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CASE REPORT

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SPONTANEOUS PERFORATION OF CONGENITALBLADDER DIVERTICULUM

ROBERT J. STEIN, DEREK J. MATOKA, PAUL H. NOH, and STEVEN G. DOCIMO

ABSTRACTtraumatic rupture of the bladder in a healthy child is extremely rare and is usually not considered duringvaluation of abdominal pain or sepsis. However, a delay in the diagnosis of bladder perforation can resultn morbid and sometimes catastrophic outcomes. We report a delayed diagnosis of spontaneous rupture of

congenital bladder diverticulum in a previously healthy child. This case demonstrates that spontaneousladder rupture may be a rare cause of abdominal complaints and sepsis when the etiology at first seemsnclear. UROLOGY 66: 881.e5–881.e6, 2005. © 2005 Elsevier Inc.

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he true incidence of congenital bladder diver-ticula is not known, but Blane and colleagues1

eported that 0.7% of 5084 children undergoingadiographic workup of the genitourinary tract werehought to have primary, congenital diverticula. Al-hough typically asymptomatic, patients with diver-icula may present with recurrent urinary tract in-ections or urinary retention, or the diverticula

ay be associated with ureteral reflux or obstruc-ion. We present a case of spontaneous rupture of areviously undiagnosed congenital bladder diver-iculum in an otherwise healthy boy.

CASE REPORT

The patient was a 9-month-old boy born at termith no past medical history who presented to the

mergency room with multiple bouts of blood-inged emesis. He had signs of peritonitis on thenitial evaluation, and the plain films were sugges-ive of bowel obstruction. Exploration revealed co-ious clear ascites that was believed to be the likelyesult of a perforated appendix; however, on fur-her examination, the bowel and appendix wereormal. The patient remained intubated postoper-tively for several days, and during this period, heequired pressors for hemodynamic support.

rom the Department of Urology, University of Pittsburgh Med-cal Center, Pittsburgh, Pennsylvania

Address for correspondence: Robert J. Stein, M.D., Departmentf Urology, University of Pittsburgh Medical Center, Liliane S.aufmann Building, 3471 Fifth Avenue, Suite 700, Pittsburgh, PA5213. E-mail: steinrj@upmc.eduSubmitted: December 28, 2004, accepted (with revisions):

tpril 14, 2005

2005 ELSEVIER INC.LL RIGHTS RESERVED

Cultures of both the ascites and urine grew Klebsiellaxytoca. A computed tomography scan with intra-enous contrast showed a right-sided perivesicalollection with multiple septations. Delayed im-ges were consistent with a right-sided bladderiverticulum and extraperitoneal urine extravasa-ion into the perivesical collection. Cystographyonfirmed that the urine extravasation was fromhe diverticulum, and the patient underwent pelvicxploration (Fig. 1).Initial cystoscopy demonstrated no evidence of

osterior urethral valves or stricture, and the neckf the diverticulum was noted to be several centi-eters cranial to the right ureteral orifice. On ex-

loration, severe induration of the right perivesicalissues was present. The bladder was opened, andhe diverticulum was carefully inverted, and a largeerforation of the end of the diverticulum wasoted (Fig. 2). The diverticulum was excised, andhe bladder muscle and mucosa were reapproxi-ated in two layers. The patient subsequently im-

roved clinically and was extubated. The Foley cath-ter was removed on postoperative day 6, and theatient continued to do well on routine follow-up.

COMMENT

Cases of spontaneous rupture of acquired blad-er diverticula have been reported more often thann congenital diverticula. In the pediatric popula-ion, perforation has occurred in association withonnective tissue disorders such as Ehlers-Danlosyndrome and Menkes’ kinky-hair disease, a rareongenital defect of copper metabolism associatedith Ehlers-Danlos syndrome type IX.2–4 In addi-

ion, Redman and colleagues5 reported spontane-

0090-4295/05/$30.00doi:10.1016/j.urology.2005.04.004 881.e5

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us rupture of a bladder diverticulum in a childith trisomy 9, a chromosomal abnormality asso-

iated with multiple genitourinary anomalies.Spontaneous rupture of a bladder diverticulum

n a healthy child, such as we report, is much lessommon. We were able to identify only one otherescription of a 1-year-old boy with a spontaneousupture of one of his bilateral periureteral divertic-la.6 Similar to our patient, he presented with ancute abdomen and sepsis and was noted to have

IGURE 1. Cystogram demonstrating (A) bladder diver-iculum and (B) urinary extravasation from diverticulum.

scites on abdominal exploration. 1

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A timely preoperative diagnosis and prompturgical management can temper the morbidityssociated with spontaneous bladder rupture.7,8

n our case, a delay in management occurred owingo the unusual presentation of an acute abdomenithout trauma in an otherwise healthy child. Weostulated that infection due to urinary stasis inhe diverticulum led to increasing edema and ob-truction of the diverticular neck with increasingntraluminal pressures and eventual perforation.his case underscores the importance of consider-

ng a possible urologic source in infants found toave ascites of unknown etiology.

REFERENCES1. Blane CE, Zerin JM, and Bloom DA: Bladder diverticula

n children. Radiology 190: 695–697, 1994.2. Jorion JL, and Michel M: Spontaneous rupture of bladder

iverticula in a girl with Ehlers-Danlos syndrome. J Pediatrurg 34: 483–484, 1999.

3. Trulock TS, Finnerty DP, and Woodard JR: Neonatalladder rupture: case report and review of literature. J Urol33: 271–273, 1985.4. Oshio T, Hino M, Kirino C, et al: Urologic abnormalities

n Menkes’ kinky hair disease: report of three cases. J Pediatrurg 32: 782–784, 1997.

5. Redman JF, Seibert JJ, and Arnold WL: Urinary ascites inhildren owing to extravasation of urine from the bladder.Urol 122: 409–411, 1979.6. Sullivan MJ, Lackner LH, and Banowsky LH: Intraperi-

oneal extravasation of urine: BUN-serum creatinine dispro-ortion. JAMA 221: 491–492, 1972.7. Zia-ul-miraj M: Congenital bladder diverticulum: a rare

ause of bladder outlet obstruction in children. J Urol 162:112–2113, 1999.8. Itoh N, and Kounami T: Spontaneous rupture of a blad-

er diverticulum: ultrasonographic diagnosis. J Urol 152:

IGURE 2. Diverticulum exposed after inverting it withorceps to an intravesical position. Two forceps andolding stitch held open the large perforation.

206–1207, 1994.

UROLOGY 66 (4), 2005