Division of Rheumatology, Saint Louis University School of Medicine, Saint Louis, MO, USA.
Department of Medicine, Quaid e Azam Medical College, Bahawalpur, Pakistan.
Am J Case Rep. 2024 Aug 29;25:e943658. doi: 10.12659/AJCR.943658.
BACKGROUND Periodic fever, aphthous stomatitis, pharyngitis, and cervical adenitis (PFAPA) syndrome is an autoinflammatory fever syndrome primarily seen in children under age 5 years, and its etiology is unknown. Most cases are resolved by the age of 10 years, and it is rare in adults. PFAPA is characterized by recurrent episodes of fever associated with pharyngitis, stomatitis, and cervical adenitis, although not all clinical features are present at initial evaluation. Diagnosis is made clinically, as there are no specific biomarkers available. Treatment includes prednisone, colchicine, interleukin-1 blockers, and tonsillectomy. We report a case of adult-onset PFAPA syndrome that responded to colchicine. CASE REPORT A 22-year-old woman presented to the Rheumatology Clinic for evaluation of recurrent fevers associated with sore throat and enlarged painful cervical lymph nodes. She was symptom-free between the episodes. Workup for infectious causes and autoinflammatory/autoimmune diseases was unremarkable. Various differential diagnoses were considered, due to her unusual presentation. After all were ruled out, PFAPA was diagnosed based on her symptoms, and she started steroids, to which she had a dramatic response and resolution of symptoms. She was then transitioned to oral colchicine, which significantly decreased flare frequency. CONCLUSIONS Being aware of PFAPA syndrome in adults is vital. A timely diagnosis can significantly improve a patient's quality of life. This case highlights the importance of considering PFAPA syndrome in the differential diagnosis of periodic febrile illnesses in adults and the role of Colchicine as prophylaxis. Larger studies are needed to understand etiopathogenesis better and develop other effective therapeutics.
周期性发热、口疮性口炎、咽炎和颈部淋巴结炎(PFAPA)综合征是一种主要发生在 5 岁以下儿童的自身炎症性发热综合征,其病因不明。大多数病例在 10 岁时得到解决,在成年人中很少见。PFAPA 的特征是反复出现发热,伴有咽炎、口疮性口炎和颈部淋巴结炎,尽管初始评估时并非所有临床特征都存在。由于没有特异性生物标志物,因此通过临床诊断。治疗包括泼尼松、秋水仙碱、白细胞介素-1 阻滞剂和扁桃体切除术。我们报告一例成人发病的 PFAPA 综合征,对秋水仙碱有反应。
一名 22 岁女性因反复发热伴有咽痛和增大的疼痛性颈部淋巴结就诊于风湿病诊所。她在发作之间无症状。感染原因和自身炎症/自身免疫性疾病的检查均无异常。由于她的不寻常表现,考虑了各种不同的诊断。在排除所有其他诊断后,根据她的症状诊断为 PFAPA,她开始接受类固醇治疗,症状明显缓解。然后她转为口服秋水仙碱,显著降低了发作频率。
了解成人中的 PFAPA 综合征至关重要。及时诊断可以显著提高患者的生活质量。本病例强调了在成人周期性发热疾病的鉴别诊断中考虑 PFAPA 综合征的重要性,以及秋水仙碱作为预防药物的作用。需要更大的研究来更好地了解病因发病机制并开发其他有效的治疗方法。