Journal of Pediatric Surgery Case Reports 79 (2022) 102235

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Journal of Pediatric Surgery Case Reports

journal homepage: www.elsevier.com/locate/epsc

Anorectal malformation diverted with transverse colostomy

associated with recurrent urinary tract infections
Tasnia Islam a, Fariha Nusrat a, Mohammad Kabirul Islam b, Mahboob Hossain a, *
a
Microbiology Program, Department of Mathematics and Natural Sciences, BRAC University, Dhaka, Bangladesh
b
Department of Paediatric Surgery, Square Hospitals Ltd., Dhaka, Bangladesh

A R T I C L E I N F O A B S T R A C T

Keywords: Anorectal malformation (ARM) is the absence of a functioning anus with an abnormal attachment
Anorectal malformation of the anus or rectum with urinary or female genital systems in a newborn child. It is considered a
Recto urethral fistula birth defect and is associated with comorbidities that may affect the baby from the early stages of
Urinary tract infection life. We report a case of an eight-month-old boy with recurring urinary tract infection, fever, and
Posterior saggital AnoRectoPlasty leakage of blood-filled fluid from his previously constructed transverse colostomy stoma due to
the absence of anus and was diagnosed to have ARM with rectourethral fistula. Posterior Saggital
AnoRectoPlasty was performed and the condition of the baby was stabilized with no further
report of urinary tract infection. For ARM with rectourethral fistula, the preoperative care of
colostomy and urgent corrective surgery is obligatory to curtail the effects of ARM and associated
anomalies on the affected neonate.

1. Introduction
Anorectal malformation (ARM) is a congenital defect that affects many neonates globally. Anomalies occur in the distal anus and
rectal, as well as the urinary and genital tracts [1,2]. Around 1 in every 5000 live births are affected by ARM that including both sexes,
with a slightly greater prevalence in males [1–3]. The prognosis for each ARM case depends on the severity of the case, which may
range from minor defects to life-threatening complications that are often associated with other anomalies of the lower gastrointestinal
and urogenital systems, added with or without gynaecological issues, orthopaedic, spinal, cardiac and neurological complications [1,
3–5]. Such associated anomalies have a 60% incidence rate [6]. However, more than half of these associated anomalies fall under the
urogenital category [7]. The most common form of ARM that affects male babies is rectourethral fistula, which arises due to an
abnormal connection of the rectum to the urethra, (either bulbar or prostatic form) and according to several studies as reviewed by
Murphy and colleagues, 28–36% males with ARM are affected by this form of abnormality [8,9]. It is vital to identify and correct any
ARM during the first few months of life to normalize such congenital defects and therefore, surgery is necessary. Our report describes a
medical case of an eight-month-old male baby with a recto-bulbous urethral fistula who has been suffering from recurrent urinary tract
infection (UTI) and underwent corrective surgery.

2. Case presentation
An eight-month-old male baby was admitted with complaints of high-grade fever with cold for three days. He had a history of the
increased urinary frequency with straining and passage of blood per urethra during each passage of urine. Some blood mixed fluid also

* Corresponding author. Microbiology Program, Department of Mathematics and Natural Sciences, BRAC University, 66-Mohakhali, Dhaka, 1212, Bangladesh.
E-mail address: mmhossain@bracu.ac.bd (M. Hossain).

https://doi.org/10.1016/j.epsc.2022.102235
Received 7 February 2022; Received in revised form 22 February 2022; Accepted 4 March 2022
Available online 5 March 2022
2213-5766/© 2022 Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license
(http://creativecommons.org/licenses/by-nc-nd/4.0/).
T. Islam et al. Journal of Pediatric Surgery Case Reports 79 (2022) 102235

came out through the previously made transverse colostomy stoma, done for ARM with recto urethral fistula, 6 months back. The
mother also noticed the passage of some stool, mixed with urine per urethra a few days back. Distal loopogram X-ray (Fig. 1) was
performed for routine evaluation of ARM which showed the passage of contrast materials in the urethra along with a mixture of stool,
confirming recto urethral fistula associated with ARM. During admission, his pulse was 110/min, body temperature was 100 ◦ F and
oxygen saturation was 94% in room air. There was an absence of oedema or dehydration. The Lymph nodes were impalpable.
After admission to the inpatient department (IPD), the patient was administered intravenous Paracetamol for pyrexia, and
nebulization was given for the spasm in both lungs. Injection Ceftriaxone was also started, suspecting respiratory tract infection
associated urinary tract infection but the general condition of the baby did not improve. Gradually, he developed abdominal distention
with no passage of stool through the colostomy after 24 h. His bowel sound was absent at that time. The fever and chest infection did
not subside. So, a nasogastric (NG) tube was inserted and suction was given every 2 h.
As the condition was not improving, the patient was then shifted to the Paediatric Intensive Care Unit (PICU) four days after
admission. In the meantime, routine examination of urine indicated the presence of albumin, leukocytes, red blood cells, and pus cells
in the urine. An Ultrasonogram of the whole abdomen indicated distal bowel obstruction with dilated loops. Other abdominal organs
were found to be normal except for mild hepatomegaly. A urine culture report revealed the presence of Enterococcus faecalis with a
colony count of 105 cfu/ml. The antibiotic sensitivity report is presented in Table 1. However, no microbial growth was observed in the
aerobic and routine blood culture and sensitivity tests.
In the PICU, the patient was kept in ‘Nothing by mouth’ and new antibiotics like injection Meropenem and injection Vancomycin
were introduced omitting Injection Ceftriaxone. Injection Paracetamol and nebulization were also continued. With this treatment,
gradual improvement of the baby was observed. So, subsequently, the NG tube was removed and breastfeeding was resumed from the
third day in PICU. After 5 days of starting new antibiotics, the Haemogram and C-reactive protein (CRP) levels were found satisfactory.
So, the intravenous antibiotics were continued for 5 days and the baby was discharged with oral linezolid for one more week. During
discharge he was afebrile, well alert, the chest was clear. He was taking food orally and was maintaining adequate urine output with a
proper functioning Colostomy stoma.
Two months later, the baby was readmitted for surgery. After stabilizing the general condition of the baby, a new anus was
constructed by Posterior Saggital Anorectoplasty (PSARP). The congenital recto urethral fistula was also ligated during the surgery. No
further complications were observed during the post-operative period. Oral feeding was started from the third post-operative day. The
newly formed anus was kept patent by regular anal dilatation by metallic anal dilators. The colostomy was also closed after 3 weeks of

Fig. 1. Distal loopogram X-ray lateral view showing a recto urethral fistula connected to the bulbar urethra.

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T. Islam et al. Journal of Pediatric Surgery Case Reports 79 (2022) 102235

Table 1
Antibiotic sensitivity test of patient urine culture (routine and aerobic) and sensitivity report.

Antibiotics Sensitivity

Penicillin S
Amoxicillin S
Gentamicin (12 μg) S
Ciprofloxacin I
Nitrofurantoin S
Linezolid S
Vancomycin S

Key: R-resistant, S- sensitive, I- intermediate.

doing PSARP. The baby is now doing well and gaining bodyweight satisfactorily. He has been advised to have regular anal dilatation.
He has been passing stool through the new anus and did not have any episode of UTI after the surgery.

3. Discussion
Our case describes an eight-month-old patient with transverse colostomy done for Anorectal malformation (ARM), although high
ARM with rectourethral fistula was identified after admission for corrective surgery. The anorectal malformation is specified as a
congenital deformity that can range from the presence or absence of meconium to imperforated anus, presence of meconium in urine,
or perineal reflex. Based on its severity and the particular relationship between the rectum and sphincter complex, ARM can be
classified as low or high. While low-type anomaly can present with an enterourinary tract fistula [10], a high-type ARM usually in­
dicates a broad fistulous connection between the rectum and urinary tract [11,12]. As in this case, loopogram revealed passage of
contrast materials in the urethra and mixture of stool, which eventually confirmed the presence of rectourethral fistula, and it was then
identified as high ARM.
Determining the type of ARM is essential to sketch out the treatment plan. Since rectourethral fistula associates high-type ARM, a
diverting colostomy is performed first regardless of the patient’s age [13]. It helps to divert the feces away from the urethra and
prevents unwanted infection. The second phase of the treatment is performed two to three months after colostomy when the patient
has had a chance to grow. Conventionally, a three-stage anorectoplasty is performed; however, in recent years, minimal invasive
laparoscopic correction of ARM associated with recto-vesicle, rectourethral or rectobulbar fistula has been attempted during neonatal
period [14,15].
Regardless of the treatment method, colostomy care is essential as it comes with a high incidence of complications. There are many
reported complications associated with colostomy in infants; which include but are not limited to colostomy prolapse, urinary tract
infection, intestinal obstruction, stomal ulceration, and bleeding [16–18]. Such complications could be highly correlated to our case.
Before the patient could be prepared for the second phase of the treatment, he was experiencing recurrent UTI and did not respond well
to intravenous Ceftriaxone. He also developed abdominal distention as there was no fecal discharge. A urine culture report revealed the
presence of Enterococcus faecalis. Just as in adults, Escherichia coli is identified as the main bacterial etiology for neonatal and infant UTI
[19,20]. However, infant UTI with Gram-positive organisms like E. faecalis is rare [20] which is an unusual aspect of our case and it
also influences the choice of antibiotic. Since the patient did not have any prior history of UTI during the neonatal period, it is assumed
that such recurring febrile UTI could have occurred as one of the complications of the colostomy. While renal anomalies are very
common with anorectal malformation [21], the presence of rectourethral or rectovesical fistula puts the patient at an increased risk of
UTI. Unattended and prolonged UTI may progress to severe complications such as sepsis and therefore instant treatment is necessary.
Several complications, one of which is sepsis, are a major drawback of late diagnosis and therapy of ARM [22]. Electrolyte imbalance
may also occur due to colonic absorption of urine, which has a significant impact on the outcome of the patient [23]. Besides, patients
with high-ARM have been reported with a high incidence (86%) of UTI [21]. In this case, reinfection with UTI was probably due to the
choice of ‘looped transverse colostomy’. A malfunctioning loop colostomy can cause distal movement of stool and faecal impaction in
the distal stoma which increases the incidences of UTI, although this is still a topic of debate [24,25]. On the other hand, a transverse
stoma is nearly impossible to clean which imposes a health hazard as a hygienic stoma is important to prevent infections such as UTI
[21,24]. Other complications associated with transverse colostomy include stoma prolapses [23]. However, a ‘divided colostomy’ in
the descending or sigmoid colon is more a preferred choice for ARM [26]. Since the faecal stream is completely separated in this type of
colostomy, it prevents spillover into the urinary tract and the risk of UTI is reduced. The two stomas between of proximal and distal end
of the colon are kept well separated, thus reducing the overall rate of infections [26]. The procedure has added advantages of reduced
stoma prolapse and better access to distal colostogram for identification of rectourethral fistula [23].
ARM is an inborn defect and hence it should be diagnosed at birth. However, low-income countries, such as Bangladesh still
struggle due to lack of awareness and limitations in the healthcare system which is why congenital anomalies remain a significant
reason for infant mortality. Many deliveries are performed by non-professionals especially on the rural side of the country. As a result,
many birth defects easily go unnoticed. Some cases have been described of late presentations of ARM are from low-income countries
[27]. In case of anorectal malformations, along with delayed diagnosis, another factor contributes to the life-threatening outcome that
is delaying colostomy and the subsequent treatment procedures. Often parents are reluctant about the surgical procedures considering
the age of the child which is a misconception and is highly responsible for crucial complications. Delay in diagnosis, associated
complications, and the added issue of undernourishment are some major reasons behind increased mortality rates of children suffering

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from ARM in low-income countries [28]. Moreover, there are a few reported cases of ARM-associated complications in Bangladesh.
However, a 2019 study observed many different subtypes of ARM in Bangladesh [29]. It revealed a higher incidence ARM without
fistula and with vestibular fistula compared to other complications. However, very less information is found on UTI-associated high
ARM, one of the threatening conditions in neonates and infants. Besides good management of ARM, this case also emphasizes the
importance of colostomy care at home until any definitive surgery is performed.

4. Conclusion
The lack of awareness and knowledge regarding ARM cases in low-income countries induces delayed diagnosis and treatment.
However, early diagnosis of the ARM type is necessary and the correct surgery should be performed as soon as possible, which may
lessen the severity of complications and suffering of the affected child.

Patient consent
Consent to publish the case report was not obtained. This report does not contain any personal information that could lead to the
identification of the patient.

Funding
This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

Authorship
All authors attest that they meet the current ICMJE criteria for Authorship.

Declaration of competing interest

The authors declare that they have no known competing financial interests or personal relationships that could have appeared to
influence the work reported in this paper.

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