Left inguinal appendix in an HIV patient: A case report and review of literature

Abstract

INTRODUCTION

The presence of the appendix in an inguinal hernia sac is rare, with an estimated incidence of 0.51–1% of all inguinal hernias. An inguinal appendix is most commonly referred to as Amyand's hernia.

PRESENTATION OF CASE

A 59-year-old HIV positive male presented to our center with a left painful inguinal mass. The preoperative diagnosis was a left inguinal hernia. Intraoperatively, the sac was found to contain a non inflamed appendix; the appendix was reduced back to the peritoneal cavity and the patient underwent a tension free prosthetic left inguinal hernia repair.

DISCUSSION

Most cases of inguinal appendices are right-sided and are diagnosed intraoperatively; left-sided cases as we encountered are rare and most likely the result of cecal mobility. Preoperative diagnosis of the entity is difficult and most cases are diagnosed intraoperatively. A CT scan is not necessary unless other pressing differentials need to be ruled out. Most authors agree that if the appendix is not inflamed, appendectomy, concurrently with herniorrhaphy, should not be performed to avoid perioperative septic complications.

CONCLUSION

Surgical management of inguinal appendices carries a risk of septic complications. This is especially pertinent to our case, considering the immunocompromised status of our patient. The decisions in the operating room were geared toward limiting septic potential.

1. Introduction

The presence of the appendix in an inguinal hernia sac is rare, with an estimated incidence of 0.51–1% of all inguinal hernias. The incidence of an inflamed appendix is 0.13%.^1–6 The finding of an inguinal appendix is most commonly referred to as Amyand's hernia. The term was coined after Claudius Amyand, sergeant-surgeon to King George II of England, who performed the first recorded successful appendectomy in 1735 that was encountered in a right inguinal hernia sac.⁷ Similarly, in 1886 Robert Hall performed the first recorded appendectomy in the United States. Hall's finding was also an inflamed appendix contained in an inguinal hernia sac. Even less common is herniation of the appendix through the left inguinal canal, since the majority of reported cases describe a right inguinal appendix. Also what is unique to our case is the presentation of the pathology in an HIV positive patient. To our knowledge, only one other case of Amyand's hernia concurrent with HIV has been reported.¹ There is likely no causal relationship between the herniation and HIV status of the patient. However, the HIV status of the patient clearly impacted the intra-operative decisions and post-operative management strategies.

Most cases of Amyand hernias are indirect inguinal hernias⁸ and are usually found in patients above the age of 60 years.⁹ Cases in neonatal and pediatric population have been reported but less frequently so.¹⁰ The majority of the patients are male.¹ Fewer cases are reported in females, and when they do present, the females tend to be post-menopausal.¹¹ Rare locations of appendices are not limited to the inguinal canal; cases of appendices being found in femoral, umbilical and trochar site hernias have also been reported.^12,13

2. Case report

A 59-year-old HIV positive male on antiretroviral therapy (CD4 count of 321 and undetectable viral load), with a history of right inguinal herniorrhaphy, herpetic infection, hypertension, benign prostatic hypertrophy, and newly diagnosed coronary artery disease, was evaluated for a one year history of left groin reducible swelling and pain. The patient denied any obstructive gastrointestinal symptoms. On physical examination, a small reducible left inguinal hernia was noted with no local inflammatory signs and the scrotal sac was empty of peritoneal contents. After cardiac workup and medical optimization, he was admitted through same day surgery for an elective left inguinal hernia repair. Upon exploration of the left groin, an indirect inguinal hernia was identified and the hernia sac was noted to contain the appendix (Fig. 1). The hernia sac was ligated, the contents were reduced back to the peritoneal cavity, and the defect at the internal inguinal ring was repaired using a large size plug, while the floor of the inguinal canal was reinforced using a mesh. The patient tolerated the procedure well and was discharged home on the day of the procedure. His post-operative course was uneventful.

Fig. 1.

Left inguinal incision showing the appendix (A), spermatic cord (B), cecum (C), and conjoint tendon (D).

3. Discussion

The most common clinical presentation of an Amyand hernia as reported by Sharma et al.³ and Kaymakci et al.¹⁴ is a painful inguinosacral or inguinoscrotal mass. Most cases are preoperatively misdiagnosed as a frank incarcerated or strangulated inguinal hernia.⁶ Factors that should prompt the clinician to consider the possibility of an inguinal appendix being more likely than a frank inguinal hernia are the absence of clinical and radiological findings suggestive of intestinal obstruction.^1,15 Some authors suggest that the pain of an incarcerated appendix tends to be crampy and episodic as opposed to dull and constant as is usually seen in strangulated bowel occurring in a frank inguinal hernia.¹⁶ Very rarely the entity presents as prodromal appendicitis.

Considering the anatomy of the appendix, it is understandable why the majority of cases of inguinal appendices are right-sided.¹⁷ Additionally, right inguinal hernias are more common than left inguinal hernias.^17,18 The finding of a left inguinal appendix has been documented in a relatively fewer number of cases and postulated mechanisms of a left-sided occurrence include situs inversus, intestinal malrotation, and cecal mobility.^17,19

The pathophysiology of appendiceal inflammation in Amyand is unlikely to involve obstruction as is with the case of normal appendicitis. Inflammation in Amyand likely involves ischemic events and trauma; the blood supply of an incarcerated, possibly adhesed appendix, is reduced and the abnormal location of the organ accounts for it being more vulnerable to traumatic events.¹⁶ As it relates to the extent of inflammation and infection, a host of resulting pathologies has been documented, ranging from inflammation of the appendix confined to the inguinal sac, to abscess, perforation of the appendix, necrotizing fasciitis, and intraperitoneal involvement.^{2,11,16,19–21} Strangulation of the appendix has also been documented.²²

Preoperative diagnosis is difficult, and since it is commonly misdiagnosed as a frank inguinal hernia, further diagnostic work up once the diagnosis of frank inguinal hernia is presumed is not routine.¹⁵ The majority of cases are diagnosed intraoperatively. When Amyand is suspected, ultrasound and CT scan can assist.²³ A few authors elaborate on the accuracy of a CT scan in detecting Amyand.^14,24 Even with a CT scan however, preoperative determination are often difficult as reported by Kueper et al. Based on the inconsistencies in reports of a CT scan making an accurate preoperative diagnosis,²² we believe that the routine practice for approach of a frank inguinal hernia should be adhered to, especially considering that often times the result of a CT scan does not significantly alter the course of management. In the majority, if not all of the instances, a surgical intervention is necessitated regardless of CT findings.²⁵ A CT scan can be considered if other pressing differentials need to be ruled out.

Losanoff et al. proposed the following classification based on the extent of inflammation; type 1: having no inflammation, type 2: inflammation limited to the sac, and type 3: with the disease extending beyond the sac to surrounding tissue, abdominal wall and intraperitoneal space.⁴ The 4th type was proposed to include compounding intra-abdominal pathology such as masses, which may have a causal relationship with the extent of incarceration.

Ioannidis et al. encountered a case of a strangulated appendix in an inguinal sac with no inflammation and proposed that the Losanoff's classification be extended to a 5th type to include such findings. Strangulation results in a compromised blood supply that negatively affects the integrity of appendiceal tissue. This allows for bacterial translocation.²² It is likely that strangulation is the beginning in a pathophysiological spectrum of an alternative inflammation mechanism. Considering this, we believe that such a case of strangulated appendix is clinically indistinct from an inflamed appendix and should be treated as such. Hence we propose that a strangulated appendix in an inguinal sac with no inflammation or infection beyond the sac should be included in Losanoff's type 2 criteria.

Authors generally agree that if the appendix is not inflamed as in Losanoff's type 1, appendectomy should not be performed because of the risk of peritoneal contamination.⁶ In the middle aged and the elderly population, the risk of future appendicitis is minimal when compared to younger patients and hence prophylactic appendectomy is not warranted. A counter argument points to the potential of a left sided appendix resulting in future hassles associated with an atypical appendicitis presentation.¹⁸

If the appendix is inflamed as in Losanoff's types 2, 3 and usually in 4, appendectomy is the treatment of choice. Appendectomy would normally be performed through the herniotomy. In cases where pathology extends beyond the sac, as in Losanoff's type 3 and 4, inferior midline laparotomy allows for better exposure, control and exploration. In the event of extensive infection to surrounding tissue with resulting necrotizing fasciitis, debridement will also be necessary.

Prosthetic herniorrhaphy is recommended in the setting of no inflammation and infection especially considering the longevity advantage. In the setting of inflammation and or infection as in Losanoff's types 2, 3, and 4 some authors feel that there is a septic potential with the use of mesh as compared to endogenous repair by the recommended Bassini's or Shouldice's method.⁶ Limited data suggests that appendectomy and herniorrhaphy can be managed laparoscopically.

Our patient presented clinically similar to the majority of cases of inguinal appendices reported in the literature and met the criteria for Losanoff's type 1 Amyand; a painful inguinal mass, with no history of nausea, vomiting or abdominal pain suggestive of appendicitis, nor were there clinical symptoms or radiological findings suggestive of obstruction. Preoperatively the diagnosis of frank inguinal hernia was presumed and hence no further diagnostic work up was pursued as is routine practice for an inguinal hernia. The diagnosis of an inguinal hernia was made intraoperatively. The rare left-sided occurrence of the entity found in our patient was most likely a result of a mobile cecum; in retrospect, a remote CT scan of the abdomen and pelvis on the patient had failed to reveal situs inversus or intestinal malrotation. Also unique to our case was the presentation in an HIV patient. Considering the various schools of thought pertaining to whether or not an appendectomy should be performed in a left normal appendix, what superseded all opinions in our case was the fact that our patient was immunocompromised. In our case, simple appendiceal reduction was opted to limit septic potential. The patient's HIV status was not advanced as reflected by his undetectable viral load, but had he been in a more advanced HIV stage, his status could have further affected his course including possible presentation with more advanced disease with a bearing on the chosen route of management.

4. Conclusion

A left-sided inguinal appendix is usually the result of cecal mobility. It is much less common than a right-sided inguinal appendix. Preoperative diagnosis of the entity is difficult and most cases are diagnosed intraoperatively. A CT scan is not necessary unless other pressing differentials need to be ruled out. Most authors agree that if the appendix is not inflamed, appendectomy should not be performed to avoid perioperative septic complications. This is especially applicable to patients with an immunocompromised status. In light of the fact that a frank strangulation of an appendix in an inguinal sac and Losanoff's type 2 Amyand which describes an inflamed appendix, are clinically similar, and the fact that the same management strategy is implicated, we propose that the Losanoff's type 2 criteria be extended to include appendiceal strangulation.

Conflict of interest statement

None.

Funding

None.

Ethical approval

Written informed consent was obtained from the patient for publication of this case report and accompanying image. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.

Author contributions

• Noubar Kevorkian: Data collection

• Chad Rennie: Writing

• Armand Asarian: Supervisor

• Peter Pappas: Review