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Neonatal appendicitis with perforation: A case report
J Ped Surg Case Reports 2 (2014) 353e354
Contents lists available at ScienceDirect
Journal of Pediatric Surgery CASE REPORTS journal homepage: www.jpscasereports.com
Neonatal appendicitis with perforation: A case reportq Mohamed Amin El-Gohary a, *, Salam Al jubouri b a b
Department of Surgery, Division of Paediatric Surgery, Burjeel Hospital, P.O. Box 7400, Abu Dhabi, U.A.E Al Noor Hospital, Abu Dhabi, UAE
a r t i c l e i n f o
a b s t r a c t
Article history: Received 6 July 2014 Accepted 16 July 2014
Acute appendicitis is a rare condition in neonates, with a high mortality. We report a preterm baby who presented with clinical picture of necrotizing enterocolitis, and found at laparotomy to have perforated appendicitis. Ó 2014 The Authors. Published by Elsevier Inc. All rights reserved.
Key words: Neonatal appendicitis Neonate Necrotizing enterocolitis Peritonitis
Acute neonatal appendicitis is a rare condition associated with significant morbidity and mortality. It tends to occur in premature infants, with increased perforation rate and rapid progression to peritonitis. The rarity of neonatal appendicitis together with lack of specific signs and low index of suspicion has led to delay in diagnosis and surgical intervention. It most instances the diagnosis is made during laparotomy for cases of neonatal intestinal perforation. 1. Case report Preterm baby boy born at 33 weeks of gestation by Cesarean section (CS), to a 26 years old primigravida mother. He was admitted to neonatal intensive care on day of life 2 with acute respiratory distress syndrome. On the 4th hospital day the baby was noted to have repeated bilious vomiting and mild abdominal distension. The abdomen was soft non tender with no evidence of erythema or edema. No occult blood was found in the stool. Abdominal x-ray showed gas under the diaphragm with suspicious areas of pneumo-intestinalis suggestive of perforated necrotizing enterocolitis (NEC). Sepsis screening yielded gram positive cocci for which he was started on ampicillin and vancomycin. The baby was on nasogastric suction and started on total parenteral nutrition (TPN). The baby remained clinically stable and passing regular
q This is an open access article under the CC BY-NC-SA license (http:// creativecommons.org/licenses/by-nc-sa/3.0/). * Corresponding author. Tel.: þ971 506225532. E-mail address: [email protected] (M.A. El-Gohary).
stools. A repeated abdominal x-ray done 24 h later showed significant increase in abdominal free air (see Fig. 1). A decision was made to take the patient immediately to the surgical theater for exploratory laparotomy. Laparotomy was performed via transverse supra umbilical incision. Intraoperative findings was significant for substantial amount of greenish stool inside abdominal the cavity, the source of which was not obvious at initial inspection. The whole bowel was healthy except for the right lower quadrant in which the bowel was stuck to abdominal wall with fibrinous adhesions. When adhesions was released, there was a 1 cm perforation near the tip of a retro caecal appendix (Fig 1). A formal appendectomy was performed with the baby subsequently having a smooth and uneventful postoperative recovery. Follow up at 6 months of age revealed a normal thriving baby. Histopathology confirmed gangrenous nature of the removed appendix and with no evidence of Hirchsprung’s disease. 2. Discussion Acute appendicitis is a rare cause of acute abdominal sepsis in neonates and hardly considered in the differential diagnoses. The incidence of neonatal appendicitis has been reported as 0.04e0.2% [1]. It affects males in approximately 75% of the time, and 25%e50% of all reported cases involve premature infants [2e7]. Less than 50 cases have been reported over the last 30 years [2], with mortality rate ranging between 20% and 25% [1,4,6]. The low incidence of neonatal appendicitis may be attributed to the funnel shaped appendix with wide opening into the cecum,
2213-5766/$ e see front matter Ó 2014 The Authors. Published by Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.epsc.2014.07.008
354
M.A. El-Gohary, S. Al jubouri / J Ped Surg Case Reports 2 (2014) 353e354
generally be expected to be made only at laparotomy or autopsy. Hirschsprung’s disease (HD), and isolated form of NEC limited to the appendix should always be ruled out [8,9]. In absence of suspicion of Hirschsprung’s disease, simple appendectomy with peritoneal lavage with warm saline is the preferred line of management. 3. Conclusion Acute appendicitis is a rare condition in neonates, with a high mortality rate. The rarity of neonatal appendicitis together with a lack of specific signs and low index of suspicion has led to delay in diagnosis and high mortality. References
Fig. 1. Showing perforated appendicitis.
liquid diet, the lack of fecaliths, and the presumed relative infrequency of lymphatic hyperplasia in the periappendiceal region [6]. The high morbidity and mortality associated with appendiceal perforation is attributed to the thin appendiceal wall and an indistensible cecum, facilitated by the small undeveloped and functionally non-existent omentum, small size of the peritoneal cavity allowing a more rapid and diffuse contamination and little physiological reserve [3]. Because of the rarity of the pathology and atypical signs and symptoms, the diagnosis of perforated appendicitis would
[1] Stifle D, Stallmach T, Sachet P. Acute appendicitis in neonates: complication or morgues suit generis? Pediatr Surg Int 1998;14:122e3. [2] Drapala B, Trognon B, Canarelli JP, Tamboura Tientcheu A, Bensatti L, Razarimanantsoa L, et al. Appendicite neonatal. Arch Pediatr 2000;7:896e7. [3] Arora NK, Deorari AK, Bhatnagar V, Mitra DK, Singhal PK, Singh M, et al. Neonatal appendicitis: a rare cause of surgical emergency in preterm babies. Indian Pediatr 1991;28:1330e3. [4] Deguchi E, Iwai N, Yanagihara J, Nakamura K, Doi Y. Case report: neonatal appendicitis with perforation in a premature infant. Pediatr Surg Int 1990;5: 366e8. [5] Karaman A, Cavusoglu YH, Karaman I, Cakmak O. Seven cases of neonatal appendicitis with a review of the English language literature of the last century. Pediatr Surg Int 2003;19:707e9. [6] Ruff ME, Southgate WM, Wood BP. Neonatal appendicitis with perforation. Am J Dis Child 1991;145:111e2. [7] Shaul WL. Clues to the early diagnosis of neonatal appendicitis. J Pediatr 1981; 98:473e6. [8] Kwong MS, Dinner M. Neonatal appendiceal perforation and Hirschsprung’s disease. J Pediatr 1980;96:917e8. [9] Arliss J, Holgersen LO. Neonatal appendiceal perforation and Hirschsprung’s disease. J Pediatr Surg 1990;25:694e5.
Contents lists available at ScienceDirect
Journal of Pediatric Surgery CASE REPORTS journal homepage: www.jpscasereports.com
Neonatal appendicitis with perforation: A case reportq Mohamed Amin El-Gohary a, *, Salam Al jubouri b a b
Department of Surgery, Division of Paediatric Surgery, Burjeel Hospital, P.O. Box 7400, Abu Dhabi, U.A.E Al Noor Hospital, Abu Dhabi, UAE
a r t i c l e i n f o
a b s t r a c t
Article history: Received 6 July 2014 Accepted 16 July 2014
Acute appendicitis is a rare condition in neonates, with a high mortality. We report a preterm baby who presented with clinical picture of necrotizing enterocolitis, and found at laparotomy to have perforated appendicitis. Ó 2014 The Authors. Published by Elsevier Inc. All rights reserved.
Key words: Neonatal appendicitis Neonate Necrotizing enterocolitis Peritonitis
Acute neonatal appendicitis is a rare condition associated with significant morbidity and mortality. It tends to occur in premature infants, with increased perforation rate and rapid progression to peritonitis. The rarity of neonatal appendicitis together with lack of specific signs and low index of suspicion has led to delay in diagnosis and surgical intervention. It most instances the diagnosis is made during laparotomy for cases of neonatal intestinal perforation. 1. Case report Preterm baby boy born at 33 weeks of gestation by Cesarean section (CS), to a 26 years old primigravida mother. He was admitted to neonatal intensive care on day of life 2 with acute respiratory distress syndrome. On the 4th hospital day the baby was noted to have repeated bilious vomiting and mild abdominal distension. The abdomen was soft non tender with no evidence of erythema or edema. No occult blood was found in the stool. Abdominal x-ray showed gas under the diaphragm with suspicious areas of pneumo-intestinalis suggestive of perforated necrotizing enterocolitis (NEC). Sepsis screening yielded gram positive cocci for which he was started on ampicillin and vancomycin. The baby was on nasogastric suction and started on total parenteral nutrition (TPN). The baby remained clinically stable and passing regular
q This is an open access article under the CC BY-NC-SA license (http:// creativecommons.org/licenses/by-nc-sa/3.0/). * Corresponding author. Tel.: þ971 506225532. E-mail address: [email protected] (M.A. El-Gohary).
stools. A repeated abdominal x-ray done 24 h later showed significant increase in abdominal free air (see Fig. 1). A decision was made to take the patient immediately to the surgical theater for exploratory laparotomy. Laparotomy was performed via transverse supra umbilical incision. Intraoperative findings was significant for substantial amount of greenish stool inside abdominal the cavity, the source of which was not obvious at initial inspection. The whole bowel was healthy except for the right lower quadrant in which the bowel was stuck to abdominal wall with fibrinous adhesions. When adhesions was released, there was a 1 cm perforation near the tip of a retro caecal appendix (Fig 1). A formal appendectomy was performed with the baby subsequently having a smooth and uneventful postoperative recovery. Follow up at 6 months of age revealed a normal thriving baby. Histopathology confirmed gangrenous nature of the removed appendix and with no evidence of Hirchsprung’s disease. 2. Discussion Acute appendicitis is a rare cause of acute abdominal sepsis in neonates and hardly considered in the differential diagnoses. The incidence of neonatal appendicitis has been reported as 0.04e0.2% [1]. It affects males in approximately 75% of the time, and 25%e50% of all reported cases involve premature infants [2e7]. Less than 50 cases have been reported over the last 30 years [2], with mortality rate ranging between 20% and 25% [1,4,6]. The low incidence of neonatal appendicitis may be attributed to the funnel shaped appendix with wide opening into the cecum,
2213-5766/$ e see front matter Ó 2014 The Authors. Published by Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.epsc.2014.07.008
354
M.A. El-Gohary, S. Al jubouri / J Ped Surg Case Reports 2 (2014) 353e354
generally be expected to be made only at laparotomy or autopsy. Hirschsprung’s disease (HD), and isolated form of NEC limited to the appendix should always be ruled out [8,9]. In absence of suspicion of Hirschsprung’s disease, simple appendectomy with peritoneal lavage with warm saline is the preferred line of management. 3. Conclusion Acute appendicitis is a rare condition in neonates, with a high mortality rate. The rarity of neonatal appendicitis together with a lack of specific signs and low index of suspicion has led to delay in diagnosis and high mortality. References
Fig. 1. Showing perforated appendicitis.
liquid diet, the lack of fecaliths, and the presumed relative infrequency of lymphatic hyperplasia in the periappendiceal region [6]. The high morbidity and mortality associated with appendiceal perforation is attributed to the thin appendiceal wall and an indistensible cecum, facilitated by the small undeveloped and functionally non-existent omentum, small size of the peritoneal cavity allowing a more rapid and diffuse contamination and little physiological reserve [3]. Because of the rarity of the pathology and atypical signs and symptoms, the diagnosis of perforated appendicitis would
[1] Stifle D, Stallmach T, Sachet P. Acute appendicitis in neonates: complication or morgues suit generis? Pediatr Surg Int 1998;14:122e3. [2] Drapala B, Trognon B, Canarelli JP, Tamboura Tientcheu A, Bensatti L, Razarimanantsoa L, et al. Appendicite neonatal. Arch Pediatr 2000;7:896e7. [3] Arora NK, Deorari AK, Bhatnagar V, Mitra DK, Singhal PK, Singh M, et al. Neonatal appendicitis: a rare cause of surgical emergency in preterm babies. Indian Pediatr 1991;28:1330e3. [4] Deguchi E, Iwai N, Yanagihara J, Nakamura K, Doi Y. Case report: neonatal appendicitis with perforation in a premature infant. Pediatr Surg Int 1990;5: 366e8. [5] Karaman A, Cavusoglu YH, Karaman I, Cakmak O. Seven cases of neonatal appendicitis with a review of the English language literature of the last century. Pediatr Surg Int 2003;19:707e9. [6] Ruff ME, Southgate WM, Wood BP. Neonatal appendicitis with perforation. Am J Dis Child 1991;145:111e2. [7] Shaul WL. Clues to the early diagnosis of neonatal appendicitis. J Pediatr 1981; 98:473e6. [8] Kwong MS, Dinner M. Neonatal appendiceal perforation and Hirschsprung’s disease. J Pediatr 1980;96:917e8. [9] Arliss J, Holgersen LO. Neonatal appendiceal perforation and Hirschsprung’s disease. J Pediatr Surg 1990;25:694e5.