Two-step diagnostic method in diagnosing periodic fever with aphtha stomatitis, pharyngitis and lymphadenitis, a practice from China

Cong Liang, Zhen He, Youzhong An, Jun-zhu Chen, Weiying Zeng, Hui Jin, Tao Sui,Li Yu,Ke Zhang

Research Square (Research Square)(2023)

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摘要
Abstract Objective Periodic fever with aphtha stomatitis, pharyngitis and lymphadenitis (PFAPA) is an inflammatory disease characterized by recurrent fever, which mainly affects children and has a high rate of missed diagnosis in China. This study is set to summarize the clinical features of PFAPA children and to develop a feasible diagnostic method. Methods Base on our previous experience, a two-step method consisting of preliminary assessment with disease recognition and subsequent verification was used when diagnosing PFAPA in children. The clinical data of the PFAPA children diagnosed from June 17, 2020 to November 3, 2022 were collected and summarized, and the two-step diagnostic method was validated according to the CARRA-PFAPA diagnostic criteria. Results A total of 260 children with periodic fever were included in this study, 8 of them did not meet the criteria of suspected PFAPA. Of the rest, 29 lost follow-up and 9 had no more episode during follow-up. One child was excluded from PFAPA by prednisone trial. The children diagnosed with PFAPA had recurrent fever beginning at a median age of 30 months, but diagnosis was made at 13 months later. The cycle duration was 30 days and the attack lasted 4 days. Exudative tonsillitis, oral ulcer, cervical adenitis and abdominal pain were presented in 93.6%, 51.4%, 43.9% and 33.1%, respectively. Leukocytosis and elevated C-reactive protein were presented in 88.6% and 91.2% of the children during fever episode, and all returned to normal when reexamined in the interval. Cytokines were detected in 36 cases during episodes, interleukin-6 increased in all cases, interferon-γ increased in most cases, but all returned to normal during asymptomatic interval. The height and weight of the children were lower than those of normal. 39.5% of the children had a positive family history. If criteria “normal growth and development” removed, only 15 of the 213 children diagnosed with PFAPA did not fulfill the CARRA-PFAPA criteria for less than 6 episodes at the time diagnosing, but all up to the standards in follow-up. Conclusion PFAPA should be considered for children with recurrent fever, especially those with tonsil exudation. The two-step diagnostic method provides the indications for corticosteroid use, and thus may be appropriate in clinical practice.
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periodic fever,aphtha stomatitis,pharyngitis,lymphadenitis,two-step
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