Duplication Of The Urinary Bladder: A Case Report From Northwest Nigeria

ABSTRACT 

Duplication of the urinary bladder is a rare congenital anomaly. Complete and incomplete forms are known in literature. We present a case of complete duplication of the urinary bladder, with right blind-ended bladder with right mega-ureter, and left draining bladder with normal single left ureter, and single urethra in a three-year-old boy. The patient had a single-phase excision of right-sided, blind-ended urinary bladder, and reconstruction of a refashioned right ureter to the left-sided urinary bladder of a sagittal type DUB, with full recovery and normal urinary function afterwards. 

Key words: Bladder duplication, mega-ureter, congenital anomaly,   

INTRODUCTION

Complete duplication of the urinary bladder is rare congenital anomaly, with few cases (less than 50) reported in the last century [1]. Only few cases have been reported from Nigeria, and this is the first from Kebbi state in northwest, Nigeria. The aetiology of DUB remains obscure, although some hypotheses have been proposed. Two explanations were offered by Abrahamson as follows: (a) Excess narrowing between the vesico-urethral and urogenital segments of the ventral cloaca (b) A supernumerary cloacal septum resulting in indentation of the epithelial lining of the urinary bladder [2].  

CASE PRESENTATION 

History and Presentation

We present a case of a 6-year-old boy, who presented on account of right loin pain and fever of one week duration at Federal Teaching Hospital, Birnin Kebbi. There was history of similar recurrent abdominal pain of three years duration. Pain was located in the right loin, of gradual onset, radiating to the right suprapubic region. There was no known aggravating but it was relieved by pain killers. Pain was usually severe enough to prevent him from moving in bed. There was no vomiting, abdominal distension, or change in bowel habits. There was no jaundice or right upper quadrant pain. There was no dysuria, frequency, straining on micturition or terminal dribbling. He had no history of swimming, haematuria, or passage of stones in the urine. There was no previous pelvic or abdominal surgery. Child experienced early morning facial puffiness and significant weight loss during previous episode about five months ago. Fever was low grade, intermittent, but not associated with chills or rigors. 

The patient had been taking to peripheral hospitals during previous episodes with pain killers and antibiotics usually administered with subsequent improvement. He was not a known sickle cell disease or asthma patient. There was no similar illness in the sibling. He was a product of term pregnancy delivered via spontaneous vaginal delivery, said to have cried immediately after delivery. There was no history of febrile illness neither in the mother during pregnancy, nor in the child during neonatal period. He was the second of four children in a monogamous setting, and he was yet to be enrolled in school then.

Physical Examination

General examination revealed a young boy/male toddler, who was conscious, and was not in any distress. He was febrile (temperature was 38.1^oC). He was not pale, jaundiced, cyanosed, or dehydrated. He had no significant peripheral lymph node enlargement, and there was no pedal oedema.     

Cardiovascular system revealed a pulse rate was 104 bpm, jugular venous pressure was not raised and his cardiac apex beat was at the 6^th left intercostal space, lateral to mid-clavicular line. Heart sounds were the first and second only. 

Respiratory rate was 20 cpm, and he had no sign of respiratory distress. Chest was symmetry, tactile vocal fremitus was normal and equal bilaterally. Percussion notes were resonant bilaterally. Breath sounds were vesicular. 

Abdomen was full, and moved with respiratory. There was a mass felt in the right iliac fossa region, extending to the suprapubic region. It was soft, fluctuant, compressible but immobile. The examining hand could not get below it. It measured 18cm by 10cm. Bowel sound was present and normoactive.

Urogenital system showed was normal external genitalia, uncircumcised phallus and urethral meatus. The right side of the scrotal sac was empty. 

Laboratory investigations conducted were as follows: Electrolytes, Urea, and Creatinine were all within normal range, HB genotype was “AA”. Complete blood count was 8.24 X 10⁶/ul, RBC 3.87 X 10⁶/ul, Platelets 306 X 10⁶/ul, Packed cell volume was 36.6%, HB 12.2g/dl, MCV 94.6fl, MCH 31.6pg, MCHC 33.3g/dl. Differentials: Granulocytes 57.3%, Lymphocytes 39.3%, and Mid cells 3.4%. Electrolytes, Urea, Creatinine: K 3.1mmol/l, Na 142 mmol/l, Cl 103mmol/l, Ur 1.8mmol/l, Cr 60mmol/l.

Intravenous Urography showed severe right hydro-uretero-nephrosis, mild left hydroureter, right vesico-ureteric reflux, and normally locate kidneys. Micturating Cystourethrogram revealed severe right sided hydronephrosis causing a mass effect on the urinary bladder. Chest radiography (X-ray) showed normal lung fields, and no enlarged cardiac silhouette.

An assessment of Double urinary bladder was made. 

Management  

Child’s parents were counseled on the nature and the condition and the need for surgical intervention was duly explained. Patient was admitted, a pint of blood was grouped and crossmatched, and he was prepared for surgery. Patient’s nutrition and fluid input were optimized, and he was prepared for surgery. Surgical Procedure involved gaining access to the peritoneal cavity via a midline incision. Operative findings include double urinary bladder, right hydronephrotic kidney, dilated and tortuous right ureter that emptied into a blind ended urinary bladder. There was normally positioned left kidney and normal ureter emptying into a normal urinary bladder, which had a connecting urethra. Access was gained to the peritoneal cavity via a midline incision. The dilated tortuous ureter was mobilized, and excised with the blind-ended bladder. Dilated ureter was tailored/re-fashioned and reimplanted into the normal bladder. The post-operative period progressed with improvement in symptoms with subsided suprapubic pain. Urinary flow was maintained and Electrolyte/Urea/Creatinine done was within normal range throughout. 

DISCUSSION

Duplication of the urinary bladder (DUB) can occur in the coronal or sagittal plane. The coronal plane type results when duplication of two urinary bladders occurs with one lying in front of the second, and a fibromuscular septum running obliquely along posterior-superior to antero-inferior plane, separates the two urinary bladders. A urethra each is connected to each urinary bladder in the coronal type. The sagittal type of DUB, the two urinary bladders lie side by side, being separated by a muscular wall. Each urinary bladder has its own ureter from its ipsilateral kidney, and also has its own urethra, each positioned side by side [3]. In this case, which is a form/variant of the sagittal type, the right urinary bladder was blind-ended, with no urethra, though, it had a right sided ureter from its ipsilateral right kidney (with hydronephrosis present). The left urinary bladder was connected to its normal appearing ureter from a normal appearing left kidney, and a single (only) draining urethra. This morphology makes this case further rare. There was no other associated anomaly in this case, and a similar case was reported almost two decades ago by Oguzkurt in 2006 [4].   

Other congenital anomalies that are known to be associated with DUB include certain forms of duplication of external genitalia; duplication of vagina and urethra; duplication of lower gastro-intestinal tract (GIT); and fistulas between urethra, vagina, and rectum [5]. Physical examination done in this case revealed none of the aforementioned anomalies. Thus, this index case appeared to be an isolated case of double urinary bladder. Skeletal anomalies associated with DUB include diastasis of the symphysis pubis, duplication of lower lumbar vertebral column, meningococele, and spina bifida [6]. However, none of these skeletal anomalies was found in this case. The report of only few cases in the literature, has made it difficult to have a consensus in the methods of treatment. Various surgical treatment modalities have been offered with consideration for the associated anomalies and clinical situation [7]. The index patient was surgically managed by excision of the right blind-ended bladder, separation of the right ureter from the excised right bladder, re-fashioning of the right mega-ureter, and re-attachment via a nipple-valve into the functional urinary bladder.       

CONCLUSION

Double urinary bladder is a rare congenital urological condition. Few cases have been reported worldwide. A high index of suspicion and relevant radiological investigations aided the diagnosis. The patient had a surgical repair with return of urinary flow and subsequent normal renal function.  

Ethical Approval and Consent to Participate

The authors hereby certify that appropriate consent was ob­tained with the patient consent form. Patient’s father gave con­sent for the patient’s clinical information to be reported in a jour­nal. He understood that the patient’s name and other identifying details would not be published. 

Conflict of Interest: The authors declare no conflict of interest.

Funding: There are no funding received for this study.  

REFERENCES

1. Berrocal T, Novak S, Arjonilla A, Gutierrez J, Prieto C, Urrutia MJ. (1999) Complete duplication of bladder and urethra in the coronal plane in a girl: Case report and review of the literature.  Pediatr Radiol. 1999;29:171-3.  

2. Abrahamson J. Double bladder and related anomalies: clinical and embryological aspects and a case report. Br J Urol. 1961;33:195-8. 

3. Hoffman AD, Leroy AJ. Uroradiology: Procedures and anatomy. In: Kelalis PP, King LR, Belman AB, editors. Clinical pediatric urology. 3rd ed. Philadelphia. PA: Saunders; 1992. pp. 66-116.   

4. Oguzkurt P, Ozalevli SS, Alkan M, Kayaselcuk F, Hicsonmez A. Unusual case of bladder duplication: complete duplication in coronal plane with single urethra and no associated anomalies. Urology. 2006;68(5):1121.e1-1121.e11213.   

5. Cifti AO, Senocak ME, Buyukpamukcu N, Hicsonmez A.  Complete duplication of the bladder and urethra: A case report and review of literature. J Pediatr Surg. 1995;30:1605-6. 

6. Ravitch MM. Hindgut duplication- Doubling of colon and genital urinary tracts.  Ann Surg. 1953;137:588-601.  

7. Mirshemirani AR, Sadeghyian N, Mohajerzadeh L, Molayee H, Ghaffari P.  Diphallus: Report on six cases and review of literature.  Iran J Pediatr. 2010;20:353-7.