Donaire-Castaños Juan Carlos, Demelo-Rodríguez Pablo, Ordieres-Ortega Lucía, Pardo-Sánchez Susana, Galeano-Valle Francisco
Department of Internal Medicine, Hospital General Universitario Gregorio Marañón, Madrid, ESP.
School of Medicine, Universidad Complutense de Madrid, Madrid, ESP.
Cureus. 2024 Nov 8;16(11):e73283. doi: 10.7759/cureus.73283. eCollection 2024 Nov.
Kikuchi-Fujimoto disease (KFD) and adult-onset Still disease (AOSD) are two rare conditions whose association poses a significant diagnostic challenge. KFD is characterized by subacute necrotizing lymphadenitis of unknown etiology, primarily affecting young adults, and often presents with fever and posterior cervical lymphadenopathy. AOSD is a systemic inflammatory disorder of unclear origin, defined by high-spiking fever, lymphadenopathy, hepatosplenomegaly, hyperferritinemia, and leukocytosis. To date, only 15 cases of the coexistence of both conditions have been reported, providing valuable insight into their pathophysiology. We present the case of a 32-year-old Moroccan woman with prolonged fever, arthralgia, and axillary and mediastinal lymphadenopathy. After extensive evaluation, a lymph node biopsy confirmed the diagnosis of KFD, and she met the Yamaguchi criteria for AOSD. Treatment with oral prednisone was initiated, leading to rapid resolution of fever and normalization of acute-phase reactants. This case aligns with previously documented patterns in the literature, indicating a potential shared pathogenic mechanism. The lack of specific autoantibodies in these overlap cases emphasizes the need for clinicians to look for atypical clinical presentations when diagnosing these conditions. While corticosteroids have shown effective symptomatic control in the 16 reported cases, further research is necessary to explore targeted therapies, as many patients lack adequate follow-up regarding long-term management and response to treatment.
菊池-藤本病(KFD)和成人斯蒂尔病(AOSD)是两种罕见疾病,它们的关联给诊断带来了重大挑战。KFD的特征是病因不明的亚急性坏死性淋巴结炎,主要影响年轻人,常表现为发热和颈后淋巴结病。AOSD是一种病因不明的全身性炎症性疾病,其定义为高热、淋巴结病、肝脾肿大、高铁蛋白血症和白细胞增多。迄今为止,仅报告了15例两种疾病共存的病例,这为它们的病理生理学提供了有价值的见解。我们报告一例32岁摩洛哥女性病例,该患者长期发热、关节痛,伴有腋窝和纵隔淋巴结病。经过广泛评估,淋巴结活检确诊为KFD,且她符合AOSD的山口标准。开始口服泼尼松治疗后,发热迅速消退,急性期反应物恢复正常。该病例与文献中先前记录的模式相符,表明可能存在共同的致病机制。这些重叠病例中缺乏特异性自身抗体,这强调临床医生在诊断这些疾病时需要寻找非典型临床表现。虽然皮质类固醇在16例报告病例中显示出有效的症状控制,但由于许多患者缺乏关于长期管理和治疗反应的充分随访,因此有必要进一步研究以探索靶向治疗方法。