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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 9  |  Issue : 1  |  Page : 86-88

Multiple idiopathic cecal perforation in a term neonate


1 Department of Pediatrics, Tata Motors Hospital, Jamshedpur, Jharkhand, India
2 Abhisek Child and Maternity Centre, Jamshedpur, Jharkhand, India

Date of Submission06-Aug-2019
Date of Decision19-Oct-2019
Date of Acceptance21-Oct-2019
Date of Web Publication29-Jan-2020

Correspondence Address:
Dr. Kumar Mitrabhanu
Department of Pediatrics, Tata Motors Hospital, Telco, Jamshedpur - 831 003, Jharkhand
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jcn.JCN_88_19

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  Abstract 


Neonatal spontaneous colonic perforation in term neonates is a rare phenomenon, cecal perforation being seldom reported. We report a case of spontaneous cecal perforation in a term, large for gestational age neonate, who became symptomatic on day 2 of life with vomiting and abdominal distension. On exploration, three large full-thickness cecal perforations with necrotic cecum were noted. Histopathologic samples were negative for necrotizing enterocolitis and Hirschsprung's disease. Clinical awareness of this rare entity could help in preventing delay in imaging and prompt surgical management.

Keywords: Cecum, idiopathic perforation, neonate


How to cite this article:
Mitrabhanu K, Ghosh S, Sharan R, Nath S. Multiple idiopathic cecal perforation in a term neonate. J Clin Neonatol 2020;9:86-8

How to cite this URL:
Mitrabhanu K, Ghosh S, Sharan R, Nath S. Multiple idiopathic cecal perforation in a term neonate. J Clin Neonatol [serial online] 2020 [cited 2020 Jul 14];9:86-8. Available from: http://www.jcnonweb.com/text.asp?2020/9/1/86/277232




  Introduction Top


Gastrointestinal perforation is a neonatal surgical emergency. Although perforation is commonly observed in the small intestine during the neonatal period, perforation of the colon is a rare condition. In preterm infants, colonic perforation is caused mostly due to necrotizing enterocolitis (NEC). It also may be associated Hirschsprung disease, meconium plug syndrome, small left colon syndrome, idiopathic perforations, anorectal malformations, stercoral perforations, and rarely cystic fibrosis.[1],[2],[3] In term and near-term infants, Hirschsprung disease is the leading cause, followed by NEC and spontaneous idiopathic perforation.[1],[2],[3],[4] Idiopathic perforation of the cecum in a term newborn is extremely rare and is seldom reported. We report a case of multiple idiopathic cecal perforation in a term male neonate.


  Case Report Top


A 4270-g male child of 40 weeks gestation age was born to a 27-year-old second gravida mother by elective cesarian delivery. Apgar scores were 8 and 9 at 1 and 5 min, respectively. He had no external congenital anomalies, passed meconium, and put on breastfeeds on day 1 of life. On the 2nd day of life, he developed nonbilious vomiting and poor feeding with a distended but soft abdomen without any redness of the abdominal wall. A plain abdominal radiograph showed dilated bowel loops. The baby was managed with empirical antibiotics, intravenous fluids, and trophic feed volumes after sending blood culture. However, abdominal distension increased, and the baby developed bilious vomiting and respiratory distress on the 3rd day of life. The baby was kept nil orally with nasogastric decompression and respiratory and fluid support. Suspecting intestinal obstruction, urgent laparotomy was planned by the pediatric surgeon. On exploration, extensive fecal peritonitis was noted. In gut tracing, three large full-thickness perforations were identified in the cecum [Figure 1]. The cecum was congested and necrotic. Resection of necrotic segment with proximal ileostomy was done. Multiple colonic and rectal biopsies were taken. The postoperative course was complicated by persistent septic ileus. The baby developed disseminated intravascular coagulation and pulmonary hemorrhage. The baby could not be salvaged despite aggressive antibiotics and multiple fresh frozen plasma and platelet transfusion. All colonic and rectal biopsies revealed nonspecific inflammatory signs and excluded NEC and Hirschsprung disease.
Figure 1: Three full thickness perforations over the caecum

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  Discussion Top


Hirschsprung disease is perhaps the only condition where a routine correlation to a cecal perforation has been made in case studies.[1],[2],[4] The incidence of 3%–4% is however largely biased toward long-segment Hirschsprung disease. Idiopathic perforation of the cecum has almost always been reported in isolated case reports as a rare entity.[4],[5] Most of these cases are diagnosed after the possibilities of a probable etiology have been carefully excluded.

Another etiology that is closely related is NEC. However, NEC is generally found in preterm babies. In preterm infants, most frequently, the ileum and jejunum are involved if perforation occurs. Intestinal mucosal injuries stemming from low-flow states or hypoxia and related bacterial invasion have been demonstrated as the most important etiological factors in the pathogenesis of NEC.[2],[3],[6]

In recent studies, idiopathic perforation in neonates has been defined as localized intestinal perforation unrelated to NEC. There could only be speculation about the cause of perforation in these infants. It has been suggested that ischemia is the common factor, but this etiology may be related to hypoxic neonates. Another problem commonly seen in very low birth weight infants is the immaturity of the intestine.[2],[4],[7],[8] Resch et al. suggested that the combination of episodes of hypoxia, resulting in ischemic injury to the gut, together with the immaturity of intestine could result in perforation in these infants.[9] In the present case, an unknown vascular insult causing cecal ischemia and perforation and progressing to hypercoagulopathy could be hypothesized.

Neonates with spontaneous intestinal perforations can be treated by drain alone, drain and laparotomy later, or primary laparotomy. An optimal management plan depends on the site of perforation, degree of contamination, and hemodynamic stability of the neonate. Among neonates undergoing primary laparotomy, majority require enterostomy formation. Secondary surgery during the neonatal period due to recurrent intestinal pathology may be required in half of the babies with spontaneous intestinal perforations.[10] In cecal perforations, unless Hirschsprung disease is ruled out, primary repair becomes impossible, and a stoma is usually done. Biopsies are recommended in all cases. The availability of frozen sections may help for an immediate decision, but its availability in most operation theaters is a limitation.[1],[2],[5],[6] In our patient, we had to perform an ileostomy as the ileocecal valve could not be salvaged.

The morbidity of these patients remains high, with the stoma output needing extensive resuscitation and intensive care unit care. In spite of efforts, the mortality rate in most studies is close to 50%.[1],[2],[9]


  Conclusion Top


Spontaneous perforation of the cecum without a demonstrable cause is sufficiently rare. Along with a careful history, it is extremely important to perform a histopathology in all cases. The management varies depending on the cause. Even with the best care, babies with proximal gut perforations tend to have a poorer prognosis than babies with colonic perforation.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the legal guardian has given his consent for images and other clinical information to be reported in the journal. The guardian understands that name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Grover J, Ali Q. A study of peritonitis in newborns and infants. Glob J Res Anal 2019 May 18;7(12).  Back to cited text no. 1
    
2.
Yu L, Tian J, Zhao X, Cheng P, Chen X, Yu Y, et al. Bowel perforation in premature infants with necrotizing enterocolitis: Risk factors and outcomes. Gastroenterol Res Pract 2016;2016:6134187.  Back to cited text no. 2
    
3.
Singh S, Rawat J, Wakhlu A, Kureel SN, Pandey A. Six-year retrospective analysis of colonic perforation in neonates and infants: Single centre experience. Afr J Paediatr Surg 2012;9:102-5.  Back to cited text no. 3
  [Full text]  
4.
Saraç F, Ataoǧlu E, Tatar C, Hatipoǧlu HU, Abbasoǧlu L. Neonatal colonic perforation. Ulus Cerrahi Derg 2015;31:44-6.  Back to cited text no. 4
    
5.
Shah J, Singhal N, da Silva O, Rouvinez-Bouali N, Seshia M, Lee SK, et al. Intestinal perforation in very preterm neonates: Risk factors and outcomes. J Perinatol 2015;35:595-600.  Back to cited text no. 5
    
6.
Demir N, Tuncer O, Melek M, Kaba S, Bulan K, Peker E. Idiopathic neonatal colonic perforation. J Neonatal Surg 2014;3:6.  Back to cited text no. 6
    
7.
Digray NC, Mengi Y, Goswamy HL, Thappa DR. Colorectal perforations in neonates with anorectal malformations. Pediatr Surg Int 2001;17:42-4.  Back to cited text no. 7
    
8.
Siddiqui MM, Burge DM. Neonatal spontaneous colonic perforation due to cystic fibrosis. Pediatr Surg Int 2008;24:863-4.  Back to cited text no. 8
    
9.
Resch B, Mayr J, Kuttnig-Haim M, Reiterer F, Ritschl E, Müller W. Spontaneous gastrointestinal perforation in very-low-birth-weight infants – A rare complication in a neonatal intensive care unit. Pediatr Surg Int 1998;13:165-7.  Back to cited text no. 9
    
10.
Drewett MS, Burge DM. Recurrent neonatal gastro-intestinal problems after spontaneous intestinal perforation. Pediatr Surg Int 2007;23:1081-4.  Back to cited text no. 10
    


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