Hypereosinophilic syndrome (HES) is a rare disorder that is characterized by hypereosinophilia and organ damage, caused by the infiltration of eosinophils. In rare cases, the urinary bladder may also be involved. The current case report presented a 56-year-old male with gross hematuria and hypereosinophilia. The diagnosis of eosinophilic cystitis associated HES was established. Oral prednisone with a slow tapering regimen was administered as the primary treatment for the patient, which achieved partial hematological remission and complete relief of cystitis during a six-month follow-up period. Although eosinophilic cystitis is not commonly the primary manifestation of HES, eosinophilic cystitis should be taken into consideration following the onset of urinary symptoms in patients with HES.
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