Introduction

The vermiform appendix in a femoral hernia sac is an uncommon operative finding, and appendicitis in a femoral hernia sac is even more infrequent [1, 2]. De Garengeot reported the presence of a vermiform appendix in a femoral hernial sac in the early eighteenth century, and the condition is aptly named after him [1–3]. The incidence of this disease is estimated to be between 0.5 and 5% of all femoral hernias [1, 2, 4]. Due to the infrequent occurrence, De Garengeot hernia has been sparingly reported as case reports and small case series.

We present a retrospective case series of seven consecutive patients presenting with De Garengeot hernia over a 16-year period at a tertiary referral centre. This report is probably the largest case series of its kind.

Materials and methods

From January 1991 to September 2006, 457 femoral hernia repairs were performed at Sawai Man Singh Hospital, Jaipur. Of these, the operation register identified nine patients with a record of appendicectomy and femoral hernia in a single operative session. Two of these patient’s operative records had mere findings of incidental femoral hernia during appendicectomy and were therefore excluded from the case series.

Therefore the hospital records of seven patients were reviewed retrospectively. The demographic characteristics, documented history, operative notes, biochemical and radiological investigations, perioperative outcome, histopathology and recurrence rates were evaluated.

Results

We have summarised the base line characteristics of these seven patients in Table 1. Six of seven patients were admitted as an emergency with an acute onset of right infra-inguinal lump, while one patient was admitted for investigation of right groin lump following failed antibiotic therapy misdiagnosed as inflamed inguinal lymph node. The median duration of symptoms at presentation was 5.2 (range 2.5–21) days. All patients had an abdominal X-ray that failed to reveal any abnormality, while one patient who presented with chronic groin lump of 3-week duration had an ultrasound of the abdomen and pelvis showing a hernia containing bowel in the right groin (Fig. 1). White cell count was normal in all our patients.

Table 1 Baseline characteristics of the patients
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Fig. 1
figure 1

Preoperative ultrasound of the groin

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Table 2 represents the perioperative summary of our patients. None of the patients were diagnosed preoperatively with De Garengeot hernia. Intraoperatively, the macroscopic assessment of the appendix by the surgical team was used to determine the appendiceal inflammation. Three out of seven patients had an abnormal appendix. Two of these patients had inflamed appendix, while one patient had a perforated appendix in the femoral hernia sac. The appendix was normal in all other patients (n = 4). Due to suspicion of infection, all patients received preoperative third-generation cephalosporin and metronidazole and were operated upon within 10 h of admission. Patients with pathological appendix further received a 5-day course of third-generation cephalosporin and metronidazole.

Table 2 Operative and postoperative summary of the patients
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All patients were operated upon using an infra-inguinal incision. In patients with normal appendix (n = 4), the appendix was simply reduced and the femoral hernia was repaired using a prolene mesh. In patients with inflammed appendix (n = 2), appendicectomy was done via an infra-inguinal incision and the femoral canal was closed using interrupted prolene sutures because of fear of mesh infection. In the patient with a perforated appendix (n = 1), a separate right iliac fossa incision was carried out in addition to an infra-inguinal incision to facilitate a good pelvic wash out and proper exposure of the appendix and caecum. The repair of the hernia was again done with interrupted prolene sutures. This patient did suffer from a minor wound infection, which settled with antibiotic therapy. The median postoperative stay of the patients with pathological appendix was 3 days (range 2–21) in comparison to 2 days (range 1–3) for patients with normal appendix. No recurrence has been noted to date and there was no mortality in the perioperative period.

Discussion

Rene Jacques Croissant De Garengeot, a Parisian surgeon, first reported an appendix in a femoral hernia sac in 1731, but it was 5 decades later in 1785 that Hevin performed the first appendicectomy in a femoral hernia sac [1]. The diagnosis of De Garengeot hernia in the present series was based on the following criteria—a femoral hernia sac containing either normal appendix, inflamed appendix or perforated/gangrenous appendix. This condition, though rare, is well documented in the literature [5, 6]. The incidence of presence of an appendix in femoral hernia is reported to be 0.5–5% [1–5]; the reason for this wide variation is the paucity of cases and no published large case series. The present series had an incidence of 0.15%, which is well below the figures quoted above.

Controversy reigns regarding the pathogenesis of De Garengeot hernia [5–8]. According to the congenital theory, the abnormal attachment of the appendix to the caecum, secondary to different degrees of intestinal rotation, gives rise to a pelvic appendix, which has a high risk of entering the hernial sac of the pelvic peritoneum [7, 8]. Other theories propose appendicitis in femoral hernia to be either a primary obstructive event or secondary to constriction of the appendix by a tight neck of hernia sac in a less roomy pelvis [5, 6, 9]. The patient with the perforated appendix in the present study had a wide hernia neck, but the other two patients with inflamed appendix had a comparatively narrow neck of hernia. We thus infer that pathogenesis of De Garengeot hernia is a combination of both the primary and secondary events mentioned above. The infra-inguinal approach in this series had the drawback of limited surgical exposure and therefore the operative notes fail to comment on the position of the appendix. We thus feel that the congenital theory still requires more data for acceptance.

Femoral hernia occurs more frequently in females in an approximate ratio of 2:1 to males [10, 11]. An analysis of case reports quotes the mean age of De Garengeot hernia patients to be 69 years [7]. In contrast to this, the median age of our patients was 55 (range 35–89) years. The female:male ratio was approximately 2:1.

Preoperative diagnosis of this condition is difficult. The clinical signs and symptoms of De Garengeot hernia are that of incarcerated femoral or inguinal hernia and include vague abdominal pain and tenderness and an erythematous groin lump [3, 7]. The signs of appendicitis are overshadowed by a tight femoral hernia neck and pelvic rigidity: this anatomical feature prevents the spread of inflammation to the peritoneal cavity, and thus even patients with perforated appendix rarely present with florid peritonitis [10, 12, 13]. All patients in our study presented with groin lumps on the right side, with no features of bowel obstruction. Nguyen et al. [7] report that the symptoms of the patient may be chronic (present for up to 15 years) or acute. In the majority of our patients (n = 6) the duration of symptoms was 1–2 days. However, the oldest patient in our series presented with a tender groin lump of >3 weeks duration and was initially treated for inflamed inguinal lymph node. White cell count is an inconsistent investigation and was normal for all our patients. Few other case reports too report the same findings as ours [9, 11].

Abdominal X-ray does not aid in the diagnosis of De Garengeot hernia, but assists in recognising associated small bowel obstruction [14]. Ultrasound has been used to assess the groin in unsuspected cases [9]. CT scan of the groin has been established to be highly sensitive recently [6, 8, 10], but an evident incarcerated femoral hernia leaves little room for radiological investigations. Yet in the unsuspected cases or if in doubt, contrast-enhanced CT scan of the groin may confirm the diagnosis. We feel retrospectively that abdominal X-rays done on our patients were unwarranted as none had features of obstruction and all these patients were exposed to unnecessary radiation.

The treatment of this disease entity is emergency surgery. Incidental appendicectomy is not a preferred practice anymore [14], henceforth reduction of the appendix and primary mesh repair was performed on four out of seven patients with normal appendices. Choice of repair in a femoral hernia containing pathological appendix is debatable. Generally prosthetic material is not preferred in a contaminated field due to the risk of infection [11, 14], but a few reports have mentioned mesh repair even in the presence of an inflammed appendix with no postoperative infection [1, 8, 9]. In the present series, all patients were approached with an infra-inguinal incision, but the patients with inflammed appendix (n = 2) had an appendicectomy and hernia repair with interrupted sutures through the same incision. Perforated appendix was encountered in one patient, and an appendicectomy was performed by a separate abdominal incision and the hernia repair was undertaken with interrupted sutures via a separate groin incision. A thorough pelvic wash out was performed before closure. Still, this patient suffered superficial skin infection of the infra-inguinal incision, which responded to antibiotics. Laparoscopy has been described as a useful aid in the evaluation of incarcerated groin hernia [15], reduction of hernia [16] and also resection of the bowel in the event of strangulation [17]. High operative costs and not so readily available laparoscopic instruments in a third world set up prevented the use of laparoscopy in the present study.

The infection rate of the De Garengeot repair is reported to be approximately 29% [7]. This is in contrast to an infection rate of 14% in our series, which is probably due to early surgical intervention and strict adherence to surgical principles. Grave complications such as necrotising fasciitis and death have been reported [7–9], but the present series had no mortality and no major complications.

Conclusion

De Garengeot hernia is a rare condition. Preoperative diagnosis of this surgical entity is unlikely in an emergency presentation, and the condition becomes evident only during the intraoperative period. Choice of repair in the event of a pathological appendix is still debatable and needs more data to establish proper guidelines. Laparoscopy may be the procedure of choice in the future for the operative diagnosis and subsequent repair.