© 2 0 0 5 B J U I N T E R N A T I O N A L | 9 5 , 4 11 – 4 1 3 | doi:10.1111/j.1464-410X.2005.05311.x 411 Original Article CYSTITIS GLANDULARIS IN CHILDREN CAPOZZA et al. Cystitis glandularis in children NICOLA CAPOZZA, GIUSEPPE COLLURA, SIMONA NAPPO, MAURO DE DOMINICIS, PAOLA FRANCALANCI* and PAOLO CAIONE Divisions of Paediatric Urology and *Pathology, ‘Bambino Gesù’ Children’s Hospital, Research Institute, Rome, Italy Accepted for publication 20 October 2004 Presented at the 15th ESPU Meeting, Regensburg, Germany, April 21–24, 2004 gross haematuria in the first patient and frequency and urgency in the second. The third patient was asymptomatic and the lesion appeared as a wide thickening of the bladder wall on follow-up ultrasonography for previous surgery. In all patients, a polypoid bladder mass was found at cystoscopy and diagnosed at histology. The endoscopic resection, with long-term antibiotic prophylaxis, was the treatment of choice, with no recurrence at 12–30 months of follow-up. CONCLUSION Cystitis glandularis has been rarely described in children, and is probably related to chronic or recurrent infections or an inflammatory reaction. Its potential premalignant significance is still the subject of debate. KEYWORDS cystitis glandularis, urinary tract infections, paediatric OBJECTIVE To assess the characteristics of cystitis glandularis in children. PATIENTS AND METHODS Three cases of cystitis glandularis in children are described, occurring in boys aged 9–13 years. The presenting symptoms were INTRODUCTION Cystitis glandularis is a proliferative and metaplastic disorder of the bladder mucosa rarely reported in children; even in adults its incidence and clinical significance are not known exactly. The occurrence of cystitis glandularis is closely related to other proliferative changes of the bladder mucosa, e.g. submucosal masses of epithelial cells (‘Brunn’s nests’), epithelial crypts and the subepithelial fluid-filled cysts of cystitis cystica. In cystitis glandularis the transitional epithelium undergoes metaplasia into a columnar type, which may or may not secrete mucus. It is frequently found lining the cysts of cystitis cystica, and many authors therefore term it as ‘cystitis cystica glandularis’ [1–4]. The diagnosis is generally made on histopathological examination. The correlation of cystitis cystica glandularis with the development of bladder tumours, e.g. adenocarcinoma and TCC, is debatable. Only a few cases of cystitis glandularis have been reported in children [5,6] and here we describe three more. PATIENTS AND METHODS Patient no. 1 was born with the exstrophy- epispadias complex; he had primary closure of the exstrophic bladder within the first 24 h of life and genital reconstruction at 20 months old. He had partial continence with spontaneous transurethral voiding every 60–90 min, a voided volume of ª 120 mL, no residual urine and recurrent UTI (mostly Escherichia coli). He presented at 9 years old with gross haematuria and no other symptoms. On ultrasonography a small polypoid bladder lesion (1.5 cm diameter) was found. Cystoscopy confirmed the presence of the lesion, in a diverticulum of the bladder wall, which was completely resected. Histology showed a polypoid lesion with evidence of cystitis cystica and glandularis. Continuous antimicrobial prophylaxis was restored; at 2.5-years of follow-up there was no recurrence. Patient no. 2 was operated at 16 months old to correct an ectopic megaureter in a duplex kidney. At the follow-up, recurrent UTIs from E. coli and Pseudomonas spp were recorded. He presented at 9 years old with irritative bladder symptoms (frequency, urgency) and an irregular bladder wall on ultrasonography. At cystoscopy a single polypoid bladder mass was found and completely resected. After 2 years the follow-up was uneventful and histology showed cystitis glandularis. Patient no. 3 was operated at 1 year old to correct a left obstructive megaureter; the recovery was uneventful. At follow-up there was no recurrent UTI reported by the parents and the dilatation of the upper urinary tract recovered almost completely. At 13 years old the annual ultrasonography showed a wide bladder mass (3 cm diameter) on the right lateral bladder wall and smaller multiple lesions on the left side (Fig. 1). At cystoscopy multiple polypoid lesions were found (Fig. 2a,b) and biopsied. Histology showed cystitis glandularis (Fig. 3). The lesion was completely resected by transurethral endoscopy and at the 1-year follow-up there was no recurrence detected. In all cases the diagnosis was confirmed at histology; specimens were examined during surgery and completely resected endoscopically. Histology showed bladder mucosa with oedematous lamina propria, epithelial hyperplasia and characteristic glandular structures lined with mucus- secreting cubic and columnar cells (Fig. 3). DISCUSSION The cause of cystitis cystica glandularis is debatable; the ‘embryological theory’, currently abandoned, considered that the mucus-secreting cells of cystitis glandularis might arise by inclusion of intestinal mucosa in the bladder from the primitive cloaca or from the residue of the omphalo-mesenteric duct [1,3,4]. The intestinal metaplasia of cystitis glandularis is a result of a chronic