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Journal of Pediatric Surgery (2012) 47, E33–E38

Post meningocele repair urinary ascites in a neonate—arare presentation☆

Ritesh Ranjan⁎, Paras R. Kothari, Rahul K. Gupta, Abhaya R. Gupta, Gursev Sandlas,Parag J. Karkera, Krushna K. Kesan

Department of Pediatric Surgery, LTMMC and LTMG Hospital Sion, Mumbai, Maharashtra 400022, India

Received 10 June 2011; revised 30 January 2012; accepted 31 January 2012

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Key words:Meningocele repair;Urinary ascites;Neonate

Abstract A full-term female neonate with a large lumbosacral meningocele developed suddenabdominal distension and urinary retention after meningocele repair. An erect abdominal radiographshowed homogeneously opacified areas with a paucity of intestinal gas. Abdominal ultrasound revealedascites with multiple internal echoes. With a provisional diagnosis of hollow viscus perforation, anemergency laparotomy was performed, which failed to reveal any leak from the urinary tract. However,fluid analysis confirmed the diagnosis of urinary ascites. The patient developed repeated episodes ofurinary retention after catheter removal. She was started on clean intermittent catheterization (CIC) toensure adequate bladder drainage. At 6 months of follow-up, ultrasound of the urinary tract, voidingcystourethrogram, and magnetic resonance imaging of the spine were all within normal limits. The CICwas discontinued, and the patient was observed. At present, she is voiding normally with a good stream.Failure to establish normal micturition after meningocele repair and CIC requirements suggested aneurogenic cause. Bladder rupture, secondary to spinal shock resulting in bladder atonia, could not beruled out. The perforation (leading to urinary ascites) could be owing to bladder atonia and spinal shockrather than detrusor sphincter dyssynergia.© 2012 Elsevier Inc. All rights reserved.

Ascites in the neonatal period is most commonly urinaryin origin and the end result of a leak from any area in theurinary tract. King [1] in 1836 was the first to report a case ofurinary ascites in a fetus owing to rupture of the urinarybladder. Reports of urinary ascites in the neonatal periodowing to bladder rupture have appeared sporadically sincethen. Most of the neonates were boys with obstructivepathologies such as posterior urethral valves as the cause ofurinary ascites. Even more rare are cases of urinary ascites

☆ Conflict of interest: NIL.⁎ Corresponding author. Tel.: +91 9022225821, +91 9833112941.E-mail address: riteshranjan06@gmail.com (R. Ranjan).

022-3468/$ – see front matter © 2012 Elsevier Inc. All rights reserved.oi:10.1016/j.jpedsurg.2012.01.074

owing to nonobstructive urinary tract pathologies, seenmostly in female neonates. Those presenting at birth areassociated with difficult labor, have a worse prognosis, andwarrant prompt action. This report describes our experiencein managing a case of urinary ascites after operative repair ofa lumbosacral meningocele in a female neonate.

1. Case report

A full-term, 2.5-kg, female neonate presented with a largeskin-covered transilluminating lumbosacral meningocele

E34 R. Ranjan et al.

(Fig. 1A and B). The lower limb motor power, bladder, andbowel continence were normal. An anocutaneous reflex waspresent. The infant had good tone, cry, and activity levels.There was no history of cyanosis or features of birthasphyxia. The systemic examination was essentially normal.The abdomen was soft and nontender on palpation. Theinfant passed urine normally. Echocardiogram and ultra-sound of brain, kidneys, ureters, and bladder were withinnormal limits.

Meningocele repair was performed on day 6 of life andwas uneventful. The infant was fine for the first 5 days afterthe procedure, was feeding well, and passed urine and fecesnormally with good wound healing. On sixth postoperativeday, she developed sudden abdominal distension and urinaryretention. The abdomen was dull on percussion, suggestingpresence of free fluid. The bowel sounds were sluggish on

Fig. 1 A, Lumbosacral meningocele. B, Transillumination of themeningocele.

ig. 2 Anteroposterior (A) and lateral (B) views of contrastystogram on the 10th day after exploratory laparotomy showingo leak.

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Fig. 3 Voiding cystourethrography done at 6 months of ageshowing smooth-lined bladderwall, no leak, nor vesicoureteric reflux.

E35Post meningocele repair urinary ascites in a neonate

auscultation. An erect abdominal radiograph showed homo-geneously opacified areas with paucity of intestinal gas.Abdominal ultrasound examination showed the presence of

Fig. 4 Magnetic resonance imaging of the s

ascites with multiple internal echoes. The patient wascatheterized with a Foley catheter, and urine output wasmonitored. The abdominal distension gradually increasedover the next 6 hours, resulting in respiratory distress.

With the provisional diagnosis of hollow viscus perfora-tion, an emergency exploratory laparotomy was performed.Approximately 450 mL of clear ascitic fluid was drained. Allthe abdominal organs were normal. There was no obviousrent or rupture in the kidneys, ureters, and bladder. Theabdomen was closed with a tube drain in situ. The asciticfluid was examined and showed a creatinine level of 4.61mg% compared with the serum creatinine at that time of 3.64mg% (fluid: serum creatinine ratio was 1.27). The bladdercatheter was maintained for 10 days because the gross andmicroscopic evaluations of the ascitic fluid suggested aurinary origin. The serum and urine creatinine valuesdecreased to 0.59 mg% and 1.58 mg%, respectively, onday 8 of catheterization. A contrast cystogram was done onthe 10th postoperative day. The filling phase showedsmooth-lined bladder, closed neck, with no leakage ofcontrast or vesicoureteric reflux (Fig. 2A and B). Voidingand postvoid films were normal with no residual urine.Because the abdominal distension had decreased consider-ably, the abdominal drain and catheter were removed, afterwhich the patient developed repeated episodes of urinaryretention. However, there was no change in lower limb motorpower and sensation postoperatively along with presence of anormal anocutaneous reflex. She was started on cleanintermittent catheterization (CIC) to ensure adequate bladder

pine with no tethering of the spinal cord.

Table 1 Case reports of urinary ascites in literature (in neonates) along with their etiology and postulated hypothesis

No. Author No. of patients reported(parentheses mention thesex of the patient)

Patient's profile Proposed hypothesis for urinaryascites/conclusionof the study

1 Kennelly andRitchie [4]

1 (female) A case of spontaneous perinephricextravasation owing to elevatedintravesical storage pressures in agirl with myelodysplasia. VCUG afterurinoma drainage—right-side grade 4reflux along with perirenal extravasationof contrast

Sustained elevated detrusor leakpoint pressure with neurogenicbladder resulted in pyelosinusbackflow and forniceal rupturethat caused extravasation

2 Alkan et al [5] 1 (male) A 5-day-old boy presented withabdominal distension and impairmentof renal function. Tests revealed urinaryascites and renal insufficiency thatspontaneously resolved aftertransurethral urinary drainage.

It was a case of vesicoureteralreflux with Hutch diverticula.This rare complication should beconsidered in neonates with highintrapelviureteric and intrarenalpressure because of high-gradevesicoureteral reflux withparaureteral diverticula.

3 Redman et al [6] 1 (female) A female neonate with myelodysplasia,hydrocephalus and ascites

Spontaneous rupture of hollowviscus. All previously reportedcases of neurovesical dysfunctionand ascites in neonates havebeen women.

4 Howatt [7] 1 (female) A full-term female infant with a lumbosacralmeningomyelocele First report of urinaryascites as a complication of myelodysplasiain a female neonate

Spina bifida has not been asfrequently described as a cause ofurinary ascites in view of thegross obstructive changes theyfrequently present with inneonates.

5 Cywes et al [8] 19 19 neonates with urinary ascites of whom14 had posterior urethral valves

Obstructive pathologies are themain cause of urinary ascitesin neonates.

6 Brereton et al [9] 1 (female) A female neonate with thoracolumbarmeningomyelocele and urinary ascitesdiagnosed 8 days after birth

Result of vigorous manualpressure to effect vesical emptying

7 Mann et al [10] 1 (female) A female infant with ruptureof the urinary bladder

Neurogenic cause. The patientwas found to have a congenitalneuroblastoma with completeblock of the spinal canal in thelumbar region.

8 King [1] 1 A fetus with urinary ascites that followedrupture of bladder.

–

9 McDonald andMurphy [11]

2 (1 maleand 1 female)

2 neonates with urinary ascites. Confirmedwith ascitic fluid analysis and MCUdemonstrating leak from the posteriorwall of bladder.Both the cases were managed conservatively.

–

10 Vasdev et al [12] 4 4 neonates with spontaneous urinarybladder rupture and secondary urinary ascites.Three were managed conservatively, and 1needed surgical repair.

Ischemia played an important rolein the pathogenesis of neonatalbladder rupture.

11 Morrell et al [2] 2 2 neonates with otherwise normalgenitourinary system having urinary ascites.Both the infants described had traumaticrupture of the fundus of urinary bladder.

Definite episodes of hypoxia andhypotension in the first and astormy obstetric history leading tohypoxia in the second suggesteda definite role of ischemia in thegenesis of spontaneous bladderperforation and urinary ascites.Biochemical analysis of ascitic

E36 R. Ranjan et al.

Table 1 (continued)

No. Author No. of patients reported(parentheses mention thesex of the patient)

Patient's profile Proposed hypothesis for urinaryascites/conclusionof the study

fluid is an important modality todiagnose urinary ascites because,in urinary ascites only, the ureaand creatinine concentrationsexceed those of plasma.Conservative management is thetreatment of choice and thebladder should be catheterized toarrest distension and allowhealing. Paracentesis should bedone to control distension andcollect fluid for analysis but onceurinary drainage is adequatelyestablished it can be safelydiscontinued. Spontaneous ruptureof the bladder carries a goodprognosis provided there are noimmediate complications andassociated abnormalities.

12 Murphy et al [13] 1 A case of neonatal urinary asciteswithout demonstrable obstruction inthe urinary tract.

–

E37Post meningocele repair urinary ascites in a neonate

drainage. There was no evidence of renal compromise at 6months of follow-up, and she had achieved all the growthand developmental milestones expected for her age. Wecould not perform urodynamic study in this patient becausethis facility is not available for infants in our country.However, at 6-month follow-up, we repeated a voidingcystourethrogram (VCUG) that was essentially normal (Fig.3). Ultrasound of the kidneys, ureters, and bladder was alsonormal. Magnetic resonance imaging of the spine revealedno evidence of tethering of the spinal cord (Fig. 4). In view ofthese normal investigations, we discontinued CIC and keptthe patient under observation. She has been voiding normallywith a good stream since then. There is no evidence ofbladder wall thickening or significant postvoid residual urinein the subsequent ultrasound reports.

2. Discussion

A review of literature was undertaken that showed thatour patient indeed had urinary ascites, proven conclusivelyby the ascitic fluid analysis report. We believe thatnonvisualization of a urinary leak intraoperatively could beowing to the small size of the leak or owing to the fact thatthe leak occurred from a location that was not easilyvisualized. Although the clinical scenario strongly pointed tourinary ascites as the cause of abdominal distension, the

ultrasound report did not. Because of the infant's rapidlydeteriorating condition and respiratory distress, we decidedto proceed with an urgent laparotomy. Voiding cystoure-thrography now would not have improved the condition ofthe patient, and moreover, the internal echoes in theultrasound report were suggestive of hollow viscus perfora-tion rather than urinary ascites. The intraoperative findingsand fluid analysis confirmed that this was a case of urinaryascites and not of intestinal perforation. We performed acontrast cystogram on the 10th postoperative day that was,with no leak, observed from the bladder. Because she wasunable to void completely at the time, CIC was started.

Very few reports have been published describing anassociation between myelodysplasia and urinary ascites. Thepresence of urine in the ascitic fluid is suspected when thecreatinine content is greater than that of plasma. An asciticfluid creatinine/urine creatinine ratio greater than 1 (N1.0) ishighly suggestive of urinary ascites [2]. Voiding cystourethro-graphy and intravenous urography sometimes reveal the natureand site of obstruction, condition of the upper tracts, and thesite of leak. Intravenous urography was not performed in thispatient because the serum creatinine was high (3.64 mg%).

Weller and Miller [3] have classified infants with urinaryascites into 3 categories: the largest group of patients arethose with posterior urethral valves; children with complexcaudal anomalies, most commonly anorectal anomalies andurethral atresia; and children with miscellaneous lesions suchas ureteroceles, ureteral atresia, myelomeningocele with

E38 R. Ranjan et al.

neurogenic bladder, and extrinsic lesions such as presacralneuroblastoma. Various case reports of urinary ascites alongwith their etiology and postulated hypothesis are discussed inbrief in Table 1.

Our patient developed urinary ascites after meningocelerepair without any demonstrable urinary tract obstruction.The gross and biochemical evaluations of ascitic fluid werehighly suggestive of urinary ascites. Urgent decompressionof the abdominal distension and urinary drainage remains themainstay of management and proved curative in our patient.The perforation (leading to urinary ascites) might be theresult of bladder atonia owing to spinal shock rather thandetrusor sphincter dyssynergia.

The clinical course and findings could not conclusivelyprove the cause of urinary ascites in our patient. Anyhypothesis, such as neural damage during surgery leading tospinal shock or congenitally present neuropathic elementleading to dyssynergia or urethral obstruction, is onlyspeculative and warrants further investigations.

References

[1] King TW. Case of disease in the fetus: impervious urethra;accumulated urinary secretion; dilated ureter; bursting of the distended

bladder, peritonitis and ascites of the fourth month of fetal life. Guy'sHosp Rep 1837;2:508.

[2] Morrell P, Coulthard MG, Hey EN. Neonatal urinary ascites. Arch DisChild 1985;60(7):676-8.

[3] Weller MH, Miller K. Unusual aspects of urinary ascites. Radiology1973;109:665.

[4] Kennelly MJ, Ritchey ML. Perinephric urinoma secondary toneurogenic bladder in myelodysplasia. J Urol 1995;153:458-60.

[5] Alkan M, Ozcelik Z, Keskin E, et al. Fetal urinary ascites in a neonatewithout detectable obstructive uropathy or neurogenic bladderetiology. J Pediatr Urol 2009;5(2):151-3.

[6] Redman JF, Rader LE, O'Donnel PD. Urinary ascites in femaleneonate with myelodysplasia. Urol Int 1987;29(6):616-7.

[7] Howatt JM. Urinary ascites complicating spina bifida. Arch Dis Child1971;46(245):103-105.

[8] Cywes S, Wynne JM, Louw JH. Urinary ascites in the newborn with areport of two cases. J Pediatr Surg 1968;3(3):350-6.

[9] Brereton RJ, Philp N, Bayukpamukcu N. Rupture of the urinarybladder in children: the importance of the double lesion. Br J Urol1980;52:15.

[10] Mann CM, Leape LL, Holder TM. Neonatal urinary ascites: a report of2 cases of unusual etiology and a review of the literature. J Urol1974;111:124.

[11] McDonald J, Murphy AV. Neonatal ascites from spontaneous ruptureof the bladder. Arch Dis Child 1971;46:103.

[12] Vasdev N, Coulthard MG, De la hunt MN, et al. Neonatal urinaryascites secondary to urinary bladder rupture. J Pediatr Urol 2009;5(2):100-4.

[13] Murphy D, Simmons M, Guiney EJ. Neonatal urinary ascites in theabsence of urinary tract obstruction. J Pediatr Surg 1978;13(6):529-31.