Abstract
Introduction: Kikuchi-Fujimoto’s disease (KFD) or histiocytic necrotizing lymphadenitis is a benign and self-limited disease of unknown etiology mainly affecting young women. Although the association with systemic lupus erythematosus (SLE) is well described, no case of drug-induced lupus erythematosus (DILE) associated KFD has not been reported so far. Case report: We herein report a 25-year old Caucasian woman, with no medical history and no medication except for oral estrogen-progestin contraception (levonorgestrel-ethinylestradiol), who presented with cervical lymphadenopathy, fever and arthralgia without weight loss, night sweats or skin involvement. An exhaustive infectious disease screening was negative and lymph node biopsy revealed histiocytic necrotizing lymphadenitis suggesting KFD. Autoimmune screening tests evidenced high titers of anti-histone antibodies suggesting DILE induced by estrogen-progestin medication. The patient received a short course of non-steroidal anti-inflammatory treatment for painful lymphadenitis and arthralgia. Oral levonorgestrel-ethinylestradiol contraceptive medication was stopped and KFD and DILE completely recovered with a long-term disappearance of anti-histone antibodies. Conclusion: We report the first case of KFD associated-DILE following oral levonorgestrel-ethinylestradiol medication. Even though levonorgestrel-ethinylestradiol induced lupus is well known, the association with KFD has never been reported and the physiopathology remained unknown. Keywords: Kikuchi-Fujimoto disease, Lupus, Levonorgestrel-ethinylestradiol
Highlights
Kikuchi-Fujimoto’s disease (KFD) or histiocytic necrotizing lymphadenitis is a benign and self-limited disease of unknown etiology mainly affecting young women
We report the first case of KFD associated-drug-induced lupus erythematosus (DILE) following oral levonorgestrel-ethinylestradiol medication
The diagnosis is made by lymph node histology: histiocytic necrotizing lymphadenitis which is characterized by patchy areas of necrosis, with eosinophilic materials and nuclear debris and the absence of neutrophils [1, 2]
Summary
We report the first case of KFD associated-DILE following oral levonorgestrelethinylestradiol medication.
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