Eosinophilic cystitis: A rare cause of hematuria

Seema Rohilla; Nitin Jain; Dhara B Dhaulakhandi; Shaveta Jain

doi:10.4103/2148-7731.152116

CASE REPORT

Year : 2015 | Volume : 2 | Issue : 2 | Page : 41-43

Eosinophilic cystitis: A rare cause of hematuria

Seema Rohilla¹, Nitin Jain¹, Dhara B Dhaulakhandi², Shaveta Jain³
¹ Department of Radiodiagnosis and Imaging, Post Graduate Institute of Medical Sciences, Pt. Bhagwat Dayal Sharma University of Health Sciences, Rohtak, Haryana, India
² Department of Biotechnology and Molecular Medicine, Post Graduate Institute of Medical Sciences, Pt. Bhagwat Dayal Sharma University of Health Sciences, Rohtak, Haryana, India
³ Department of Obstetrics and Gynecology, Post Graduate Institute of Medical Sciences, Pt. Bhagwat Dayal Sharma University of Health Sciences, Rohtak, Haryana, India

Date of Web Publication

25-Feb-2015

Correspondence Address:
Dr. Nitin Jain
House no 629, Sector 14, Rohtak - 124 001, Haryana
India

Source of Support: None, Conflict of Interest: None

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DOI: 10.4103/2148-7731.152116

Abstract

Eosinophilic cystitis (EC) is an uncommon inflammatory condition of the bladder which can mimic malignancy both clinically and radiologically. It is characterized by eosinophilic infiltration of the bladder wall. High level of suspicion is needed to arrive at the diagnosis which is important because it is a self-limiting condition and in most cases surgical intervention is not required. We present one such case in a child whose symptoms totally resolved on conservative treatment.

Keywords: CT scan, eosinophilic cystitis, hematuria

How to cite this article:
Rohilla S, Jain N, Dhaulakhandi DB, Jain S. Eosinophilic cystitis: A rare cause of hematuria. Sifa Med J 2015;2:41-3

How to cite this URL:
Rohilla S, Jain N, Dhaulakhandi DB, Jain S. Eosinophilic cystitis: A rare cause of hematuria. Sifa Med J [serial online] 2015 [cited 2023 Mar 23];2:41-3. Available from: https://www.imjsu.org/text.asp?2015/2/2/41/152116

Introduction

Eosinophilic cystitis (EC) was first reported in 1960 by Brown ^[1] and Palubinskas ^[2] independently. It has been attributed to food allergens, ^[3] infectious agents, drugs (salicylazosulfapyridine, tranilast, Coumadin, intravesical thiotepa and mitomycin), ^{[4],[5],[6],[7],[8]} parasites (Toxocara cati, Echinococcus granulosus and Sparganum) ^{[9],[10],[11]} and as hypersensitivity to chromate catgut sutures ^[12] as well as indwelling catheter. ^[13] Association with charcot-leyden crystals, celiac disease, lupus anticoagulant and additional viral and bacterial agents has also been found. ^[14] Our patient had eosinophilia but association with any particular allergen could not be established.

Case Report

A 7-year-old female child presented with suprapubic pain during micturition, frequency, urgency and terminal hematuria for 8 months. She had been repeatedly diagnosed as a case of urinary tract infection and treated with antibiotics by different doctors (three times during this 8 month period) without any relief of symptoms.

Hematological examination showed Hb = 10 g/dl, WBC count of 12000/mm ³ with differential leukocyte count of polymorphs 40%, lymphocytes 28%, mononuclear cells 2% and eosinophils 30% (absolute eosinophil count being 3600/mm ³ ). Patient had significant eosinophilia. Urine sediments showed more than 40 erythrocytes per high power field. No pus cells or eosinophils were detected in urine. Repeated urine cultures were negative. Ultrasound of abdomen showed gross thickening of right lateral wall and right halves of anterior and posterior walls of urinary bladder. Contrast enhanced computed tomography (CT) scan showed more than 1cm thickening of bladder wall in the same area with moderate enhancement [Figure 1]a and b. Bilateral kidneys and ureters were, however, normal. Taking into consideration the presentation, age and laboratory data (marked eosinophilia), radiological diagnosis of eosinophilic cystitis was made.

Figure 1: (a) Axial non-contrast CT scan of the pelvis showing thickening of right lateral wall and right halves of anterior and posterior walls of urinary bladder (b) Axial contrast enhanced CT scan of the pelvis showing moderate enhancement of the thickened wall of urinary bladder

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The patient received 9 weeks of tapered prednisone therapy. The patient showed total symptomatic relief after 9weeks of therapy. Ultrasound picture normalized after 12 weeks. Blood eosinophil count also normalized after 6 weeks. The patient is doing well after 17 months.

Discussion

EC was first recognized as an inflammatory bladder condition by Duke in 1922, ^[3] but was first characterized by Brown and Palubinskas independently in 1960. ^[1],[2] It is characterized by massive eosinophilic infiltration of the bladder wall and is widely believed to be due to allergy to food, drugs etc. IgE may bind to various antigens activating mast cell degranulation and eosinophil attraction. Activated eosinophils may release cytotoxic cationic proteins that could induce tissue damage. ^[15] Peripheral eosinophilia is present in 43% of patients with EC but elevated serum IgE is uncommon. ^[16] Our patient also had eosinophilia. Another possible pathogenic mechanism is dysregulation of cytokines mainly involving interleukin 4 and 5 (IL-4 and IL-5). ^[17] Yoshino and Moriyama treated a patient of EC successfully with suplatast tosilate. ^[18] This drug inhibits the production of IgE from B cells and cytokines from mast cells. ^[19] Low production of IL-5 leads to inhibition of eosinophil differentiation and activation thus preventing further release of cytotoxic cationic proteins and hence tissue damage. ^[20] Thus the two proposed mechanisms of action are actually interlinked.

Cytotoxic cationic proteins thus released cause tissue damage seen histologically as tissue eosinophilia, mucosal edema and hyperemia, and muscle necrosis in acute phase (deep biopsy is needed for proper diagnosis). Chronic phase shows fibrosis, mast cells and plasma cells along with squamous metaplasia and cystitis cystica but tissue eosinophilia is inconspicuous. ^[14] Development of secondary malignancy has, however, never been reported. ^[21] Eosinophiluria is rare because eosinophils are either degraded rapidly or there occurs little mucosal shedding from the urothelium. ^[22]

It is a benign condition which can mostly be managed conservatively, but high level of suspicion is warranted to consider this diagnosis. As it may closely mimick infection and malignancy which are much more common conditions, misdiagnosis and mismanagement is common specially in cases where urine culture is positive. It should be considered in a patient with bladder wall thickening of more than 10mm with preserved mucosal lining on CT (as in our case), with or without peripheral blood eosinophilia. However, deep biopsy is definitive.

Though it is a benign condition, it is important to diagnose and manage it properly as it is associated with numerous complications such as hydroureteronephrosis (due to inflammation obstructing intramural ureter), uremia, eosinophilic ureteritis, eosinophilic cholangitis, retroperitoneal fibrosis, spontaneous bladder perforation with persistent vesical fistula and vesicoureteric reflux. ^[14] Our patient had an uneventful recovery.

In non-responsive cases, electrocoagulation, endoscopic resection of bladder mass, partial cystectomy, radical cystectomy with reconstruction or prostatectomy should be considered. ^[14]

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