Abstract

BackgroundKikuchi-Fujimoto's disease (KFD), also called histiocytic necrotizing lymphadenitis, is a rare, idiopathic and self-limited condition usually characterized by cervical lymphadenopathy and fever, most often affecting young patients. Aetiology is unknown. Differential diagnosis includes mainly malignant lymphoma, tuberculous lymphadenitis and systemic lupus erythematosus (SLE), so early diagnosis is crucial. Pleuropulmonary involvement due to isolated KFD has been seldom reported.Case Presentationa 32-year-old man, on treatment for iatrogenic hypothyroidism, was admitted due to high grade fever and painful cervical lymphadenopathies. KFD was diagnosed by lymph node biopsy. Some days after admission the patient got worse, he developed generalized lymphadenopathy, bilateral pleural effusion and interstitial lung disease. All of them resolved with prednisone and after two years of following up he remains asymptomatic and without evidence of any other associated disease.ConclusionPleural effusion and interstitial lung disease are very uncommon manifestations of KFD. In our experience, treatment with oral prednisone was effective.

Highlights

  • Kikuchi-Fujimoto’s disease (KFD), called histiocytic necrotizing lymphadenitis, is a rare, idiopathic and self-limited condition usually characterized by cervical lymphadenopathy and fever, most often affecting young patients

  • Case Report A 32-year-old Caucasian man, with a history of hyperthyroidism six months previous due to toxic multinodular goiter, treated with radioactive iodine, who developed iatrogenic hypothyroidism and on initiation of substitutive treatment, was admitted due to persistent fever (39-40°C), malaise and painful cervical lymphadenopathies that had been present for two weeks

  • Thoracocentesis was performed with the following results in pleural fluid: pH 7.39 (6.8-7.6), glucose 102 mg/dl (60-100), proteins 3.2 g/dl (0-3), lactic dehydrogenase (LDH) 1694 IU/L (0200), 50 leukocytes/ml (0-300); stains and cultures for bacteria and mycobacteria were negative and malignant cells were not found on cytologic examination

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Summary

Background

Kikuchi-Fujimoto’s disease (KFD), or histiocytic necrotizing lymphadenitis, was first described in Japan in 1972 as a self-limiting disease mostly affecting the cervical lymph nodes of young individuals, mainly females [1]. Chest X-ray was clear in both lung fields and computed tomography of the neck, thorax and abdomen revealed only lymphadenopathy affecting bilateral cervical and one mediastinal lymph nodes (Figure 1). The patient’s condition worsened over the following days, with high-grade daily fever, increasing size of cervical lymph nodes, the development of axillary lymphadenopathy and mild dyspnoea on exertion, with decreased breath sounds in both lung bases on physical examination. Thoracocentesis was performed with the following results in pleural fluid: pH 7.39 (6.8-7.6), glucose 102 mg/dl (60-100), proteins 3.2 g/dl (0-3), LDH 1694 IU/L (0200), 50 leukocytes/ml (0-300); stains and cultures for bacteria and mycobacteria were negative and malignant cells were not found on cytologic examination. After two years of follow-up appointments the patient remains asymptomatic, physical examination and chest X-ray are absolutely normal and serum antinuclear antibodies remain negative

Discussion
20. Sharma OP
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