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Journal of Pediatric Surgery Case Reports
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Enterocutaneous fi stula due to neglected strangulated hernia
Riana Pauline Tamba
a,∗, Dorothy Rachmadia
a, Bertha Kawilarang
baDivision of Pediatric Surgery, Department of Surgery, Faculty of Medicine, Universitas Indonesia, Cipto Mangunkusumo, National General Hospital, Jakarta, Indonesia
bFaculty of Medicine, Universitas Pelita Harapan, Indonesia
1. Introduction
Inguinal hernia is a frequent surgical condition encountered in pe- diatric age group. It may get strangulated at times which require prompt intervention before it develops further into complications such as enterocutaneousfistula. Enterocutaneousfistula is an extremely rare complication that is under reported in literature. Its scarce incidence mainly occurred in developing countries. We report a case of one month old boy with enterocutaneousfistula due to neglected strangulated in- guinal hernia that perforated. He presented with feculent discharge coming out of the right scrotum, episodes of bilious vomiting and ab- dominal distension prior to admission. This complication primarily occurred due to delayed diagnosis and intervention. Patient underwent succesful debridement, herniotomy as well as resection and ileo-ileal end-to-end anastomosis. Fortunately postoperative management was uneventful. This case highlights the need for prompt diagnosis and re- ferral for surgery whenever pediatric patients present with neglected strangulated hernia. Awareness on this serious complication must also be imposed to further prevent this complication.
2. Case presentation
A one month old boy was admitted with a history of intermittent re- ducible swelling at right inguino-scrotal area of one month duration. He was born at term with good postnatal condition. Patient was still able to pass stools normally through rectum with feces presenting as soft and no bloody or mucoid discharge. The parents also noticed that the swelling intermittently enlarged when patient strained or cried. However, 10 days previously patient was admitted at another hospital as his abdomen seemed distended and he had episodes of bilious vomiting resembling stool. These symptoms then abruptly subsided with the onset of fecal discharge from the right scrotum and the inguinal swelling became irre- ducible. He was then referred to our center for further evaluation.
Examination revealed the boy weighed 3.1 kgs at presentation. He was well with no signs of dehydration, hypothermia or cyanosis. Vital signs were heart rate 132 beats per minute, respiratory rate 32×/
minute, and oxygen saturation 99%. Abdominal examination wasflat, no appearance of loops of bowels, no defense muscular and positive bowel sounds. At inspection there was a discharge of fecal matter
coming out of afistula with fecal odor (Fig. 1). Surrounding skin that overlied the perineal area was erythematous and inflamed. The left inguino-scrotal area was normal. Examination of other systems did not show abnormality.
The patient's blood parameters were hemoglobin 9,2 gr/dL, hema- tocrit 25%, white cell count 21,020 cell/mm3and platelet 822,000 cell/
mm [3]. C-reactive protein to evaluate sepsis was not done. Serum electrolytes were Na+ 131 mmol/l, Cl− 110 mmol/l, K+ 5.0 mmol/l and no signs of electrolyte imbalance. Ureum and creatinine were within normal limits.
Afistulogram was done at the previous hospital using barium-based contrast material (Fig. 2). Fistulogram showed afistulous tract where segment of ileum was found in the right scrotum. The patient was planned for exploration (seeFig. 3).
Patient underwent exploratory laparotomy under general anesthesia through incision at right inguinal crest. After identification of spermatic cord the hernia sac was incised. This revealed a hernia sac containing loop of ileum which created afistulous tract in the right scrotum. The distal ileum had perforated through right scrotal skin with fecal dis- charge. The ileum loop was released from adhesions by blunt dissec- tion. The segment of ileum loop appeared necrotic hence a resection and ileo-ileal end-to-end anastomosis was done. The ileum loop was put back to intraabdomen, and after double ligation of proximal sac a herniotomy was performed. An excision offistula was done at scrotum followed by careful debridement of scrotal skin and adjoining tissue rinsed with povidone iodine and NaCl 0,9% solution. The scrotal wound was left unsutured to heal by secondary intention. Intraoperative bleeding was approximately 20 cc.
Patient was given cefotaxime 3 × 100 mg, paracetamol 3 × 40 mg and metronidazole 3 × 150 mg postoperatively. His postoperative condition was uneventful with abdomen appearedflat and presence of bowel sounds. Postoperative wound appeared closed well with no leakage. Attention to daily wound care, nutrition, and sepsis control were taken.
3. Discussion
Inguinal hernia in infants is a common congenital defect with in- direct type being the majority. Incidence of inguinal hernia range from
https://doi.org/10.1016/j.epsc.2018.07.017 Received 20 July 2018; Accepted 21 July 2018
∗Corresponding author. Jl. Salemba Raya No. 6, Jakarta Pusat, 10430, Indonesia.
E-mail addresses:[email protected],[email protected](R.P. Tamba).
Journal of Pediatric Surgery Case Reports 38 (2018) 9–11
Available online 02 August 2018
2213-5766/ © 2018 The Authors. 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/).
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3% to 5% in term infants and 13% in premature infants [1]. More cases are found in boys than in girls (ratio of 9:1) [2]. The main cause of defect is processus vaginalis that remains patent thus connecting in- traabdomen and inguinal cavities.
In this patient parents have observed appearance of swelling at right inguinal area since birth, however didn't seek further medical advice for the condition. There is a tendency to abandon these conditions as hernias typically appear intermittent and initially may not be associated with pain or discomfort in child.
Complications of neglected inguinal hernia vary from incarceration to risk of reduced testicular circulation caused by spermatic cord
compression. Risk of incarceration is amongst the frequently found complication, with rates as high as 50% in prematures atfirst trimester, 30% in mature neonates < 1 year and 15% in mature neonates > 1 year old [2]. The higher risk in infancy may be due to narrower deep inguinal ring in infants that easily predispose to incarceration [3].
In our case, patient is still younger than one year of age, thus at a higher risk of incarcerated hernia. Although incidence of incarceration decreases with age, it must always be considered in all children with hernias at any age.
Before admission, symptoms of vomiting and abdominal distension subside after fecal discharge protrude from the right scrotum.
Additionally bowel sounds were present that further indicated a de- compressing intestine through afistula tract. This showed that what may be an uncomplicated inguinal hernia atfirst have progressed into a complicated hernia with perforation creating an enterocutaneousfis- tula.
The pathophysiology of an enterocutaneousfistula in this case is due to perforation that creates potential communication between small intestine and the epidermis [4]. The leaking bowel contents macerate surrounding tissue, suppurate through fascia forming afistulous com- munication with the skin. The thin overlying skin in inguino-scrotal area also further predisposes formation offistula [3].
In our patient, afistulography was already done by the previous hospital. This imaging modality was in fact not necessary in this case to characterize the enterocutaneous fistula, as evidence of strangulated hernia perforation was already made obvious fromfindings of clinical signs and physical examination.
An enterocutaneousfistula due to neglected strangulated hernia is an extremely rare complication in infants. To date there are only eight cases of scrotalfistula in infants that have been reported [5]. Majority of these cases occurred in developing countries with inadequate health facilities such as Nigeria, Iran and India [5]. Other cases of en- terocutaneous fistula in pediatrics are secondary to complication of abdominal surgeries.
Noting that morbidity of strangulated inguinal hernia in newborn is high [6], prompt treatment were taken to prevent more necrosis of Fig. 1.Fistula tract seen in right scrotum (arrow) (April 2018).
Fig. 2.Fistulogram done at the previous hospital showed perforation of distal ileum in right scrotum (May 2018).
R.P. Tamba et al. Journal of Pediatric Surgery Case Reports 38 (2018) 9–11
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tissues and further infarction of testis. The surgical procedures carried out in this patient were herniotomy, excision offistula, debridement, resection and ileo-ileal end-to-end anastomosis.
Theoretically, in irreponible inguinal hernia without evidence of strangulation, a manual reduction was done under sedation followed by herniotomy 48–72 h later [5]. However this was not applicable in our center and on this case. At our center, for strangulated hernia with no signs of perforation, patient was sedated with rectal diazepam followed by careful observation of whether the hernia spontaneously reduce on its own within 8 h time. If this approach is succesful then 24–48 h later an elective surgery is planned, whereas if failed then a prompt surgical repair is done. In this patient, since there were both evidence of strangulation and perforation hence we proceeded with prompt surgical approach.
4. Conclusion
This rare case report highlights the importance of an early diagnosis and referral for prompt surgical approach of infant inguinal hernia. Our case showed that there is a great sense of urgency for strangulated hernia repair in order to reduce further complications. Parents should also be encouraged and made aware of inguinal swellings in children and to actively seek early medical intervention. With proper recogni- tion and surgical approach we can help prevent morbidity and mor- tality of enterocutaneousfistula due to neglected strangulated hernia in infant.
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
No funding or grant support.
Authorship
All authors attest that they meet the current ICMJE criteria for Authorship.
Conflicts of interest
The authors declare that there are no conflicts of interest regarding the publication of this paper. The following authors have nofinancial disclosures: (RPT, DR, BK).
Appendix A. Supplementary data
Supplementary data related to this article can be found athttps://
doi.org/10.1016/j.epsc.2018.07.017.
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[4] Amjad A, Qazi S, Nazir Z. Enterocutaneousfistulae in children–a management challenge. J Coll Phys Surg Pakistan 2008;18(8):526–7.
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Fig. 3.IntraoperativefindingsA. Necrotic segment of ileum (arrow) B. Resection and ileo-ileal end-to-end anastomosis performed C. Post-resection and anastomosis.
R.P. Tamba et al. Journal of Pediatric Surgery Case Reports 38 (2018) 9–11
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