Case Report

Enthesitis-related arthritis in Kikuchi-Fujimoto disease

Authors

Yogesh Preet Singh¹, Vikas Agarwal¹, Narendra Krishnani² and Ramnath Misra¹

1. Department of Clinical Immunology, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Lucknow, 226014, India
2. Department of Pathology, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Lucknow, 226014, India

Received:

7 May 2007

Accepted:

19 March 2008

Published online:

10 May 2008

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Abstract

Histiocytic necrotizing lymphadenitis or Kikuchi-Fujimoto disease (KFD) is a rare, benign and self-limiting disorder that characteristically presents with fever and cervical lymphadenopathy. Articular manifestations in the form of arthralgias are common but frank arthritis is distinctly rare and dactylitis has not been reported yet. Herein, we describe a young boy who presented with arthritis and dactylitis as the initial manifestation of KFD. A 14-year-old boy presented with a two-week history of fever, generalized lymphadenopathy and asymmetric polyarthritis, enthesitis and dactylitis of the toes. Two years earlier he presented with arthritis of the knee and ankle joints, which lasted for 12 months. However, he had been asymptomatic for one year. Investigations revealed anemia, leukopenia and raised acute phase reactants. Work-up for infectious etiology, systemic lupus erythematosus and leukemia and lymphoma was negative. Excision biopsy of the cervical lymph node confirmed KFD. Fever, lymphadenopathy and leukopenia dissipated with nonsteroidal anti inflammatory drug therapy, but the arthritis persisted. A trial of methotrexate led to the resolution of the arthritis.

Key words

Enthesitis-related arthritis - Dactylitis - Leukopenia - Necrotizing lymphadenitis - Kikuchi-Fujimoto disease (KFD) - Juvenile idiopathic arthritis (JIA)