INTRODUCTION
Amyand’s hernia is named after Claudius Amyand, who described it in 1735 for the first time. It is used to denote presence of appendix in hernial sac. It may be present in any age group, mostly present in adults, have male predominance, and usually right-sided. The herniated appendix may or may not be inflamed and decided for appendectomy accordingly.
CASE DETAILS
Case 1
A 2-year-old male child presented with large inguinal hernia on right side. The swelling was present for last one year and used to appear on crying or straining and disappeared on lying down. It was gradually increasing in size. There was no history of recurrent cough or constipation. There was no history of any irreducibility. Per-abdomen examination was within normal limits, with normal genitalia and bilateral descended testis. He was planned for elective surgery after pre-anesthetic fitness. Blood investigations were within normal limits. On exploration of right inguinal canal, large hernia sac with cecum and appendix as content was found [Figure 1]. The contents were reduced and right herniotomy was done. Appendix was healthy and not removed. The child has no recurrence of hernia at follow-up period of 2 years.
Figure 1: Intra-operative picture of right inguinal hernia with appendix and cecum as contents
Case 2
A 7-month-old male baby presented with left inguinal large irreducible hernia for 1 day. Parents gave history of left inguinal swelling since 2 months of age and was increasing in size. There was history of swelling being irreducible once in past and was reduced at a local hospital nearby. There were no signs of obstruction at presentation to us, but hernia was irreducible. Blood investigations were normal except low Na+ levels (129 meq/L). After initial resuscitation, baby was taken for emergency operation. On left inguinal exploration, cecum with appendix were contents [Figure 2], which were gently reduced and left herniotomy done, without appendectomy. Baby has no recurrence at follow-up period of 2.5 years.
Figure 2: Intra-operative picture of left inguinal hernia with appendix and cecum as contents
Case 3
An 1-month-old neonate presented with bladder outlet obstruction. On physical examination, neonate had bilateral palpable kidneys and right inguinal hernia. Micturating cystourethrography showed bilateral grade IV/V reflux. Laboratory investigations showed normal hemoglobin, total leucocyte count and platelets, blood urea was 83 mg/dl, and creatinine was 1.4 mg/dl. Baby was planned for endoscopy and right herniotomy. At inguinal exploration, long appendix was found as hernia content [Figure 3]. It was reduced without appendectomy and herniotomy being done. Baby has no recurrence of hernia at 1-year follow-up and currently being managed with chronic kidney disease.
Figure 3: Intra-operative picture of right inguinal hernia with appendix as content
DISCUSSION
Inguinal hernia is one of the most common surgical condition. Amyand’s hernia is rare in pediatric age group, especially in neonates and infants.[1] It accounts for 0.4% to 1% of inguinal hernias.[2] It has male predominance and has been reported in patients aged 3 weeks to 92 years.[2–6] It is usually seen on right side.[7] It may be in relation to normal location of appendix in right iliac fossa and also right inguinal hernias being more common as compared with left inguinal hernias. Rarely, it can be seen on left side in situs inversus, long appendix mesentry, highly mobile cecum, or malrotation cases.[2,6]
The appendix in hernia sac may be inflamed and appendicitis associated with Amyand’s hernia accounts for 0.1% of all appendicitis cases.[3] It may have been caused by extra-luminal obstruction or local trauma. Short or narrow neck of hernia sac may cause hampered blood supply and inflammation.[7] Losanoff et al.[8] classified these into 4 types in adults depending of degree of inflammation and sepsis. Although, it does not seem to have implications in children. The differential diagnosis on physical examination may include strangulated hernia/omentocele, testicular tumor, acute hydrocele, inguinal adenitis, etc., The presentation is usually similar to any inguinal hernia. There might be erythema or tenderness, due to incarceration of contents. In majority, the diagnosis is per-operative. The pre-operative diagnosis is difficult as it is a rare entity and presentation is similar to that of any inguinal hernia. Ultrasound can reliably differentiate it from other inguinoscrotal swellings.
The treatment for Amyand’s hernia depends on inflammation status of appendix. Literature still debates removal of appendix in all cases. The appendix should be preserved as far as possible, if there are no signs of appendicitis or appendicolith. If appendix is normal, it can be repositioned and herniotomy done in children. Johari et al.[9] recommended prophylactic appendectomy irrespective of inflammation, especially in left-sided Amyand’s hernia to avoid possible confusion of diagnosis in future. We did not do appendectomy in our case as there were no features of appendicitis nor features suggestive of malrotation in past. To best of our knowledge, less than 20 cases of left-sided Amyand’s hernia have been reported in literature so far.[10,11]
Cigsar EB et al.,[6] in their 11 years of review, found that all cases of Amyand’s hernia were male, with a mean age of 16.7 months (15 days to 8 years). It was right-sided and incidental finding in 80.4% cases each. None had recurrence. Sharma et al.[4] in their study of 18 cases over 15 years found that the mortality may be upto 30% especially in sepsis. Manatakis DK et al.[12] did a 20-year systematic review on Amyand’s hernia and found that 91% of cases were in males and a left-sided Amyand’s hernia was observed in 9.5%. Among acute cases, mortality was 1.8%. All of our presented cases were male, with one left-sided AH and none had mortality or recurrence. None of our patient had sepsis and all were doing good in follow-up.
CONCLUSION
The pediatric surgeon should be aware of inadvertent finding of appendix in hernia sac, especially on left side.
Declaration of patient consent
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 initial s will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
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