成人发病型 PFAPA 综合征:用秋水仙碱成功治疗。

IF 1 Q3 MEDICINE, GENERAL & INTERNAL American Journal of Case Reports Pub Date : 2024-08-29 DOI:10.12659/AJCR.943658
Amulya Balagani, Farina Tariq, Muhammad Zaheer
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引用次数: 0

摘要

背景:周期性发热、口腔炎、咽炎和颈部腺炎(PFAPA)综合征是一种自身炎症性发热综合征,主要见于 5 岁以下儿童,病因不明。大多数病例在 10 岁前即可痊愈,成人病例很少见。PFAPA 的特点是反复发热,伴有咽炎、口腔炎和颈腺炎,但在初次评估时并非所有临床特征都存在。由于没有特异性的生物标记物,因此只能通过临床诊断。治疗方法包括泼尼松、秋水仙碱、白细胞介素-1 受体阻滞剂和扁桃体切除术。我们报告了一例对秋水仙碱有反应的成人型 PFAPA 综合征病例。病例报告 一位 22 岁的女性因反复发热伴咽喉痛和颈部淋巴结肿大疼痛到风湿病诊所就诊。在两次发作之间,她没有任何症状。对感染原因和自身炎症/自身免疫性疾病的检查没有发现异常。由于她的表现不寻常,医生考虑了各种鉴别诊断。在排除了所有病因后,根据她的症状确诊为 PFAPA,并开始使用类固醇,她对类固醇的反应非常强烈,症状也得到了缓解。随后,她开始口服秋水仙碱,这大大降低了病情发作的频率。结论 意识到成人 PFAPA 综合征至关重要。及时诊断可大大改善患者的生活质量。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。为了更好地了解发病机制并开发其他有效的治疗方法,还需要进行更大规模的研究。
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Adult-Onset PFAPA Syndrome: Successful Management with Colchicine.

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.

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来源期刊
American Journal of Case Reports
American Journal of Case Reports Medicine-Medicine (all)
CiteScore
1.80
自引率
0.00%
发文量
599
期刊介绍: American Journal of Case Reports is an international, peer-reviewed scientific journal that publishes single and series case reports in all medical fields. American Journal of Case Reports is issued on a continuous basis as a primary electronic journal. Print copies of a single article or a set of articles can be ordered on demand.
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