Vesical manifestations of chronicgranulomatous disease in children: Its relation to eosinophilic cystitis

doi:10.1016/0090-4295(91)80112-K

Urology

Volume 37, Issue 5, May 1991, Pages 463-466

https://doi.org/10.1016/0090-4295(91)80112-K Get rights and content

Abstract

Two children with chronic granulomatous disease involving the bladder are reportedTheir clinical course was characterized by lower urinary tract symptoms, decreased vesical capacity unilateral ureterovesical junction obstruction, and an intense eosinophilic infiltrate on biopsy specimens in each. Chronic granulomatous disease and eosinophilic cystitis appear to have remarkably similar clinical manifestations. A review of urinary tract involvement in chronic granulomatous disease and its comparison with eosinophilic cystitis is presented.

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Eosinophilic cystitis is a rarely seen condition in the pediatric population with indistinct symptomatology and non-standardized treatment protocols. We review our experience of treating patients with this condition.
We retrospectively reviewed the records of four patients from a single institution who have been diagnosed and treated for eosinophilic cystitis. In addition, the literature was reviewed for cases of pediatric eosinophilic cystitis. Our patients were added and compared to this cohort.
Our patients included 3 females and 1 male who range in age from 5 days to 18 years (5 days, 1 month, 7 years, 18 years). Both of the infants presented with a suprapubic mass and bilateral hydroureteronephrosis. The two older patients both had dysuria while the 18 yo also complained of fatigue, flank pain, and hematuria. Only 2 of the 4 patients were found to have significant peripheral eosinophilia and only one patient had eosinophiluria. All of the patients were diagnosed via cystourethroscopy with biopsy. Treatment in each case consisted of a combination of steroids, antihistamines, and antibiotics.
The presentation of eosinophilic cystitis is varied and diagnosis requires a high index of suspicion. Cystourethroscopy with biopsy is essential to establish the diagnosis as there is no typical appearance of the lesions or presenting signs/symptoms. Most cases of eosinophilic cystitis are responsive to medical therapy although in some cases recurrence may be noted.
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Reported genitourinary involvement in patients with CGD includes bladder granulomata, ureteral obstruction, and UTI [1]. In addition, there have been isolated reports of the occurrence of pseudotumors and tumors of the bladder, eosinophilic cystitis, end-stage renal failure, immune complex and chronic glomerulonephritis, prostatic abscess in an adult patient, xanthrogranulomatous pyelonephritis, and renal amyloidosis in patients with CGD [9,10-19]. A recent series in Europe revealed a 23% occurrence of kidney or urinary tract involvement with infection being the most common manifestation.
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In the case of drug sensitivities, specific agents include clometacin, coumadin, cyclophosphamide, mitomycin C, penicillin, thiotepa, and tranilast.83–88 Associations with other disorders known to cause increased peripheral blood eosinophilia are described, as is an association with X-linked chronic granulomatous disease.89–92 Allergic diatheses are reportedly more common in women and children; older men may be affected after bladder injury, urologic procedure, or in the setting of prostate disease.93
Benign diseases of the bladder often present diagnostic challenges to practicing pathologists due to their diverse nature and ability to mimic a variety of epithelial or mesenchymal neoplasms. Categories of benign bladder disease include infectious cystitis, noninfectious cystitis, reactive proliferative processes, and benign processes that secondarily involve the bladder. An understanding of the key clinical and morphologic features of these lesions and the useful ancillary techniques specific for these entities is critical to the correct diagnosis of these lesions. This article reviews the key features of these benign bladder diseases and highlights methods to distinguish these lesions from other benign and malignant processes involving the bladder.
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Benign bladder tumors are rare in children. A number of descriptive terms have previously been used to describe inflammatory tumors with myofibroblastic proliferation or eosinophilic infiltration. We present our experience with these tumors and review the literature.
We retrospectively reviewed the records of all children presenting with a benign focal bladder mass during the last 5 years, including 2 girls and 3 boys 2 to 12 years old (mean age 7).
Presenting symptomatology included irritative voiding symptoms, suprapubic pain and hematuria. All bladder masses were diagnosed by ultrasonography and all patients underwent transurethral resection. Three children who had an inflammatory bladder tumor with myofibroblastic features required open tumor excision with preservation of the bladder. In the 2 remaining children urine culture was positive preoperatively and the diagnosis was an inflammatory bladder tumor with eosinophilic infiltration. Transurethral resection of the mass was performed without partial cystectomy.
Inflammatory myofibroblastic and eosinophilic tumors represent forms of focal cystitis with a tumefactive component. Differentiation from the malignant bladder neoplasms of childhood has important consequences for therapy. Management of these benign but aggressive inflammatory lesions involves local control by endoscopic or open surgery with bladder preservation. We encourage refined use of the terminology for inflammatory myofibroblastic and eosinophilic tumors.
Eosinophilic granulomatous cystitis in children
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We report 3 cases of eosinophilic granulomatous cystitis in children. All presented with hematuria, urinary urgency, and dysuria. Treatment with oral corticosteroids led to rapid and complete recovery in 2 of the 3 cases. In the third case recovery was prolonged without the use of steroids. Because of the presumed immunologic nature of this disease, previous authors have recommended the use of corticosteroids in its management. A review of the literature suggests that the disease is probably of immunologic nature and that corticosteroids shorten symptomatic recovery from this disease.

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Vesical manifestations of chronicgranulomatous disease in children: Its relation to eosinophilic cystitis

Abstract

Clin Perinatol

Pediatr Clin North Am

Urology

J Urol

J Urol

J Pediatr

J Pediatr

J Pediatr

Urology

J Urol

J Urol