connective tissue diseases [5, 6]. Gouty arthritis and FMF rarely coexist. We describe an unusual case of gouty arthritis with systemic inflammatory reactions, including persisting high fever and protracted polyarthritis.
Case report A 59-year-old male patient was admitted to our hospital for polyarthritis and persistent high fever ([38.5 °C). Seven years earlier, he developed recurrent episodes of polymyalgia and polyarthralgia that subsided spontaneously within a couple of weeks. There was no medical history of periodic fever among his family. His first symptoms, present 2 weeks prior to hospital admission, were symmetrical arthralgia in the wrist and elbow joints with joint swelling, which developed to polyarthralgia of the symmetrical upper and lower extremities (elbows, knees, ankles and metatarsophalangeal joint). He visited the local clinic because of the persistent polyarthralgia and accompanying polymyalgia and high fever. He was diagnosed with palindromic rheum
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