Amyand's hernia: Appendix in hernia or hernial appendicitis?

Praveen Mathur; Priyanka Mittal; Ajay Kumar

doi:10.4103/jiaps.jiaps_64_22



ORIGINAL ARTICLE

Year : 2023 \| Volume : 28 \| Issue : 3 \| Page : 206-211

Amyand's hernia: Appendix in hernia or hernial appendicitis?

Praveen Mathur, Priyanka Mittal, Ajay Kumar
Department of Pediatric Surgery, Sawai Man Singh Medical College, Jaipur, Rajasthan, India

Date of Submission	13-May-2022
Date of Decision	09-Jan-2023
Date of Acceptance	09-Jan-2023
Date of Web Publication	02-May-2023

Correspondence Address:
Priyanka Mittal
Department of Pediatric Surgery, Sawai Man Singh Medical College, Jaipur - 302 004, Rajasthan
India

Source of Support: None, Conflict of Interest: None

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DOI: 10.4103/jiaps.jiaps_64_22

Abstract

Purpose: The presence of an appendix in the inguinal hernia sac is defined as Amyand's hernia (AH). This study intends to present the authors' experience in dealing with this entity and also to present a discussion on possible need of updating its definition, classification, and management.
Materials and Methods: A retrospective analysis of records of all pediatric patients undergoing surgery for congenital inguinal hernia in a single unit from January 2017 to March 2021 was done. Patient's demographics, clinical presentation, preoperative investigations, peroperative findings, and postoperative outcomes were recorded and analyzed.
Results: AH was found in eight patients. All were boys. The median age of presentation was 20.5 months (range 2 months to 36 months). The mean duration of symptoms was 2 days (range 2 to 4 days). All patients presented with incarcerated inguinoscrotal swelling (right sided = 5, left sided = 3); associated with pain. An abdominal radiograph and ultrasonography were done for all. All patients underwent emergency surgery. Exploration was done for all through an inguinal incision. The appendix was found inflamed for two patients, and appendectomy was done for the same. None of the patients underwent incidental appendectomy. Wound infection, secondary appendicitis, and recurrence were not seen for any of the patients. The authors have also proposed a revised definition and classification of AH.
Conclusion: AH is an interesting entity and many questions like the need for incidental appendectomy remain unanswered. An updating of the definition and classification system can probably offer some solution in this regard. However, more research is warranted in this regard.

Keywords: Amyand's hernia, appendicitis, appendix, hernial appendicitis, inguinal hernia

How to cite this article:
Mathur P, Mittal P, Kumar A. Amyand's hernia: Appendix in hernia or hernial appendicitis?. J Indian Assoc Pediatr Surg 2023;28:206-11

How to cite this URL:
Mathur P, Mittal P, Kumar A. Amyand's hernia: Appendix in hernia or hernial appendicitis?. J Indian Assoc Pediatr Surg [serial online] 2023 [cited 2023 May 31];28:206-11. Available from: https://www.jiaps.com/text.asp?2023/28/3/206/375527

Introduction

Inguinal hernia is defined as a protrusion through the lower abdominal wall into the inguinal canal.^[1] In spite of being the most common surgery performed, time and again it is known to have eccentric presentations, in terms of sac contents. The presence of a vermiform appendix, irrespective of the presence or absence of inflammation, in an inguinal hernia sac is referred to as Amyand's hernia (AH).^[2] This entity is named after Claudius Amyand, who first described this condition in 1735 in an 11-year-old male presenting with a discharging fecal fistula whose course tracked to a perforated appendix in the hernia sac.^[3] It was then only when the first appendectomy was reported in English language literature.^[3]

Most of the times, it masquerades clinically as an incarcerated or strangulated hernia; hence it is usually an intraoperative diagnosis.^[4] The role of radiological investigations and need to do an incidental appendectomy have always been a topic of debate. With this background, the authors are presenting their experience in the management of eight pediatric patients of AH presenting in a single unit over 4 years at a tertiary referral center and teaching medical institute, and present a discussion on possible need of updating of definition and classification AH, so that these queries can find an evidence-based answer someday.

Material and Methods

A retrospective analysis of records of all pediatric patients undergoing surgery for congenital inguinal hernia in a single unit from January 2017 to March 2021 was done. Only patients having vermiform appendix as sac content were included in the study. All patients were managed as per institutional protocol. Patient's demographics, clinical presentation, preoperative investigations, peroperative findings, and postoperative outcomes were recorded and analyzed. Written and informed consent were taken from all parents' before submission of images and information about the patients.

Results

A total of 926 children underwent surgery for a congenital inguinal hernia during the study period. AH was found in eight patients. All of them were males. The median age of presentation was 20.5 months (range 2 months to 36 months). The mean duration of symptoms was 2 days (range 2 to 4 days). All patients presented with incarcerated inguinoscrotal swelling (right sided = 5, left sided = 3); associated with pain. The nature of pain was reported to be episodic for three patients, whereas for five patients it was continuous dull aching pain. Fever and anorexia were present in two patients. None of the patients had bilious vomiting, abdominal distension, diarrhea, and abdominal or groin wall inflammation. Two patients had history of upper respiratory tract infection. Blood investigations were within the normal range for four patients, whereas four patients had elevated total leukocyte counts. Radiological investigations comprised abdominal radiographs which showed bowel gas shadow within the ipsilateral hemiscrotum in three patients [Figure 1], and ultrasonography (USG) which was suggestive of bowel loops with preserved vascularity in seven patients, and echogenic material with increased vascularity in one patient. Computerized tomography (CT) was not done preoperatively for any of the patients. All patients were hemodynamically stable and were operated within 24 h of admission. Exploration was done via inguinal incision for all cases. Five patients had appendix alone in the sac; whereas three patients had sliding ileal loops and cecum along with the appendix [Figure 2]a. The neck of the sac was narrow for two patients and wide for six patients. All patients had an entire length of appendix within the sac. Two out of eight patients had inflamed appendix [Figure 2]b. A fibrous band extending from the appendix to the scrotum and attached to the right testis was found in one patient. All patients underwent reduction of contents and a high ligation of sac at a deep inguinal ring. Appendectomy was performed in two patients with inflamed appendix, whereas normal appendix was preserved in six patients. None of the patients underwent repair of the posterior wall of the inguinal canal. Histopathological examination was suggestive of acute appendicitis in both patients who underwent appendectomy. The postoperative course was uneventful for all. Six patients were discharged within 24 h and two patients (who underwent appendectomy) were discharged after 48 hours of surgery. None of the patients had surgical site infection. Radiological evaluation comprising barium meal follow through and contrast-enhanced computed tomography (CECT) abdomen was done for all left-sided AHs during follow-up visits; but none of them had malrotation of gut or situs inversus. None of the six patients in whom the appendix was reduced had secondary appendicitis. No recurrence was detected in any patient over a close follow-up of 1 year (as per institutional protocol).

Figure 1: Abdominal radiograph showing bowel gas in the ipsilateral hemiscrotum

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Figure 2: (a) Intraoperative photograph showing macroscopically normal appendix along with ileal loops in a left-sided Amyand's hernia (b) Intraoperative photograph showing an inflamed appendix (at tip) along with caecum and ileal loops in a right-sided Amyand's hernia

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Discussion

Inguinal herniotomy is the most common procedure performed by a pediatric surgeon, with the reported prevalence of inguinal hernia ranging from 0.8% to 4% in the pediatric population.^[5] AH is the amalgamation of two most common conditions of general surgery, namely, inguinal hernia and appendicitis. Hence, it merits attention. The latest data reveal its prevalence between 0.4% and 0.6%, which might reach 1% in pediatric patients; whereas coexisting appendicitis is reported in 0.1% of cases.^[4] Increased incidence in the pediatric population as compared to the adults can be ascribed to more likelihood of patency of the processus vaginalis.^[6] The incidence of AH in our study was 0.86%. However, incidence of inflammed appendix within the hernial sac was 0.2%. Both of these occurrences are in close agreement with the literature.^[4] Right-sided AH is more common than left-sided AH. This correlates with an increased prevalence of right-sided inguinal hernia and normal anatomical location of the appendix.^[7] The same trend was seen in our study, as right AH (n = 5) was more common than left AH (n = 3). Male preponderance has been reported for this entity, although cases have been reported in females but are generally found in the postmenopausal group.^[8] All of the patients were males in our study group.

With respect to pathophysiology, two points need to ponder upon, first, whether the entrance of the appendix to the inguinal sac is an accidental event or there are some predisposing factors behind it, and second whether the appendicitis is secondary to hernia or just a coincidence.^[4] As far as the first question is concerned, the fibrous connection between the appendix and the right testis has been reported, which in combination with patent processus vaginalis, can guide the passage of the appendix through the inguinal canal.^[4] The occurrence of AH in neonates^[9] and two premature twins,^[6] is a pointer toward a congenital situation. A congenital fibrous band was seen in one of our cases. The long appendix pointing toward groin has also been described as one of the reasons behind the presence of an appendix as a hernia content.^[10] AH on left has been described to be associated with situs inversus, intestinal malrotation, and mobile cecum.^[11] In all three cases in experience of authors, a mobile cecum was the cause, as they were able to rule out situs inversus and intestinal malrotation on the computed tomography (CT) abdomen and barium meal postoperatively. Our findings are in concordance with Gupta et al.,^[12] Cankorkmaz et al.^[13] and Kaymakci et al.^[14] As far as hernial appendicitis is concerned, inguinal muscle contraction can be the reason behind it. This can trigger inflammation and if untreated can cause ischemia, gangrene or perforation of the appendix. Fortunately, the neck of the hernia sac usually contains the inflammation within it, thereby preventing its spread to the abdominal cavity.^[4]

AH is a great clinical mimicker, with a gamut of differential diagnosis being strangulated hernia, epididymo-orchitis, and torsion of the testis; however, appendicitis within the hernial sac is rarely ever considered.^[15] The nature of pain can raise an alarm in favour of AH. However, this holds true for an inflamed appendix where a patient will give a history of initial periumbilical pain followed by right lower abdominal pain. But when present in a pediatric population, eliciting history as far as the nature of pain is concerned, becomes more difficult. In our study population, only three patients gave a history of episodic and crampy pain, whereas for five patients it was continuous and dull aching pain. History of anorexia was present in two patients. Fever and leukocytosis are inconsistent findings^[8] and were found in two and four patients, respectively. None of the patients had signs of bowel obstruction. If not addressed well in time, AH can also result in scrotal fistula, appendiceal perforation with periappendicular or appendicular abcess, intra-abdominal abscess, necrotizing fascitis of the anterior abdominal wall, epididymo-orchitis or testicular abscess, and rarely an in situ arterial thrombosis.^[16] However, no such presentation was seen in our study population.

Radiological investigations can help in arriving at a diagnosis for any pathology, but hernia is a clinical diagnosis. Here, imaging is confined at the most to abdominal radiographs (X-rays) and USG abdomen and inguinoscrotal region. X-rays are of little help as they will show only nonspecific findings. In our series, bowel gas shadow in the ipsilateral hemiscrotum was seen in three cases. USG can be of more help, but radiologists will usually report a hernia with bowel loops as content with or without vascularity.^[15] For seven cases, USG was suggestive of bowel loops as content with preserved vascularity, and for one patient, it was suggestive of echogenic material with increased vascularity. CECT abdomen can be of more help. Vermillion et al. are credited with the first preoperative diagnosis of AH using abdominopelvic CT.^[17] However, it is not advised once a clinical diagnosis of a complicated inguinal hernia is arrived at.^[18] Moreover, preoperative diagnosis is not going to change management in a pediatric patient in any case. Authors believe that the role of CT abdomen or any other specific investigation like barium meal is indispensable postoperatively; and that too in left AH (to rule out malrotation and situs inversus). None of our cases of left AH had any abnormality on the CT abdomen and/or barium meal, thereby inferring mobile cecum as the etiology behind it. This finding of the authors is in agreement with the available literature.^{[12],[13],[14]} CT abdomen can instead aid in picking up other concomitant pathologies.^{[19],[20],[21],[22],[23]}

The standard treatment of any pediatric inguinal hernia is inguinal exploration followed by reduction of contents and high ligation of sac; and exploratory laparotomy in case of a complicated inguinal hernia with generalized peritonitis. Vermillion et al. are credited with the first laparoscopic repair of AH, following a preoperative diagnosis.^[17] However, it is not a common practice in pediatric incarcerated or strangulated inguinal hernias.

Once an AH is encountered, two main concerns are; should incidental appendectomy be performed or not, and should meshplasty be done or not. The domain of pediatric surgery is restricted to the former aspect only. Hence, the authors have confined their discussion to the merits and demerits of appendectomy. Proponents of incidental appendectomy say that it reduces future morbidity and risk of emergency appendectomy.^[24] Old literature also suggests that manipulation of a normal appendix may provoke secondary appendicitis,^[25] hence advocating incidental appendectomy. However, these arguments lack scientific evidence; as no such issues have been reported in the literature; and also in the era of laparoscopic surgery, manipulation of the normal appendix with instruments during diagnostic laparoscopy, is not uncommon. To the best of our knowledge, such complications have not been reported in the literature.^[8] Opponents of incidental appendectomy say that it violates aseptic principles of a clean contaminated wound, leading to unnecessary superficial or deep infection.^[8] Increased dissection might require enlargement of the incision leading to weakening of tissues and hence increased chance of recurrence.^[4] Moreover, the appendix can be used in multiple ways such as Malone, Mitrofanoff urinary diversion, urethral patch, and biliary antireflux procedures.^[26] In experience of the authors, they had normal appendix in six patients; for whom simple reduction followed by the high ligation of sac was done. Appendectomy was performed in two patients who were found have a inflamed appendix intraoperatively. Both of them were discharged after 48 h of surgery. None of our patients had secondary appendicitis, wound sepsis or recurrence over a close follow-up of 1 year. The authors results are in agreement with Cigsar et al.^[26] Keeping this discussion and our experience in mind, authors will not advocate appendectomy for a noninflamed, nonfecolith containing appendix; as they are less likely to come back with secondary appendicitis. This holds truer for our pediatric population with prolonged life expectancy, where the appendix can be put to many uses. The authors would also emphasize that appendix should be removed only when found inflamed or if it is containing fecolith (as these patients are more likely to come back with appendicitis).

With this background, authors would like to emphasize that every AH is an inguinal hernia to begin with and, only an irreducible, incarcerated or strangulated inguinal hernia has an opportunity to earn a title of AH intraoperatively. We never know how many inguinal hernias that we reduced in wards or outpatient departments, actually contained appendix. Hence, term AH should be restricted only to the cases of hernial appendicitis, where appendectomy was a mandate. History further substantiates it as Claudius Amyand himself removed a chronically inflamed appendix contained within the inguinal hernia sac and perforated it by a previously swallowed pin.^[3] Hence, Claudius Amyand himself did not do an incidental appendectomy. Thus, a normal appendix within an inguinal hernia sac should be considered an incidental finding, just like other hernial contents; and term AH should be revised. This also calls for modification of Losanoff's and Basson's classification^[27] [Table 1], where type 1 comprises a normal appendix. Authors hereby propose a modified Losanoff's and Basson's classification [Table 1], where only three types of hernial appendicitis will be there, hence excluding type 1 with a normal appendix. This would streamline the management, as all the entities having inflamed appendix will come under a common umbrella and all cases with normal appendix will be excluded from the study. Hence, decision regarding appendectomy would be more evidence based.

Table 1: Classification system for staging and management for Amyand's Hernia

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Conclusion

AH, if treated well in time, it does not carry any added morbidity or mortality beyond that of a typical inguinal hernia. Many questions like need to do incidental appendectomy still remain unanswered. Novel surgical techniques and research work is coming up with more and more uses of appendix day by day. Hence, avoidance of incidental appendectomy is strongly advised. Updating of definition and classification system can probably offer some solution in this regard. However, more research is warranted regarding standardization of management of this entity. Until then, a high level of cooperation and liasioning between pediatricians, pediatric surgeons, and radiologists are required, so that any interesting finding does not get missed.

Ethics statement

Ethical approval was not taken as it was a retrospective observational study and patients were managed and treated as per standard institutional protocol. Written and informed consent have been taken from parents before submission of images and information about the patient.

Acknowledgments

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

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