Incomplete duplication of urinary bladder in a male child misdiagnosed as mesenteric cyst

Aditya Pratap Singh; Ramesh Tanger; Arun Kumar Gupta; Vinay Mathur

doi:10.4103/2320-3846.194004

CASE REPORT

Year : 2016 | Volume : 4 | Issue : 3 | Page : 129-131

Incomplete duplication of urinary bladder in a male child misdiagnosed as mesenteric cyst

Aditya Pratap Singh, Ramesh Tanger, Arun Kumar Gupta, Vinay Mathur
Department of Pediatric Surgery, SMS Medical College, Jaipur, Rajasthan, India

Date of Web Publication

14-Nov-2016

Correspondence Address:
Aditya Pratap Singh
Near the Mali Hostel, Main Bali Road, Falna, Pali - 306 116, Rajasthan
India

Source of Support: None, Conflict of Interest: None

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DOI: 10.4103/2320-3846.194004

Abstract

Duplication of the lower urinary tract is a very rare congenital anomaly which is diagnosed either at birth or during early childhood. These rare malformations are most of the times accompanied by other concomitant anomalies and are therefore diagnosed immediately after birth. In some even rarer cases, there are no concomitant anomalies and symptoms thus leading to a diagnosis later in childhood. We present a case of incomplete bladder duplication in a male child with no other associated anomalies which misdiagnosed as mesenteric cyst and review the literature pertaining to this rare anomaly.

Keywords: Bladder, cyst, duplication, incomplete, mesenteric

How to cite this article:
Singh AP, Tanger R, Gupta AK, Mathur V. Incomplete duplication of urinary bladder in a male child misdiagnosed as mesenteric cyst. Saudi Surg J 2016;4:129-31

How to cite this URL:
Singh AP, Tanger R, Gupta AK, Mathur V. Incomplete duplication of urinary bladder in a male child misdiagnosed as mesenteric cyst. Saudi Surg J [serial online] 2016 [cited 2022 Aug 16];4:129-31. Available from: https://www.saudisurgj.org/text.asp?2016/4/3/129/194004

Introduction

Incomplete bladder duplication is an extremely rare condition. Since Cattirri reported the first case in 1670 ^[1] only 7 further cases have been reported in the literature. ^[1],[2],[3] Incomplete bladder duplication is often associated with other genitourinary anomalies ^[2],[3] and can even be associated with bladder exstrophy. ^[4] We present a case of incomplete bladder duplication in a male child with no other associated anomalies.

Case Report

A 2-year-old boy presented at the out-patient clinic with constipation over the last 6 months. There were no urinary complaints. Physical examination was uneventful. A single urethral meatus was observed. Routine blood investigations were in normal limits including thyroid function test. Ultrasound, showed 26 mm × 8 mm anechoic cystic lesion with wall showing gut signature lying anterior to urinary bladder likely noncommunicating duplication cyst or mesenteric cyst. The kidneys appeared of normal size and morphology. Contrast enhanced computerized tomography (CECT) abdomen, [Figure 1]a and b showed large peripheral enhancing well-defined hypodense lesion of size approximately 83 mm × 39 mm × 92 mm noted anterosuperior to urinary bladder causing displacement and compression of the adjacent bowel loops suggestive of mesenteric cyst. We repeated ultrasonography (USG) abdomen after catheterization the patient, but finding was mesenteric cyst. On surgical exploration, there were two cystic structure [Figure 1]c and d present with bilateral abdominal testis. Anterior was large than posterior one. We opened both of cystic swellings. There were two ureteric opening in the anterior swelling, after putting a feeding tube in both of swellings; feeding tube came out from the normal urethral opening. It was become confirmed that it is not a bladder diverticulum. Hence, a plane was created between two swellings and small posterior one excised with bilateral orchidopexy was done. The content of the cystic swelling was urine. There was single urethra. An intraoperative biopsy was taken. Biopsy revealed wall of cystic lesion shows a mucosal lining which is partially squamous hyperplastic transitional type of epithelium and the wall showed muscle propria, suggestive of duplication of bladder. The patient showed normal urinary continence postoperatively.

Figure 1: (a and b) Contrast enhanced computerised tomography images with coronal and sagittal plane. (c and d) Intra operative images shows incomplete bladder duplication. Black Arrow duplication and green arrow bladder

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Discussion

Urinary bladder anomalies are generally diagnosed in infancy or childhood during evaluation of cases with urinary tract infections. ^[5] However, our case was presented with constipation.

In 1961, Abrahamson ^[1] classified bladder duplication as complete or incomplete. With complete duplication, two bladders are present with separated walls of mucosal and muscular layers, and each one empties through its own urethra. The final diagnosis is made by simultaneous retrograde urethrocystograms. ^[6] In our case, both USG abdomen and CECT abdomen suggested mesenteric cyst and there were no urinary complaints, so we did not perform micturating cystourethrogram. In incomplete bladder duplication, two bladders communicate with each other and drain into a common urethra. Duplication of the bladder may occur in a sagittal or coronal plane given the axis of the septum. The sagittal variant appears to be predominant compared to the coronal one, with a ratio of 2.5:1. In our case, it was incomplete coronal bladder duplication which is very rare. According to published literature, the male to female ratio of this anomaly is described to be equal. ^{[1],[6],[7],[8],[9]} Females are more likely to present with the sagittal type of duplication and in males, there is no predominance.

The etiology of bladder duplication, despite the hypotheses proposed by Abrahamson, ^[1] remains obscure. More recently Voigt and Wentzel ^[9] raised the question of whether the etiology can be explained by the existence of a human homolog of the mouse mutant disorganization gene first described by Hummel in 1959. ^[10]

Complete bladder duplication is, in the majority of cases, associated with other anomalies. 40 cases of complete bladder duplication were reviewed by Kossow and Morales in 1973. ^[11] They found associated duplication of the lower gastrointestinal tract in 42%, external genitalia in 90% and spina bifida, meningocele or myelomeningocele in 15% of cases. Similar findings, except a lower percentage of associated duplication of the external genitalia (30%), were reported by Berrocal et al. ^[12] Associated urogenital or nonurogenital anomalies seem to depend on the axis of the septum. Nonurogenital anomalies seem to be predominantly present in complete bladder duplication with a sagittal septum, while urogenital anomalies are more common in complete duplication with a coronal septum. ^[5],[7],[12] After a literature review, only six cases of complete bladder duplication without any associated anomalies were reported. ^[8]

Depending on the position of the accessory bladder in relation to the normal bladder, bladder duplication is also classified as sagittal or coronal. Bladder duplication in the sagittal plane is more common with the two bladders side by side, each receiving urine from one ureter. ^[12] Duplication in the coronal plane is less often. The accessory bladder usually lies anterior and superior of the normal bladder. ^[12] In our case, the bladder duplication was coronal which is very rare and present posteriorly. In our knowledge, it is the first case to present posterior to the normal bladder with hugely dilated normal bladder.

To the best of our knowledge, the previously reported nine cases of incomplete bladder duplication, five presented with other anomalies and three were anomaly-free. In our patient, the 10 ^th reported case, no any additional anomalies were present either. The histology of the excised left structure from our patient showed the typical findings of the bladder wall. In particular, the existence of bladder smooth muscles could be proven by positive specific anti-a-smooth muscle actin immunohistochemistry findings. Furthermore, primary urothelial and smooth muscle cell cultures could be established in vitro with the excised tissue, thus also proving the existence of bladder duplication.

The treatment of bladder duplication cannot be standardized, due to the great variations in presentation. We agree with statements in the literature, that treatment should aim at optimization of bladder function, addressing urinary continence and minimizing the risk of infections. ^{[1],[2],[8],[9]} In our case, the presentation was constipation which is a very rare presentation.

Conclusion

In patients with a lower abdominal cystic mass, complete bladder duplication must be included in the differential diagnosis of pelvic cystic masses. In every case of lower abdominal cystic mass, it is mandatory to perform micturating cystourethrogram.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References

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