Neonatal Appendicitis and Incarcerated Inguinal Hernia: Case Report and Review of the Literature.

Neonatal appendicitis carries a high mortality rate. We describe a peculiar case presented after an incarcerated hernia manually reduced in a 4-week-old male neonate with ipsilateral undescended testis. Laparoscopy allowed a prompt recognition of an unexpected intra-abdominal life-threatening condition. Pathogenesis, treatment, and outcome are discussed on the light of a comprehensive literature review.

INTRODUCTION

Neonatal appendicitis is a rare condition with a mortality rate of 25%–75%.[12] We present a case of an incarcerated hernia in a 4-week-old male neonate with an associated ipsilateral undescended testis. Following manual reduction of the hernia, a laparoscopic approach to the hernia 36 h later revealed a necrotic appendix. Appendicectomy was completed by an open approach as well as the inguinal hernia repair and orchidopexy. The pathogenesis of this finding and the possible approaches to its management are discussed.

CASE REPORT

A full term, 29-day-old male neonate presented at the accident and emergency department of our institution with a 3-h history of a right-sided inguinal swelling.

The diagnosis of a right undescended testis had been made 4 days previously at the time of a neonatal circumcision and tongue-tie release. The right groin swelling was tender and the testis palpable separately from it. A diagnosis of irreducible inguinal hernia was made, and a difficult manual reduction obtained facilitated by administration of oral analgesia. The patient was admitted for observation with a plan for delayed surgical repair of the hernia 24–36 h later to allow the groin edema to resolve. During the admission, the patient showed no overt signs of distress. The abdomen remained soft and nondistended. Oral feeding was only slightly less vigorous than usual. On two occasions, however, he passed stools with some traces of blood. An open inguinal herniotomy was initially considered with simultaneous orchidopexy if possible. A laparoscopic approach to the hernia was preferred to enable concurrent inspection of the bowel for signs of injury or ischemia.

The patient was taken to theater 36 h after admission. Initial laparoscopy revealed dilated bowel loops with limited working space. In the right iliac fossa, an inflammatory fibrin lined mass was tenaciously adherent to the right deep inguinal ring. Mobilization of this mass revealed an inflamed cecum and a necrotic appendix. The terminal ileum was bruised and thickened. The right inguinal ring was markedly edematous so as to appear almost closed. The vas deferens and spermatic vessels were difficult to identify due to the overlying inflamed peritoneum.

The laparoscopic procedure was abandoned, and a laparotomy performed throughout a high inguinal incision. After mobilization of the cecum, a routine appendicectomy was performed. Through the same incision, an inguinal canal approach allowed the herniotomy and orchidopexy to be completed.

The postoperative course was uneventful, and the patient went home on day 3. The histopathology examination confirmed a necrotic appendix.

At 6-month follow-up, the patient was well and no signs of hernia recurrence. The right testicle was in a good position in the scrotum with the same dimensions as the contralateral.

DISCUSSION

Neonatal appendicitis is a rare condition associated with comorbidities such as prematurity, obstruction (Hirschsprung's, Amyand's hernia, meconium plug syndrome, and cystic fibrosis), cardiac, and respiratory disease.[1] The mortality rate is reported at 25%–75% even without comorbidities.[2] Inflamed appendix within the hernia sac (Amyand's hernia) accounts for one-third of the neonatal appendicitis described in the literature. Its presentation ranges from irritability associated with an irreducible hernia to systemic signs of sepsis. Anatomical characteristics could predispose neonates and infants to an incarcerated appendix.[3] However, the correlation with the incarceration is inconsistent.[456]

In our case, the patient had no overt signs of abdominal pathology except of traces of blood in the stool. The timing suggests a role of the incarceration in the pathogenesis. The presence of ecchymosis and edema on the terminal ileum demonstrated the possible herniation of this bowel segment along with the appendix. Appendicitis within the hernia sac, after attempt of manual reduction, has been reported in infants.[7] The peculiarity of our case is that the appendix was completely reduced in the abdomen but developed a severe grade of inflammation. The coexisting cryptorchidism, and consequently the short processus vaginalis, might have prompted the reduction of appendix back to the abdomen. However, the presence of ecchymosis on the terminal ileum confirmed that the incarceration, possibly associated with the forceful manual reduction, produced severe congestion on the tissues. Ischemia has been advocated as the principal factor in the pathogenesis of appendicitis in neonates.[8]

In our case, a typical necrotic appendicitis was found at pathology examination without strong evidence of ischemic mechanism.

Prematurity is one of the most frequent comorbidities for neonatal appendicitis, being premature patients prevalent even in the cohort of neonatal Amyand's hernia.[9] This suggests that the mechanical aspect is not the only causal factor.

Few publications report on the role of laparoscopy in the diagnosis of neonatal appendicitis and laparoscopic treatment is only anecdotal [Table 1].

Table 1

Literature review of neonatal inguinal hernias associated with appendicitis

The initial laparoscopic approach to correct the hernia had been chosen to preserve the inguinal canal for a subsequent orchidopexy. The risk of testicular atrophy following an orchidopexy simultaneously done to herniotomy for incarcerated hernia is significantly higher than an elective procedure, in particular in patients <6 months of age.[10] In our case, the laparoscopic approach to the hernia allowed recognition of a life-threatening condition that was otherwise unsuspected. The outcome of neonatal appendicitis associated to inguinal signs is generally favorable [Table 1]. However, in our case, the hernia was completely reduced at the surgery, and a primary inguinal approach would have missed the abdominal condition. The hernia sac isolation through the groin was easier than expected, and thus we decided to combine the correction of the undescended testis at the same time.

On the base of this experience, we strongly suggest laparoscopic exploration for an inguinal hernia that has been previously incarcerated in neonates and infants, in particular in those cases, which have had a difficult manual reduction. In our case, the inflammatory abdominal disease did not affect the results of the inguinal hernia repair and the viability of the testicle in the midterm follow-up.

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