Amyand's hernia (AH) is an inguinal hernia containing the appendix. This condition is rare in children, especially in infants. Claudius Amyand, in 1735, performed appendectomy while doing herniotomy in an 11-year-old male child; and hence his name. Preoperative diagnosis is rare and management involves herniotomy and appendicectomy if indicated.

We are reporting two cases of AH, one in a four-year-old presented with partially reducible hernia; and another in a neonate who presented with acute epididymo-orchitis like picture.

On opening the hernia sac, appendix was revealed. Herniotomy and appendectomy was done.

Case 1: A four-year-old boy presented with inguinoscrotal swelling since one year. Pain was felt on and off recently with increase in its size since one week. Both testes were palpable separately. The swelling was partially reducible. Systemic examination was normal. Patient was electively posted for right herniotomy and on opening the sac, long tubular minimally inflamed appendix with hydrocele fluid was noted. Patient underwent appendectomy, herniotomy and Jaboulay's eversion of distal sac. Patient is doing well with two and a half years of follow-up.

Case 2: A 20-day, weighing 3.0 kg male neonats was brought to us with history of right scrotal swelling, excessive crying and fever of one day duration. His antenatal scans were normal and he was delivered at term by vaginal route. Clinical examination revealed reddish tender swelling of right scrotum with left scrotum with testis was normal. Abdominal examination was normal. The scrotal Doppler ultrasound revealed features of acute epididymo-orchitis with normal testicular blood flow and associated secondary hydrocele (Figure 1A-B). Hence the baby was managed expectantly with antibiotics and analgesics; complete resolution of the acute scrotum was noticed during follow up visit.

Two weeks later in the follow-up period the baby was brought with small painless, reducible inguinal swelling; clinical diagnosis of hernia was made and herniotomy was done. Intra-operatively appendix was noted in the hernia sac, its tip reaching the scrotum and had tell-tale evidence of past appendicular inflammation. Baby underwent appendectomy with herniotomy (Figure 2A-B) had uneventful postoperative period and doing well at first month follow-up visit.

Amyand's hernia is an extremely rare condition. The rate of incidence is 0.1% of hernitomies. AH in less than one year old represent 2% of the total cases of appendicitis [1]. The pathophysiology of AH and its relationship with appendicitis are unknown [2] [3]. AH, is as rule, occurs in the right inguinal region. If left sided, it may be due to mobile cecum, malrotation, or situs inversus. The presence of a non-inflamed appendix in the inguinal hernia is three times more common in the children than in adults. This is due to persistent patency of the processus vaginalis in infancy and adulthood [2]. A congenital band extending from the appendix into the scrotum and attached to the right testis, and the funnel shaped tapering of the cecum in the neonate are two more possible pathogenetic factors [2].

Clinical presentation of an AH may be that of a normal, obstructed, or strangulated inguinal hernia, irrespective of whether the appendix is inflamed or not. Acute scrotum and abdominal pain are possible clinical presentations. Testicular torsion, especially when there is an undescended testis; inguinal lymphadenitis; epididymo orchitis and hydrocele of the spermatic cord are conditions that an AH may mimic.

The confirmatory diagnosis of an AH is made during surgical exploration of the groin, although radiological investigations like ultrasound, computed tomography will be helpful in few cases [1].

Even though appendectomy can be performed through an inguinal incision or with laparoscopy, many authors prefer to save the appendix when it is not inflamed, because of the importance of the lymphoid tissue in it, risk of wound infection and for its possible later use in urinary diversion, biliary tract reconstruction, and for antegrade bowel enemas [1]. Both of our patients had features of appendicitis and in the neonate the Amyand's hernia with acute appendicitis presented as acute scrotum masquerading as epididymo-orchitis. Hence appendicectomy was done in both the patients and they had an uneventful postoperative course.

Neonatal appendicitis is extremely rare (0.1% of appendicitis cases in infancy, which constitutes 2% of pediatric appendicitis). Premature neonates accounts 50% of these cases, and only in one third of these, inflamed appendix lies within a hernia. This low frequency rate and the fact that only 20 of neonatal cases of AH have been reported in English publications render the cases of perforated appendix in premature neonates extremely rare [2].

Classification of AHs, after Losanoff and Bason, modified by Rikki as Rikkki's classification of Ahs described in Table 1. Both of our cases belong to type 2 [4].

Literature suggests that majority will present in neonate, as obstructed or strangulated inguinal hernia and even with perforation with generalized peritonitis, systemic signs and symptoms of appendicitis are rarely evident. Our case presented like epididymo-orchitis like picture i.e. acute appendicitis [1] [2][3].

Antonios Panagidis reported neonatal perforated AH presenting as an enterocutaneous scrotal fistula [2]. Kumar et al. reported neonatal pyoscrotum and perforated appendicitis in 26 day old male baby with undescended testis, who was initially diagnosed to have testicular torsion [5]. Luchman et al. reported a case of scrotal abscess following perforated appendicitis in six day old male neonate [6].

Amyand's hernia (AH) is very rare and appendicitis in AH in neonates is extremely rare. Preoperative diagnosis is difficult unless presenting with appendicitis or its related complications. Herniotomy and appendicectomy, when indicated are recommended.

1. Gupta S, Sharma R, Kaushik R. Left-sided Amyand's hernia. Singapore Med J 2005 Aug;46(8):424–5.   [Pubmed]    
2. Panagidis A, Sinopidis X, Zachos K, et al. Neonatal perforated Amyand's hernia presenting as an enterocutaneous scrotal fistula. Asian J Surg 2014 Apr 18.   [CrossRef]   [Pubmed]    
3. Pandey V,Gangopadhyay AN, Gupta DK, Sharma SP. Gangrenous Amyand's hernia in neonate: A great clinical masquerader. Open Jornal of Pediatrics 2013;3:364–365.   [CrossRef]    
4. Murugesan MS, Rajasekaran MS. Amyand Hernia. Journal of Dental and Medical Sciences (IOSR-JDMS) 2013; 6:20–4.    
5. Kumar R, Mahajan JK, Rao KL. Perforated appendix in hernial sac mimicking torsion of undescended testis in a neonate. J Pediatr Surg 2008 Apr;43(4):e9–10.   [CrossRef]   [Pubmed]    
6. Iuchtman M, Kirshon M, Feldman M. Neonatal pyoscrotum and perforated appendicitis. J Perinatol 1999 Oct-Nov;19(7):536–7.   [CrossRef]   [Pubmed]