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Incarcerated Littre's hernia in a neonate presenting as enteroscrotal fistula

 Department of Paediatric Surgery, SMS Medical College, Jaipur, Rajasthan, India

Date of Submission12-Aug-2022
Date of Decision09-Sep-2022
Date of Acceptance26-Oct-2022
Date of Web Publication30-Nov-2022

Correspondence Address:
Rahul Gupta,
Associate Professor, Department of Paediatric Surgery, SMS Medical College, Jaipur, Rajasthan
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/jiaps.jiaps_110_22



A 27-day-old male neonate, presented with feculent discharge from the scrotum. Operative findings revealed incarcerated right inguinal hernia with perforated Meckel's diverticulum as its content, leading to enteroscrotal fistula. Resection of the Meckel's diverticulum and end-to-end ileoileal anastomosis was performed along with repair of inguinal hernia from within the abdominal cavity. The outcome was favorable. Enteroscrotal fistula due to incarceration of inguinal hernia is a rare presentation. We are adding to the literature, an extremely rare case of incarcerated Littre's hernia in the right inguinal region presenting as enteroscrotal fistula in a neonate.

Keywords: Enteroscrotal fistula, incarcerated, inguinal hernia, Littre's hernia, Meckel's diverticulum, neonate

How to cite this URL:
Gupta R, Mathur P. Incarcerated Littre's hernia in a neonate presenting as enteroscrotal fistula. J Indian Assoc Pediatr Surg [Epub ahead of print] [cited 2023 Jan 29]. Available from: https://www.jiaps.com/preprintarticle.asp?id=362391

   Introduction Top

Meckel's diverticulum presenting in the neonatal age group is rare.[1] It usually presents in neonates as small bowel obstruction, whereas perforation of Meckel's diverticulum is rare.[1] The presence of Meckel's diverticulum as a content of the hernia sac is called Littre's hernia.[2] Here, we present an extremely rare case of incarcerated Littre's hernia in the right inguinal region presenting as enteroscrotal fistula in a neonate.

   Case Report Top

A 27-day-old term male baby, weighing 2300 g, born by vaginal delivery, presented with complaints of feculent discharge from the scrotum, abdominal distension, and bilious vomiting for 2 days. The neonate was a first-born male child. There was a history of inguinoscrotal swelling which was overlooked by the parents. At presentation, the baby was hemodynamically stable. The abdomen was distended; on inguinoscrotal examination, there was a tense, tender, irreducible inguinal hernia with inflammation of the local skin and edema at the base of the penis. Scrotal examination revealed a contaminated wound in the right hemiscrotum with markedly inflamed (hyperemic) margins and feculent material coming through the wound [Figure 1]. The anal examination was unremarkable. Abdominal radiographs showed dilated small bowel loops with the presence of gas shadow in the right hemiscrotum; the testis was palpable separately [Figure 1]. Baseline blood investigations were normal. A provisional diagnosis of enteroscrotal fistula was made. The patient was optimized and a laparotomy was performed through the right lower transverse incision. Operative findings were incarcerated inguinal hernia with perforated Meckel's diverticulum (Littre's hernia) as its content, leading to enteroscrotal fistula [Figure 1]. There was an absence of peritoneal contamination. Resection of the Meckel's diverticulum and end-to-end ileoileal anastomosis was performed. The inguinal hernia was repaired from within the abdominal cavity using purse-string absorbable sutures. The postoperative course was uneventful. The histopathological examination of the resected ileum with Meckel's diverticulum confirmed mural necrosis, chronic inflammation, marked serositis, and perforated diverticulum without any heterotopic mucosa. The outcome was favorable and the patient is gaining weight on follow-up [Figure 1].
Figure 1: Preoperative photograph (a) shows feculent material coming out through a wound in the right hemiscrotum with inflamed irregular margins and inguinal swelling; the inset image reveals a distended abdomen. Abdominal radiograph (b) shows dilated small bowel loops with the presence of gas shadow in the right hemiscrotum; intraoperative photographs (c) show perforated Meckel's diverticulum with its omphalomesenteric vessel (green arrows) retrieved from the right inguinal hernia (Littre's hernia) and the site of enteroscrotal fistula (black arrow). Postoperative photograph (d) shows healed scrotal scar and right infraumbilical incision

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   Discussion Top

Meckel's diverticulum is a true diverticulum with all three layers of the small intestine. It is an omphalomesenteric duct remnant due to its incomplete obliteration. It is named after Johann Friedrich Meckel who reported it in 1809.[1],[2] It is the most common gastrointestinal tract malformation (2% of the population).[1] Although most are asymptomatic, the typical presentation is under 2 years of age with painless bright red rectal bleeding and bowel obstruction/perforation/volvulus/intussusception. In neonates, the presentation is rare and is usually with small bowel obstruction due to associated Meckel's band obstruction. Risk factors predisposing perforation of Meckel's diverticulum are a perinatal vascular insult, hypoxia, steroid therapy, and exchange transfusion.

Meckel's diverticulum presenting as a content of the inguinal hernia sac is known as Littre's hernia. It was coined by Riecke (1841) after Alexis Littré who described the entity in 1700. The usual sites of Littre's hernia are inguinal (1/2), umbilical and femoral (1/5 each), and other sites (1/10 cases).[2] Littre's hernia is very rare in neonates. A review of literature of complicated Meckel's diverticulum including incarcerated Littre's hernia in neonates revealed an absence of any similar case with enteroscrotal fistula.

Delayed presentation of inguinal hernia results in obstruction, incarceration, strangulation, and infrequent enterocutaneous fistula formation. The reported incidence of incarceration ranges from 12% to 31%.[3] Enteroscrotal fistula (scrotal enterocutaneous fistula) is a rare complication due to the incarceration of an inguinal hernia followed by strangulation.[1],[4],[5]

Enteroscrotal fistula is a problem of third-world countries due to a lack of medical knowledge, locally prevalent myths, availability of local quacks, late presentation, and unawareness among the pediatrician about the timings of surgery for inguinal hernia in children, especially neonates.

The presence of the spontaneous enteroscrotal fistula allowed decompression of the bowel and temporary reprieval of the intestinal obstruction in the present case. This allowed us time to perform preoperative optimization before any septic complications ensue. Furthermore, there is a risk of testicular ischemia and frank gangrene of the testis from vascular compression with prolonged incarceration. Proper and timely surgical management can avert such a severe complication.

Surgical exploration is performed either through the inguinal region or abdomen. The author (RG) recommends an abdominal approach in a neonate presenting with enteroscrotal fistula due to incarcerated inguinal hernia. As etiopathogenesis of perforation of Meckel's diverticulum is different from Necrotizing enterocolitis (NEC), it is safe to perform primary anastomosis instead of stoma formation following resection of Meckel's diverticulum in neonates, even if there is low birth weight. A stoma may be considered if there is hemodynamic instability or generalized peritoneal contamination.

Early diagnosis of inguinal hernia, proper counseling of parents (regarding its associated complication), timely referral to a pediatric surgeon, and appropriate surgical management is principal in preventing the associated morbidity and mortality.

To conclude, incarcerated Littre's hernia in the right inguinal region presenting as enteroscrotal fistula is extremely rare. Early diagnosis and management of inguinal hernia may prevent complications associated with delayed presentation.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

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Conflicts of interest

There are no conflicts of interest.

   References Top

Ayari F, Achour R, Boussetta A, Cheour M, Bensmail T, Kacem S, et al. Neonatal perforation of Meckel diverticulum: About two cases. Health Care Curr Rev 2017;5:183.  Back to cited text no. 1
Ayyub M, Jayagowri K, Karthikeyan S, Idhrees M, Kamaludeen MN. A surgical surprise: Meckel's diverticulum complicating as Littre's hernia. MOJ Surg 2020;8:74-6. DOI: 10.15406/mojs.2020.08.00175.  Back to cited text no. 2
Sharif M, Ijaz L, Iqbal S, Sheikh A. Enteroscrotal fistula: A rare complication of incarcerated inguinal hernia. APSP J Case Rep 2010;1:21.  Back to cited text no. 3
Rattan KN, Garg P. Neonatal scrotal faecal fistula. Pediatr Surg Int 1998;13:440-1.  Back to cited text no. 4
Orelaru FO, Reddy NS, Brahmamdam P. Perforated Meckel's diverticulum in a neonate. J Pediatr Surg Case Rep 2018;37:37-40.  Back to cited text no. 5


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