Posterior urethral valve associated with a foreign body in posterior urethra: a first of its kind report

Abstract

Posterior urethral valve and foreign body are among the important causes of male urethral obstruction. Although one is congenital and the other is acquired, both entities are rare in children with only a few reported cases. Because of myriad of symptoms associated with both conditions, a conclusive diagnosis requires both physical examination and radiological imaging. We report a first of its kind association of posterior urethral valve with foreign body in the posterior urethra in a 6-year-old male child which was eventually managed by endoscopic intervention.

Background

Posterior urethral valve (PUV) and foreign body are among the important causes of male urethral obstruction. Although one is congenital and the other is acquired, both entities are rare in children with only a few reported cases. PUV is a congenital cause of male urethra obstruction with an incidence of 1:8000 to 1:25 000.¹

Although the aetiology is antenatal, patient may present as a neonate or a child or even as an adult. Foreign bodies in urethra also have a very low propensity of occurrence in children.²

We report a first of its kind association of foreign body in urethra with posterior urethral valve in a 6-year-old male child and eventually managed by endoscopic intervention.

Case presentation

Case report

A 6-year-old male child came to the outpatient department with complaints of recurrent urinary tract infection, nocturnal enuresis and burning micturition since birth. He had recent onset history of dribbling of urine and dysuria. There was no history of haematuria, colicky abdominal pain or fever. His physical examination was unremarkable. His weight and height were both below the fifth percentile of his age, although his IQ was normal. Complete haemogram was normal with normal blood counts and serum creatinine value. Urine routine microscopy revealed few pus cells and culture revealed growth of Escherichia coli.

X-ray kidney, ureter and bladder did not reveal any radio opacity in the pelvic region. Ultrasound abdomen was done and suggested of a distended bladder with thickened walls with bilateral mild hydroureteronephrosis with significant postvoid residual urine. Micturating cystourethrogram (MCU) showed a dilated posterior urethra with distended bladder as shown in figure 1.

Figure 1.

Micturating cystourethrogram (MCU) demonstrating a dilated posterior urethra with no vesicoureteric reflux.

A diagnosis of PUV was made and it was decided to perform operative intervention. On cystoscopy, child was found to have PUV with a surprising finding of foreign body in posterior urethra as shown in figure 2.

Figure 2.

Foreign body cystoscopically seen in posterior urethra following valve fulguration.

It appeared glistening blue plastic material and was radiolucent.

First the PUV was fulgurated. The foreign body was then removed cystoscopically along its longitudinal axis with a Double J stent removal forceps, taking care not to damage the remaining healthy urethra. The plastic material measured around 1.5 cm in length and appeared tip of pen cap as shown in the figure 3.

Figure 3.

Plastic foreign body measuring 1.5 cm in length.

Differential diagnosis

Hinman syndrome.

Outcome and follow-up

On follow-up at 3 months, the child was voiding well with no recurrence of symptoms.

Discussion

A myriad of self-inserted materials have been reported in adults ranging from electrical cables, pencils, ball point pens, pen lids, copper wire, batteries, marbles, plants and vegetables, toys, pieces of latex, gloves, tip of Foley catheter and safety pins.^{2 3} But self-insertion of foreign body is rarely reported in literature in the paediatric age group.^{2 3}

How a foreign body landed up in the posterior urethra of a 6-year-old child is an anomaly that has perplexed the authors with failure to reach a conclusive answer. Multitude of theories exist on self insertion of foreign body in urethra ranging from psychiatric illness, intoxication, sexual gratification and attempts to relive itching by inserting objects in the urethra.⁴ In all possibility, the authors opine that the child could have accidentally inserted the foreign body while playing. Another hypothesis is that the child may have inserted a pen (a complete one) into the urethra trying to relieve his obstructed voiding/dysuria, the pen cap disconnected from the pen and remained in the urethra as a foreign body.

Selected psychoanalytical theories have also been put forth, for example, Kenney’s theory of impulsivity, Wise’s sado-maso-fetishistic theory and Dr Poulet’s manic masturbation hypothesis with the most prevalent motivation being masturbation and autoerotism.³ Whether these theories hold true in a paediatric child is a separate topic to discuss and debate.

A study conducted by Alibadi et al reported reasons for self-instrumentation in 15 patients and found autoeroticism in six and psychiatric causes in two patients. In three patients, however, no definite reason for self-insertion could be described.⁵

On the other hand, accidental and iatrogenic foreign bodies occur much more rarely.⁶

Obtaining a correct history, although paramount, is often difficult from patients of paediatric age group. Leading questions are asked to evoke responses, the testimony of which also needs to be confirmed with the parents/guardians. Patience and high index of suspicion is therefore required.

Patients with both PUV and foreign body can present with a wide array of symptoms—neonates generally present with poor urinary stream, outlet obstruction and urinary tract infection. Delayed presentation in childhood is also common with presentations including recurrent urinary tract infections, nocturnal enuresis and dribbling of urine.^{7 8}

The presenting symptoms of foreign body reflect irritation of the lower genitourinary tract with the most common being dysuria, lower abdominal and urethral pain, microscopic or gross haematuria, acute urinary retention, urethral discharge and fever.^{9 10}

Because symptoms can overlap, it is very hard to discern either anomaly on basis of clinical presentation alone. A radiolucent foreign body in posterior urethra coexisting with posterior urethral valve may be easily missed.

Diagnosis in most cases can be confirmed by physical examination and combination of radiological findings. Foreign bodies distal to urogenital diaphragm can be easily palpated. A pelvic X-ray, an ultrasound and a CT can help define both a radiolucent as well as radiopaque foreign body.^{3 4}

MCU is commonly done to diagnose PUV. A child with ultrasound finding of upper tract dilatation or dilated posterior urethra should undergo MCU to rule out PUV.¹¹

In our case, the foreign body was missed on both MCU and ultrasound. A part of this misdiagnosis could stem from the fact that foreign body in our case was hollow tubular plastic which could have accumulated the contrast material during MCU.

Cystourethroscopy is an important part of urological armamentarium for diagnosing and managing both these conditions. Indeed, endoscopic manipulation and extraction utilising forceps and snares can be both diagnostic and therapeutic for treatment of urethral foreign bodies. Cystoscopy-guided valve fulguration is also the treatment of choice for PUV.^{11 12}

There have been associations of urethral calculus with PUV previously reported in the literature. In the present case, we demonstrated a rare association of PUV with self-inserted foreign body. The authors believe this to be first ever documented case with aforementioned presentation. A thorough search on pub med and Google scholar did not reveal any previous literature on this association.

It is apothegmatic to keep these two similar presenting conditions in mind when evaluating any child presenting with unusual lower urinary tract symptoms. One should think of unusual causes in patients with unusual presentations, especially in paediatric age group.

Learning points.

• Posterior urethral valve and foreign body are among the important causes of male urethral obstruction.

• It is axiomatic to keep these two similar presenting conditions in mind when evaluating any child with lower urinary tract symptoms.

• It is recommended that a thorough evaluation be performed in children with unusual lower urinary tract symptoms. The diagnosis requires a combination of physical examination and radiological imaging. Treatment is cystourethroscopy which acts as both diagnostic and therapeutic.

• The family should exercise vigilance over their children to prevent risk of reinsertion of any foreign body in future.