An unusual form of incisional hernia: A case report of Littre's hernia

Abstract

Introduction

Littre's hernia (LH) is a rare condition involving Meckel's diverticulum within a hernia sac, with an incidence of <0.1 % of all complicated hernias. To this day, only a few case reports have been published concerning this entity.

Case presentation

A 48-year-old patient with a history of four midline C sections and an incarcerated incisional midline hernia was admitted to the emergency department for small bowel obstruction evolving for 12 h. Examination revealed an incarcerated midline incisional hernia. An abdominal CT scan was performed, showing a multi-orifice incisional hernia with a small bowel loop and a 3 cm abscess. The patient underwent an urgent midline laparotomy. During surgery, we found a small bowel loop with a perforated Meckel's diverticulum, located 50 cm from the ileocaecal valve, associated with a 3 cm abscess. Surgical drainage of the abscess, resection of 20 cm of small bowel, including the diverticulum, and an appendicectomy were performed. The incisional midline hernia was managed by herniorrhaphy. The post-operative course was uneventful.

Clinical discussion

Surgeons need to keep in mind the possibility of discovering Meckel's diverticulum in a hernia sac in every incarcerated hernia. Complete history intake and careful physical examination are important to uncover signs prompting clinical suspicion. Littre's hernia is rare and difficult to diagnose, with no distinguishing clinical features or physical signs.

Conclusion

Managing LH involves treating the symptomatic Meckel's diverticulum with various resection methods and the hernia itself, with mesh application being a controversial topic. Prophylactic resection remains debatable among experts.

Highlights

• •Littre’s hernia is an exceedingly rare entity with an incidence of < 0.1% of all complicated hernias.
• •While many hernia sites have been reported, no Littre’s hernia has ever been documented on an incisional site.
• •The lack of specific physical and radiological signs renders preoperative diagnosis challenging.
• •While treatment is based on diverticulum resection and hernia defect repair, mesh application is still controversial among authors.
• •Prophylactic resection of Meckel’s diverticulum remains a debatable subject, with no clear guidelines issued.

1. Introduction

A LH is characterized by the existence of Meckel's diverticulum within a hernia sac [1]. According to published literature, the incidence of LH is <0.1 % of all the reported complicated hernias, thereby rendering it an extremely rare entity [2]. While Meckel's diverticulum is the most frequent congenital malformation in adults, its complications in the form of LH are observed in <1 % of cases [3]. LH is mainly diagnosed as inguinal, femoral, and umbilical hernias, but other rare sites have also been reported (obturator, Spiegel, and ventral hernias) [3]. To the best of our knowledge, no cases of LH on the incisional site have ever been reported.

In this paper, we report the first case of Littre's hernia in an incisional midline site to document this highly rare entity and to raise awareness among young surgeons concerning the diagnosis and management of this unique disease. This case report adheres to the SCARE Criteria [4].

2. Case presentation

A 48-year-old patient, with a history of four midline C-sections and a previous surgery for an incarcerated incisional midline hernia, was admitted to our emergency department due to a reincarcerated incisional hernia evolving for 12 h. During the initial history intake, the patient did not report signs of chronic pain in the right iliac fossa or any episodes of lower gastrointestinal bleeding.

On examination, the patient had a fever of 39 degrees; she was lucid; her blood pressure was 130/70 mmHg; and her heart rate was 100 BPM. The Respiratory assessment did not show signs of acute distress. Abdominal examination revealed a 5 cm bulge developed along the midline incision. This bulge was of firm consistency, irreducible, painful spontaneously without impulsion or expansion at cough, and associated with local inflammatory signs. No abdominal distension or rebound tenderness was recorded. The rectal examination did not reveal Douglas's outcry or bloody stools.

Laboratory findings showed an elevated white blood cell count and a high concentration of C-reactive protein. No renal function impairments or electrolyte disorders were recorded. A CT scan of the abdomen with IV contrast was performed, showing a multi-orifice incisional hernia containing an incarcerated small bowel loop with signs of ischemia, in contact with a 3 cm abscess. No bowel dilatation was noted (Fig. 1).

Fig. 1.

Preoperative CT scan. (a) and (b) axial views of an arterial phase CT scan images showing the incisional hernia containing a small bowel loop and a 3-centimeter abscess.

The patient underwent an urgent midline laparotomy. During surgery, upon opening the hernia sac, we discovered an incarcerated yet viable small bowel loop with a perforated Meckel's diverticulum, located 50 cm from the ileocaecal valve. An associated 3 cm abscess was also identified (Fig. 2). A thorough exploration of the entire abdominal cavity did not reveal any additional congenital malformations or intraperitoneal effusion. Initially, surgical drainage of the abscess was performed, followed by the resection of 20 cm of small bowel using a surgical stapler, which included the perforated Meckel's diverticulum. To restore digestive continuity, we conducted a manual side-to-side anastomosis. The appendix exhibited mild inflammation. An appendicectomy was also performed with the stump secured using sutures. The incisional midline hernia was managed through herniorrhaphy utilizing absorbable, synthetic, and braided overlock sutures. The post-operative course was uneventful, and the patient was discharged a few days later.

Fig. 2.

Intraoperative images. (a), (b) and (c) showing the congestive ileal loop found in the hernia sac with a perforated Meckel's diverticulum (White arrows). The proximal loop is pointed at with the dissecting forceps in (b). The lumen of the perforated diverticulum is shown in (c).

The anatomopathological report showed a complicated Meckel's diverticulum, without any signs of ectopic tissue. The appendicular specimen did not show signs of malignancies.

3. Discussion

A Littre's hernia is an exceedingly uncommon condition that develops when Meckel's diverticulum protrudes through a herniary orifice [1]. Since the first case description by Alexis de Littre in the 18th century, it has been recommended that all cases, regardless of how they were diagnosed or treated, should be published [5]. The latest literature review published in 2018 by Schizas et al. reported that only about 53 cases have been documented throughout history [6].

While the exact pathogenesis of Littre's hernia is yet to be determined, most authors have agreed that an inflammatory event causes Meckel's diverticulum to adhere to the peritoneum prior to testes descent, thus preventing the vaginalis from closing and maintaining it in a patent state [7]. Others have suggested that the antimesenteric location on the ileum border makes Meckel's diverticulum more prone to slide through any abdominal orifices [8]. One of the most challenging aspects of LH is preoperative diagnosis; there are no distinguishing clinical features or specific physical signs that set LH apart from other hernias. However, a complete history intake revealing history of rectal bleeding, chronic pain in the right iliac fossa, and incomplete taxis of the hernia has to warn the surgeon that the case in hand might be a LH [9]. LHs are presented to the emergency departments as incarcerated inguinal, femoral, and umbilical hernias; less frequent sites were also reported, like obturator, Spigelian, and ventral abdominal hernias [6]. It's the incidence of each hernia site that differs according to studies [10]. Although contemporary imaging techniques are widely available, preoperative diagnosis of LH remains challenging. According to recent studies, even with the association of several imaging modalities (ultrasonography, CT imaging, scintigraphy, and technetium scan), it might not be possible to confirm the diagnosis of Littre's hernia [11,12]. Because of its unspecific clinical symptoms and the lack of pathognomonic radiological signs, a positive diagnosis of LH is made intraoperatively [6]. Depending on perioperative findings, LH is classified as true or mixed. True LH is more frequently encountered, and the hernia sac contains Meckel's diverticulum only [13]. Mixed LH, on the other hand, is less frequently documented and includes a segment of the small bowel alongside Meckel's diverticulum [13]. In our paper, we report the first case of mixed Littre's hernia on the incisional site. On history intake, our patient did not report any chronic symptoms suggesting a complicated Meckel's diverticulum. Preoperative radiological investigations were performed but did not affirm the diagnosis of LH. This classification provides an explanation for the associated small bowel obstruction present in mixed LH (about 34 % of all documented LH cases) and its absence in true LH-reported cases [14]. Depending on the time elapsed between the first clinical symptoms and diagnosis, Meckel's diverticulum undergoes pathological changes ranging from simple hyperemia to necrosis and perforation [15]. Some rare cases of non-incarcerated LH with diverticulum perforation were reported; this complication was probably related to acid secretion by ectopic tissue within the diverticulum [6]. Our patient reported signs of small bowel obstruction evolving for 12 h. Intraoperative findings confirmed the diagnosis of mixed LH, complicated by the perforation of Meckel's diverticulum and a 3 cm abscess. Managing LH is a diligent procedure performed in two steps: First, treat the symptomatic Meckel's diverticulum. According to published literature, the appropriate surgical procedure in cases of Meckel's diverticulum complicated by bowel obstruction, inflammation, or perforation is segmental or wedge resection [16]. The second step is treating the hernia itself. Hernia defect repair can be achieved through herniorrhaphy or tension-free mesh application [17]. While mesh application is no longer a subject for debate in incarcerated hernia [17], it's the perforation of Meckel's diverticulum and surgical field contamination that makes surgeons reluctant to apply mesh in such conditions, as only limited and low-quality evidence papers have been published concerning this issue [6,17].

In our case, we performed bowel resection with side-to-side anastomosis, associated with suture herniorrhaphy repair. Although our patient was treated successfully after presenting with a complicated Meckel's diverticulum, the question remains to be answered: was this complication preventable by a prophylactic resection? According to literature reviews, there are no clear guidelines issued concerning the management of asymptomatic Meckel's diverticulum [18]. Experts derived recommendations from their personal experience, resulting in divergent management guidelines [18]. Keeping these different directives in mind, it is advised that a silent Meckel's diverticulum in elderly patients should be respected [18]. Unlike patients younger than 50 years old, Meckel's diverticulum with a length > 2 cm should be resected due to its high risk of becoming complicated [18]. This exceptional case of Littre's hernia on an incisional site emphasizes the importance of diligent and thorough abdominal cavity exploration prior to performing incisional hernia repair. It is crucial for surgeons to carefully examine the entire abdominal cavity to identify any potential digestive malformation, especially in patients with previous surgeries [17]. Failing to do so can result in unwarranted complications like, in our case, Littre's hernia, requiring more surgeries.

4. Conclusion

Littre's hernia is an exceedingly rare complication of Meckel's diverticulum that is mostly the focus of case reports, as only about 50 papers have been documented in the literature during the previous 300 years. Management of LH is based on surgery. Treatment of the complicated diverticulum is resection with various possible modalities. The use of mesh or suture closure of the hernia defect remains a surgeon's call, depending on perioperative findings. Prophylactic resection remains a debatable subject among experts with no clear guidelines.

Consent of publication

A written consent was obtained from the patient to publish this case report.

Ethical approval

Ethical approval was deemed unnecessary by our institutional ethical committee, as the paper is reporting a single case that emerged during normal practice.

Funding

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

Author contribution

Racem Trigui: writing-Original draft preparation. Anis Hasnaoui: Conceptualization, Writing-Reviewing and Editing. Sihem Heni: Data curation. Houda Kammoun: Data curation. All authors read and approved the final manuscript.

Guarantor

Dr Hasnaoui Anis

Dr Trigui Racem

Registration for research study

N/A.

Declaration of competing interest

The authors declare that they have no competing interests.