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In our cohort, the use of steroids was effective in 25/31 patients (81 %) and also in 4 out of 5 patients that developed a recurrence of fever when we abated the steroids. The administration of steroids allowed a rapid reduction of the fever in less than 10 h after the onset of febrile episodes in all patients except in the 3 youngest children (mean age 2.5 years) that showed a persistent fever after 15 h. In a subgroup of patients with a fever recurrence after steroids withdrawal, we observed that the median time to recurrence was 28 h (range 5-120 h), and that the patients experienced a subsequent recurrence in most cases, despite a prolonged tapering of the steroid dosage [5].
There are a few challenging aspects in the daily clinical practice of pediatricians (and emergency physicians) dealing with children with fever. First is the diagnostic workup, and second, when the diagnosis of PFAPA syndrome is made, is the follow-up and management of the patients. PFAPA syndrome is the most common cause of recurrent fever in children, but its diagnosis is often challenging because of a relatively broad clinical spectrum and the difficulty in making a differential diagnosis with common fever and disease such as UTI. Moreover, in severe cases of a PFAPA flare, the association of abdominal pain is very suggestive to PFAPA syndrome, but the differential diagnosis with other abdominal pain conditions is also challenging for pediatricians. Our study is one of the largest cohorts reported with PFAPA syndrome and the results should encourage pediatricians to consider this diagnosis in the workup of recurrent fever in children.
To summarize, in our cohort we observed that PFAPA syndrome was more prevalent in girls and that the affected children were younger than those reported in the literature. No case of myocarditis or pancreatitis was observed. As in other studies, low dose steroid treatment was effective to rapidly resolve fever episodes in our cohort of PFAPA patients. Age was negatively associated with the occurrence of recurrence of fever after steroids withdrawal. Further investigations are needed to better understand the pathophysiology of PFAPA syndrome and to identify different biomarkers to help in the diagnosis of the disease.
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