Keywords
circumcision - intraperitoneal - bladder rupture - rare complication
Introduction
Male circumcision is one of the commonest surgical procedures practiced in children
worldwide. According to several studies, almost a third of the world male population
is circumcised.[1]
[2] A complication rate of up to 16% has been reported.[1] Bladder rupture is a very rare and life-threatening complication of such a procedure.
In a review of literature, only three such cases were reported with this being the
fourth.[3]
[4]
[5] We would like to report on a 7-day-old patient that suffered from an intraperitoneal
bladder rupture following neonatal male circumcision.
Case Report
Our patient presented to our emergency department on the seventh day of life with
severe abdominal distension and vomiting. A thorough history revealed that the baby
was circumcised on the second day of life following an uncomplicated cesarian section
delivery. The exact mode of circumcision was unknown to the parents. After this the
baby was found to be fussy and constantly crying and was no longer tolerating his
feeds. He then started to become lethargic and show progressive abdominal distension.
The baby was transferred to a neonatal unit with a provisional diagnosis of neonatal
sepsis. Three days later he was referred to our center to assess and manage his ongoing
abdominal distension.
General examination revealed an elevated temperature of 38°C, pulse of 155, and a
blood pressure of 70/38. The abdomen was diffusely enlarged with prominent dilated
veins, a large protuberant umbilical hernia, and mild erythema. There was no tenderness
on palpation. It was dull all over on percussion with the evidence of a transmitted
thrill. The baby was passing stool normally and there were no bilious aspirates when
an nasogastric tube was placed. Careful examination of the site of circumcision revealed
that the glans penis was ischemic with signs of inflammation at the shaft ([Fig. 1]). Laboratory investigations revealed an elevated serum creatinine level of 2.3 mg/dL,
C-reactive protein of 96 mg/L, hyperkalemia (5.7 mMol/L), hyponatremia (122 mMol/L),
and an elevated leucocyte count of 15,000/mm3.
Fig. 1 Clinical picture of the baby at first presentation. Notice the gross distended abdomen,
dilated abdominal veins, mild erythema of the abdominal wall and dry gangrene of the
distal glans of the penis.
The baby was immediately admitted and resuscitation was performed. An abdominal X-ray
failed to show any free air in the abdomen and a concomitant abdominal ultrasound
showed marked free fluid with internal echoes and bilateral moderate hydronephrosis.
A provisional diagnosis of intraperitoneal bladder rupture was made based on this
data and a conservative approach was decided. An abdominal drain was placed in the
operating room under general anesthesia yielding >500ccs of yellowish turbid fluid.
A urinary catheter was placed to drain the bladder; however, catheterization was difficult
due to involvement of the distal part of the penile urethra.
The baby remained under conservation for 2 days after which a retrograde cystogram
was performed through the catheter revealing leakage of contrast from the bladder
near the trigone into the peritoneum ([Fig. 2]). An open exploration was decided upon through a Pfannenstiel incision. Due to the
close proximity of the perforation to the ureters, we decided to approach from within
the bladder to allow us to cannulate the ureters and avoid injuring them during the
repair ([Fig. 3]).
Fig. 2 Ascending cytogram (lateral view) showing an intraperitoneal leakage of contrast
from the dome of the bladder posteriorly close to the trigone.
Fig. 3 Intraoperative view. The bladder is bivalved (opened), the ureters are cannulated
and the perforation is evident.
Two pathological perforations were appreciated near the trigone between the ureteric
orifices. These were debrided and closed in two layers with 4/0 polygalactine suture.
The bladder was then closed over a suprapubic self-retaining catheter. An abdominal
toilet was done, a drain was placed, and the incision was closed. A clear line of
demarcation was visible at the time of surgery between the gangrenous and healthy
penile tissue as almost a week had passed since the initial injury. This prompted
us to perform debridement of the dead tissue to avoid sending the patient for a second
general anesthetic.
The baby was transferred to the surgical neonatal intensive care unit where he remained
on broad-spectrum antibiotics for 3 days. Oral feeds were commenced on day 2. On day
5 postoperatively, a contrast study was done through the suprapubic catheter revealing
absence of any leakage. The suprapubic catheter was then clamped. When the baby was
voiding normally the catheter was removed. The baby was discharged on the seventh
postoperative day.
Discussion
Neonatal bladder rupture is a rare problem that has been reported secondary to several
different pathologies. The most common of these is urinary tract obstruction due to
a posterior urethral valve or anterior urethral valve.[6]
[7] It has also been reported on as a possible complication of umbilical catheterization[8] and severe neonatal urinary tract infection (UTI).[9] Only three cases of bladder rupture following circumcision have been reported in
literature till today. Two of these were due to circumcision with a Plastibell device
where the urinary tract had become obstructed due to migration and trapping of the
inner perpetual skin beneath it.[3]
[4] The third case was in a 2-year-old child and had occurred when deep hemostasis sutures
were taken following circumcision to control bleeding and the surgeon had inadvertently
damaged the distal subcoronal urethral causing urinary tract obstruction.[5]
In our patient, the situation was a little different. The circumcision was performed
by a traditional practitioner employing mostly a bone cutter. Our hypothesis is that
the distal end of the glans had become caught in the device causing a crush injury
to it and the most distal part of the urethra and this wasn't managed correctly unfortunately.
This caused an acute urinary retention that was missed on initial examination and
ultimately led to pressure build up and bladder rupture.
Our initial management was conservative through catheterization of the bladder per
urethra and drainage of the urinary ascites after proper patient resuscitation. The
decision was made for exploration due to a worsening general condition and the retrograde
cytogram that demonstrated an intraperitoneal leak. Both modalities of treatment have
been proposed to manage intraperitoneal bladder rupture; however, the inclination
is usually toward a formal surgical repair especially when there is a deterioration
of the patient's general condition. Our management was in accordance with published
guidelines from the consensus on genitourinary trauma which underlined that neonatal
bladder trauma usually requires surgical management.[10] In addition, published case reports on similar conditions ultimately resorted to
surgical intervention to as a course of management.[2]
[3]
[4]
[5]
[6]
[7]
[8]
[9]
Most authors report that an open approach but some have advocated for a laparoscopic
approach to repair bladder ruptures.[6] In any case, the repair is performed from outside the bladder. In our patient, we
opted to bivalve the bladder and perform the repair from inside due to the close proximity
of the perforation to the ureters and fear of the repair leading to ureteric obstruction.
This approach allowed us to identify and cannulate both ureters in addition to performing
a mucosal and seromuscular repair of the bladder.
Long-term monitoring of patients where suturing in done near the trigone is vital
as such patients are at a higher risk of long-term bladder dysfunction.[11] We were unable to do so in this patient unfortunately as he was lost to follow-up.
Neonatal circumcision remains a controversial subject to this day. Many studies have
been designed to assess the advantages and disadvantages of this practice. Recently
the role of neonatal male circumcision in the reduction in the risk of UTIs has been
emphasized.[12] Furthermore, it has been shown that neonatal circumcision if performed under the
appropriate controlled conditions has a much lower risk of short-term complications
than if performed at an older age. The risk of additional surgery is in fact almost
1% if circumcision was initially performed after the neonatal period.[13]
However, this statement isn't supported by many. The relationship between circumcision
and the development of UTIs according to several authors is only established in those
with a predisposition to the condition such as those boys with PUJ (pelviureteric
junction) obstruction, high-grade vesicoureteral reflux, and hydronephrosis.[14] This is why they feel that routine nontherapeutic practice of circumcision should
be restricted to those subgroups of patients. To add to this point, it was found that
the risk rate for the development of meatal stenosis and other urethral stricture
diseases was higher in religious and cultural ethnicities that practiced nontherapeutic
circumcision. This greatly adds to the health service burden.[15]
It is our message that although circumcision is performed routinely in many cultures,
it can still lead to catastrophic and life-threatening problems adding a considerable
cost to the already taxed health services especially in low- and middle-income countries.
It cannot be stressed enough that this should only be performed by a trained professional
under safe, sterile circumstances and where a proper follow-up can be provided to
detect and manage any complications as early as possible should they occur.
