Amyand's Hernia, Appendicitis, and Undescended Testis: The Amyand's Triad

A 35-day-old male infant presented with a sudden onset tender right inguinal swelling of 2-day duration [ Figure 1 ]. The infant also had abdominal distension and vomiting. He was born as a term neonate and was diagnosed with a palpable undescended testis at birth. The parents were counseled to review at 6 months of age. The clinical suspicion included torsion or an irreducible inguinal hernia. X-ray abdomen showed dilated small bowel loops confirming an obstructed inguinal hernia. The infant was taken up for urgent inguinal exploration after resuscitation. Irreducible right inguinal swelling During surgery, the child was found to have a sliding inguinal hernia with caecum and appendix as a part of the wall of the sac and small bowel as content. The appendix was inflamed and gangrenous [ Figure 2 ]. The right testis was also gangrenous. The contents were reduced, and an appendicectomy was performed. The testis remained gangrenous and necrotic after warm packs and 100% oxygen and hence was removed after proper consent. Herniotomy was completed after carefully ligating the sac and the deep ring itself was closed. Postoperatively, the infant recovered well except for wound infection which was treated with appropriate antibiotics. Inflamed appendix with gangrenous testis

Amyand's hernia was named after Claudius Amyand, who performed the first appendicectomy in an 11-year-old boy with an inguinal hernia with inflamed appendix as content.[ 1 ] Preoperative diagnosis is seldom made as it is a surprise intraoperative finding in most cases. An undescended testis presenting with an irreducible hernia may be an expected complication. Encountering an Amyand's hernia with undescended testis is very rare. Moreover, this triad of association has been reported only once in literature.[ 7 ]

Amyand's hernia has been described by various authors since first documented in 1735. Nyhus,[ 2 ] Losanoff and Basson[ 3 ] [ Table 1 ] and Bendavid have classified Amyand's hernia in adults into numerous subtypes and have suggested treatment options for various clinical presentations with the main emphasis on hernioplasty. In 2015, Raveenthiran V had proposed a modified pediatric Amyand's hernia classification[ 4 ] [ Table 1 ] reiterating that Neonatal Amyand's hernia with inflamed appendix as a separate entity. Classification of Amyand's hernias, after Losanoff and Basson and Athena's modification The incidence of an inguinal hernia with appendix as content in children is typically quoted as 0.07%–0.28%[ 5 ] much less than the reported 1% incidence in adults. The incidence of finding an inflamed complicated appendicitis is also reported at 0.08%.[ 5 ] Appendix has been reported as content in almost all forms of hernia from the more common inguinal and umbilical hernia, to the rarer spigelian and obturator hernia.[ 6 ] Management of an Amyand's hernia with an inflamed appendix is usually appendicectomy with hernia repair. Rarely, a combined laparotomy may be needed for patients with abdominal signs. Recurrence of hernia is also not uncommon and has been reported in a few cases after Amyand's hernia repair with inflamed appendix in infants. For patients with noninflamed appendix, the decision on the need for appendicectomy is left to the discretion of the treating pediatric surgeon. A free lying non-inflamed appendix can be conveniently left undisturbed and reduced. However, if the appendix is adherent to the sac or has a fecalith it may have to be removed. The previously reported case was an Amyand's hernia with appendicitis and undescended testis in a 26-day-old neonate[ 7 ] who had an appendicectomy and orchidopexy. Our case is unique, as the inflamed appendix was found as a part of a sliding inguinal hernia and also had caused gangrene of the testis, requiring an orchiectomy. We would like to name this rare triple association as “The Amyand's triad” with the components being Amyand's hernia, appendicitis, and undescended testis. The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed. Nil. There are no conflicts of interest.