Rare Co-Existence of Amyand’s and Littre’s Hernia Treated with Appendectomy and Bowel Resection Followed by Polypropylene Mesh Application

Background

Amyand’s hernia and Littre’s hernia are 2 rare types of hernia. Amyand’s hernia is a type of inguinal hernia, representing <1% of all inguinal hernias [12], it was first reported by British surgeon Claudius Amyand. He performed the first reported successful appendectomy on an 11-year-old boy by removing the perforated appendix from the right inguinal hernia sac [3–5]. Littre’s hernia is another rare hernia, which contains Meckel’s diverticulum, the most common congenital abnormality of the gastrointestinal tract [6]. Alexis Littre was the first to describe hernias containing intestinal diverticula, such us Meckel’s diverticulum [7]. Both of these rare types of hernias – Amyand’s and Littre’s – constitute less than 1% of all types of hernias. The diagnosis is often difficult to confirm preoperatively; therefore, it is almost always found intraoperatively. The difficulties that a surgeon confronts during the treatment of these inguinal hernias include: 1) the performance of appendectomy (in the case of Amyand’s) or an enterectomy (in the case of Littre’s), and 2) the need to use tension-free mesh for the hernia repair. Therefore, the surgeon has to quickly decide which type of operation to perform.

Case Report

The presented case was a 65-year-old patient who was diagnosed with a right-side inguinal hernia 10 years ago, without complaining about any inconvenience or pathological symptoms. He was a heavy smoker (1 pack/day) and occasionally social drinker, and he received medication for arterial hyper-tension (ramipril 5 mg 1×1) without experiencing any additional health problems. Based on Surgical Endoscopy guidelines (Surg Endosc 2015), the surgical team opted for an open approach because there was no recurrent inguinal hernia and the abdomen was ‘hostile’ because the patient had been submitted to laparoscopic cholecystectomy in the past [8]. During the operation, the content of the hernia sac was identified and the most intriguing finding was that it consisted of the appendix and Meckel’s diverticulum. This indicates that the type of hernia was a combination of Amyand and Littre’s hernia (Figure 1). After evaluating the macroscopic pathology of the appendix intraoperatively and taking into consideration the World Society of Emergency Surgery (WSES) Grading System [9], we came to the conclusion that there was no sign of inflammation in the appendix and the Meckel’s diverticulum (Figures 2, 3). According to Losanoff and Basson guidelines [10,11], in the case of Amyand’s hernias, the appendix must be removed in young patients regardless of the state of inflammation. Therefore, we decided to proceed with an appendectomy (Figure 3) and a partial enterectomy of the diverticulum with fully controlled conditions in order to avoid contamination that could spread the infection. After the appendectomy and the enterectomy were completed, the surgical field swab was conducted and the operation restored the hernia. In the above case, a polypropylene mesh was used according to Lichtenstein hernioplasty.

The patient was intraoperatively administered a single dose of the prophylactic antibiotic cefoxitin. He patient was discharged on the second postoperative day. At 12 months after the surgery, he shows no sign of complication or recurrence of the hernia.

Discussion

Amyand’s hernia and Littre’s hernia are rare hernias accounting for <1% of all types of hernias [1,2,7]. D’ Alia et al. reported that an inflamed appendix is even rarer than a normal appendix in a sac and determined that this proportion is about 0.08% [1]. Due to their rarity, only a few cases of these hernias have been described in the literature. The diagnosis based on the contents of the sac is almost always confirmed intraoperatively. In 1999 Weber et al. [10] confirmed the results of a previous report by Thomas [12] that only 1 of 60 patients is preoperatively diagnosed based on the contents of the sac. A difficulty arises when the surgeon must quickly decide whether to perform an appendectomy or a partial enterectomy and what technique to use for the hernia repair. According to recent international research papers, the decision depends upon the inflammation signs of the appendix or the Meckel’s diverticulum and the macroscopic pathology of each of the specimens. In this respect, the decision is often individualized. According to Losanoff and Basson’s guidelines modified by Signal and Gupta [13], in a case where there was an acute appendicitis, appendectomy must be performed without use of a mesh for repair of the hernia. If there is no inflammation, the decision is often difficult. Ofili et al. believed that in young patients, the appendix must be removed regardless of the sign of inflammation and thus insisted on incidental appendectomy. The probability of a patient with Amyand’s hernia displaying acute appendicitis during the following years is higher than in the general population. This is due to the pathophysiology of an Amyand’s hernia, because the appendix in a sac is vulnerable to adhesions, microtraumas, and tissue ischemia [13]. On the other hand, the decision to use mesh after an appendectomy of a normal appendicial specimen is up to the surgeon. Sharma et al. [15] argue that it is safe to repair a hernia after an appendectomy of a normal appendix with the use of a mesh. Our data are consistent with the above findings, since our cases were the first to use polypropylene mesh in elderly patients in absence of postoperative complications or recurrence [14,15]. The present case of hernia is of great scientific interest due to its rarity. Actually, the hernia sac contained both the appendix and Meckel’s diverticulum. We performed an appendectomy and partial enterectomy with fully controlled conditions and we restored both of the hernias with the use of a polypropylene mesh and our patient had no complication or recurrence of the hernia at 1 year after the operation. Therefore, the present report is the first to describe a successful repair in a patient with combined Amyand’s hernia and Littre’s hernia.

Conclusions

The data from our study present for the first time a case of a typical Amyand’s hernia combined with Littre’s hernia. Both the appendix and Meckel’s diverticulum were resected despite the absence of any signs of inflammation or ischemia. The procedure continued with the placement of a mesh according to Lichtenstein’s technique.