Acta chir belg, 2007, 107, 429-431

In Utero Urinary Bladder Rupture : a Case Report

D. Bataille, E. Van Hoorde, M. Cassart, Th. Roumeguere, C. Donner, P. Lingier Department of Perinatalogy, Erasme hospital, Brussels, Belgium.

Key words. Bladder rupture ; foetal ascites ; megacystis ; urachus ; urethral valves ; newborn. Abstract. We report a case of foetal urinary bladder rupture due to posterior urethral valves. A megacystis was diagnosed in a male foetus during routine second trimester ultrasound examination. The diagnosis of bladder rupture was made as, one week later, the bladder became undetectable with the appearance of ascites. During the follow-up, no oligohydramnios developed and intercurrent ascites resolved spontaneously. There are three described mechanisms releasing bladder hyperpressure : bladder diverticles, unilateral vesicoureteral reflux and bladder rupture. In this case, another mechanism might be involved : a patent urachus. The urethral valves were resected and no other surgical treatment was needed. The renal function remained normal. No long-term vesical follow-up of this pathology is available in the literature.

Introduction Posterior urethral valves are one of the most common causes of lower urinary tract obstruction in newborn males. In the most severe cases, obstruction leads to urinary retention, hydronephrosis and renal insufficiency. Extravasation of urine from the upper urinary tract can occur on rare occasions. In very few cases, rupture of the bladder has been described (3-10). Wherever it happens, the leakage is a protective event as it relieves the urinary pressure and prevents further kidney damage. Case report We report the case of a male foetus in which a megacystis (Fig. 1A) was detected during the 23rd week ultrasound examination, evoking the diagnosis of urethral obstruction. One week later, the bladder became undetectable on ultrasound while a large amount of ascites appeared (Fig. 1B). No associated anomalies were detected on careful ultrasound morphologic investigation ; in particular there was no oligohydramnios and the renal parenchyma was normal, with no ureteral dilatation or hydronephrosis. On MRI and ultrasound examination, during the 26th week of gestation, ascites were still present but the bladder was enlarged with a widened posterior urethra (Fig. 1C). The diagnosis of urethral obstruction, due to posterior or anterior valves, or congenital stenosis, or polyps, or mega-urethra, was evoked before birth. A conservative approach was chosen, as the situation seemed well tolerated by the foetus. Weekly follow-up sonograms showed a slow decrease of the ascites until their complete resorption at the 39th week of gestational age. A bladder with thickened walls, without

recurrence of urinary retention, was observed as early as the 27th week. A left uretero-hydronephrosis developed during the 39th week. The baby was delivered at 40 weeks and 2 days of gestational age, with normal extra-uterine life adaptation. Ultrasound examination showed a thickened bladder wall, a bilateral hydronephrosis and no ascites. Within the first 12 hours of life, the absence of micturition indicated the placement of a urinary tube to empty the bladder. On the first day of life, a retrograde cystography showed a bilateral grade 5 vesico-ureteral reflux, a decompensated bladder, a dilatation of the posterior urethra above a posterior urethral valve and a patent urachus as far as the umbilicus (Figs. 2A & 2B). The urethral valve was endoscopically resected on the 9th day of life. There was no more urinary retention. The renal function remained normal and the iterative sonograms showed a slow resolution of the hydronephrosis and no recurrence of the ascites. Five weeks after surgery, a retrograde cystography showed the absence of vesico-ureteral reflux, and a thickened and trabeculated bladder wall. Four months after surgery, an isotopic nephrogram showed normal values. At one year of life, the baby has normal micturitions. Discussion Posterior urethral valves (PUV) are a quite frequent condition (1/5000 to 12500 births) and the most frequent cause of lower urinary tract obstruction in male children (1). Their clinical presentations vary from mild dysuria to urinary retention with urinary tract hyperpression, bladder wall thickening and vesico-ureteral reflux. In the most severe cases, this can lead to renal dysplasia, which may cause renal insufficiency. The renal

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Fig. 1 a : A sagittal ultrasound scan on the foetal abdomen at 23 weeks of gestation showed a huge bladder (arrow). Note the absence of ascites. b : An axial ultrasound scan on the upper foetal abdomen at 24 weeks of gestation showed the presence of ascites (arrow) suggesting the bladder rupture. c : A sagittal T2-weighted MR scan on the foetal body at 26 weeks of gestation, confirming the ascites, the huge bladder and the posterior urethral enlargement (arrow).

Fig. 2 A neo-natal voiding cystography, frontal view (a) and lateral view (b). a : The frontal view showed a thickened and irregular bladder wall and a bilateral high-grade reflux. b : The lateral view better demonstrated the enlarged posterior urethra above the valve (arrowhead), and the permeable urachus (arrows).

parenchymal lesions are also due to the urinary reflux in the renal cavities and parenchyma. Besides the urological and nephrologic manifestations of the PUV, the associated oligohydramnios can lead to pulmonary hypoplasia, facial dysmorphy and deformity of the extremities, in some cases, leading to foetal death (1). The treatment is the endoscopic resection of the valves (1). There are three described protective events for the renal parenchyma, gathered under the denomination of pop-off mechanisms : urine extravasation and, therefore, the appearance of a urinoma or urinary ascites, unilateral vesicoureteral reflux and large bladder diverticulas (2). Extravasation usually originates from the calyx (3). In this particular case, there was no hydronephrosis before the rupture. This shows that the uretero-vesical antireflux valve remained functional until then and decompensated only afterwards. The complete resorption of the ascites without recurrence of megacystis or development of an oligohydramnios proves that the bladder wall was completely healed before birth and that intermittent voiding was possible. The urine was most probably eliminated through the urethra but there may be another excretory path : the patent urachus may have created a natural cystostomia. This is only a hypothesis as this entity was not identified before birth and we have no proof that the urine could be eliminated through it. There are only seven described cases of pre-natally diagnosed urinary bladder rupture : five cases due to PUV (4-8),

one case due to anterior urethral valves (9) and one in a neurogenic bladder due to spina bifida (10). The gestational age at diagnosis was between the 26th and the 34th week. They all presented with an oligohydramnios. In two, a discontinuity was observed in the bladder wall with ultrasounds (4, 5), in two others the leak was observed with colour Doppler ultrasonography (8, 9). Among these seven cases, 4 had in utero procedures : 4 ascites aspirations (5, 6, 8, 9), one had an additional in utero peritoneal-amniotic shunt (8). 3 of them had a post-natal cystorraphy (4, 7, 10). One patient died at 41 hours of life from respiratory insufficiency due to pulmonary hypoplasia (6), the others are doing fine, particularly in respect of having normal renal functions (4, 5, 7, 9), except for the baby with the neurogenic bladder who presented a mild renal insufficiency (10). In our case, diagnosis was made earlier than in the literature (24 weeks of gestation). The baby was born at term without any major problem thanks to the absence of oligohydramnios. No surgical treatment of the bladder was required. At one year, the vesical function was satisfactory. Conclusion Ante-natal urinary bladder rupture is a very rare condition that can be well tolerated by the foetus. When the condition is due to a urethral obstruction, this rupture has a positive effect on the renal function by preventing excessive upper urinary tract damage. As long as no oligohydramnios and no hydronephrosis develop, a

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conservative approach can be adopted. The long-term vesical function in this pathology is not yet known. References
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7. BETTELHEIM D., PUMBERGER W., DEUTINGER J., BERNASCHEK G. Pre-natal diagnosis of foetal urinary ascites. Ultrasound Obstet Gynecol, 2000, 16 : 473-475. 8. BLESSED W. B., SEPULVEDA W., ROMERO R., BERRY S. M., KING M. E., COTTON D. B. Pre-natal diagnosis of spontaneous rupture of the foetal bladder with colour Doppler ultrasonography. Am J Obstet Gynecol, 1993, 169 : 1629-1631. 9. MERROT T., CHAUMOTRE K., SHOJAI R., DERCOLE C., ALESSANDRINI P. Foetal bladder rupture due to anterior urethral valves. Urology, 2003, 61 : 1259i-1259ii. 10. PINETTE M. G., BLACKSTONE J., WAX J. R., CARTIN A. Enlarged foetal bladder : differential diagnosis and outcome. J Clin Ultrasound, 2003, 31 : 328-334.

Dr. P. Lingier, M.D. Hpital universitaire Erasme Route de Lennik, 808 B-1070 Bruxelles, Belgium Tel. : +32.2/555.65.76 Fax : +32.2/555.83.33 E-mail : plingier@ulb.ac.be