Gross hematuria, irritative lower urinary tract symptoms, and bilateral hydroureteronephrosis: A concerning presentation of an ultimately benign case of concurrent nephrogenic adenoma and eosinophilic cystitis

Abstract

Nephrogenic adenoma and eosinophilic cystitis are well-known, distinct pathologic bladder diagnoses that rarely occur concurrently in the published literature. We present the first adult case, and second reported case, of these two disease processes occurring synchronously. Our patient's initial complaints were the characteristic symptoms of gross hematuria and urinary urgency, which were primarily worrisome for malignancy. She had no prior genitourinary history, whereas these two diagnoses are commonly associated with chronic bladder irritation, instrumentation, and trauma. Our patient did not respond to conservative management with prophylactic antibiotics and antihistamines, however experienced full resolution of her symptoms following resection of the lesions.Keywords: benign neoplasm, eosinophilic cystitis, hydronephrosis, nephrogenic adenoma, urinary bladder neoplasm A 79 year-old female was referred to the urogynecology clinic after complaining to her internist of a one year history of intermittent, new onset urinary urgency and urge incontinence associated with vulvar burning, itching, and pressure. On presentation she also reported new onset gross hematuria within the past month. A one week trial of estrogen cream did not relieve the symptoms.She had no prior medical or surgical genitourinary history and no family history of genitourinary disorders or malignancies. She denied any tobacco or chemical exposures, was married, and had delivered two children vaginally.Her physical exam was unremarkable except for posterior forchette tenderness to palpation. Her urinalysis revealed microscopic hematuria with pyuria and no urinary eosinophils. A urine culture was negative. Her laboratory assessment revealed no abnormalities in her complete blood cell count with differential, serum chemistries, liver or renal function.An in-office cystoscopy revealed an approximately 1cm papillary, bullous bladder lesion near the right ureteral orifice. The tumor was friable with a small amount of bleeding. Catheterized cytology was benign. A CT Urogram revealed moderate bilateral hydroureteronephrosis with distal ureteral and asymmetric bladder wall thickening (Fig. 1). The combination of the patient's symptoms and the radiographic and cystoscopic findings was highly concerning for muscle-invasive bladder cancer.The patient underwent an operative cystoscopy and transurethral resection of the bladder tumor with intra-operative findings of diffusely erythematous urothelium with small papillary masses on the right posterior and lateral bladder walls. Upper tract endoscopy was delayed as there was concern for possible upper tract seeding if the lesions were malignant.Pathology revealed multiple papillary structures lined by cuboidal epithelium consistent with nephrogenic adenoma (NA), as well as abundant eosinophils in clusters within the lamina propria and the deep muscle diagnostic of eosinophilic cystitis (EC) (Fig. 2). The patient was treated with a one month course of daily anti-histamines and prophylactic dose trime...