Eosinophilic Cystitis Presented as a Manifestation of Hypereosinophilic Syndrome: A Case Report and Review of the Literature

Abstract

Background: Hypereosinophilic syndrome (HES) is a group of disorders marked by the sustained overproduction of eosinophils, in which eosinophilic infiltration and inflammatory substance release cause damage to multiple organs. Eosinophilic cystitis (EC) is an inflammatory disorder caused by eosinophilic infiltration of the bladder wall. Although EC is often associated with eosinophilia, it has been rarely reported as a manifestation of HES. We report a case of EC as a primary manifestation of HES. The patient was a 27-year-old male with a history of complete intracardiac repair of tetralogy of Fallot who presented with an acute onset of dysuria accompanied by eosinophilia (7.5 × 10³/µl, 60% of white blood cells). Ultrasonography and MRI of the bladder showed a bladder mass, a biopsy of which revealed eosinophilic infiltration and degranulation. Methods: We performed a literature search in PubMed from 2001 to 2012 to find patients with EC who may have had HES. Results: There were 4 patients with HES who had EC including the present case. Of 14 patients reported as EC in whom the eosinophil count was described, 5 had eosinophils of ≥1,500/µl. None of the 5 patients had secondary causes for eosinophilia. Of the 9 patients with definite or probable HES, 7 patients (78%) were male and 5 patients (56%) showed a concomitant eosinophilic gastrointestinal disorder. Conclusion: HES may not be uncommon as the cause of EC. Thorough evaluation and close monitoring are warranted in EC patients with elevated eosinophils.

Recurrent feline gastrointestinal eosinophilic sclerosing fibroplasia and presumptive eosinophilic cystitis in a domestic short-haired cat: a case report

“…Reports of eosinophilic cystitis in dogs and humans are very rare (Fuentealba and Illanes 2000;Evason and Carr 2007;Ozaki et al 2008;Gelberg 2010;Koyima et al 2013;Jiang et al 2014) and to the best of our knowledge the condition has yet to be described in cats. In this case a biopsy of the urinary bladder wall would be necessary to confirm the presumptive diagnosis of eosinophilic cystitis, since eosinophils in FNAB could also represent peripheral eosinophilia and/or eosinophil infiltration in the bladder wall as part of another inflammatory process.…”

“…Eosinophilic cystitis is a rare manifestation of urinary bladder inflammation with an unascertained aetiology that has only been reported in dogs and humans (Fuentealba and Illanes 2000;Evason anddoi: 10.17221/134/2016-VETMED Carr 2007;Ozaki et al 2008;Gelberg 2010;Koyima et al 2013;Jiang et al 2014).…”

Eosinophilic cystitis: three cases, and a review over 10 years

“…In two patients, recurrence of symptoms occurred while tapering prednisone 7 23. Two patients had relapse of symptoms after discontinuing prednisone,24 27 and steroid treatment was reinitiated. Symptoms recurred in one patient after ended prednisone treatment while tapering montelucast,18 and in another while tapering antihistamine 28.…”

“…The most common causative and associative factors to EC are listed in table 3. Less common possible causes of EC were mononucleosis,20 recurrent UTI,20 32 chronic granulomatous disease,23 BK polyoma virus infection,13 blood cord transfusion,14 bladder stones,2 13 previous pelvic trauma,19 nephrogenic adenoma,29 parasitic infection8 32 and hypereosinophilic syndrome 7 12 27…”

“…Finally, a parasitic infection was detected in one patient who was then treated accordingly 8. The most common combination of medications used, were antihistamines with steroids 2 10 11 16 23 27 31 33. Twelve patients (21%) underwent surgery as the only treatment 2 13 30 35 36…”