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CASE REPORT
Year : 2020  |  Volume : 13  |  Issue : 5  |  Page : 564-566  

Do pneumoperitoneum in a stable extreme low-birth-weight infant always need laparotomy? A clinical conundrum


1 Department of Neonatology, KK Women's and Children's Hospital, Singapore
2 Department of Neonatology, KK Women's and Children's Hospital; Department of Paediatrics, Duke NUS Medical School, Yong Loo Lin School of Medicine, Lee Kong Chian School of Medicine, Singapore
3 Department of Paediatric and Neonatal Surgery, Columbia Asia Referral Hospital, Yeshwanthpur, Bengaluru, Karnataka, India

Date of Submission21-Jul-2019
Date of Decision03-Jun-2019
Date of Acceptance06-Sep-2019
Date of Web Publication7-Sep-2020

Correspondence Address:
Krishna Revanna Gopagondanahalli
Department of Neonatology, KK Women's and Children's Hospital, 100 Bukit Timah Road
Singapore
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/mjdrdypu.mjdrdypu_212_19

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  Abstract 


A premature female neonate born at 27 weeks of gestational age with a birth weight of 1000 g, developed spontaneous pneumoperitoneum on day 10 of life. The baby remained clinically stable with constant serial blood gases and inflammatory markers. In view of clinical stability, she was managed conservatively with parenteral nutrition and intravenous antibiotics and monitored closely for any clinical deterioration. After 7 days of conservative management, serial radiological images showed complete resolution of pneumoperitoneum. This case challenges the default notion of presumed necrotizing enterocolitis/spontaneous intestinal perforation and urgent laparotomy in an extreme preterm baby with pneumoperitoneum. Successful conservative management of this case also demands a question whether all pneumoperitoneum should be subjected to laparotomy.

Keywords: Idiopathic pneumoperitoneum, necrotizing enterocolitis, nonsurgical pneumoperitoneum, spontaneous intestinal perforation


How to cite this article:
Gopagondanahalli KR, Si Min SG, Chandran S, Sai Prasad T R. Do pneumoperitoneum in a stable extreme low-birth-weight infant always need laparotomy? A clinical conundrum. Med J DY Patil Vidyapeeth 2020;13:564-6

How to cite this URL:
Gopagondanahalli KR, Si Min SG, Chandran S, Sai Prasad T R. Do pneumoperitoneum in a stable extreme low-birth-weight infant always need laparotomy? A clinical conundrum. Med J DY Patil Vidyapeeth [serial online] 2020 [cited 2020 Sep 29];13:564-6. Available from: http://www.mjdrdypv.org/text.asp?2020/13/5/564/294351




  Introduction Top


Pneumoperitoneum is a radiological finding of free air in the peritoneal cavity. Its etiology in neonates is usually a perforated viscus until proven otherwise. The most common cause of pneumoperitoneum in the neonatal population is necrotizing enterocolitis (NEC) followed by other intestinal pathologies.[1] The standard of care across all neonatal intensive units is an urgent laparotomy. There is also a group of preterm and term infants who show complete resolution of pneumoperitoneum without any surgical intervention, but their incidence and mechanism is unclear.[2],[3] We present a case of conservatively managed pneumoperitoneum in an extremely low-birth-weight preterm neonate born at 27 weeks of gestation age with good outcome. To the best of our knowledge, this is the first reported case of extreme preterm with spontaneous resolution of pneumoperitoneum without concomitant pneumothorax.


  Case Report Top


A female baby was born vaginally at 27-week gestational age to a primigravida mother with unremarkable antenatal history. Her birth weight was 1000 g. She required surfactant therapy and assisted ventilation for 3 days due to respiratory distress and was subsequently managed on continuous positive airway pressure (CPAP) support. Breast milk was introduced on day 2 of life and progressed according to our institution's protocol.

On day 10 of life, she had feed intolerance with abdominal distension, but remained clinically stable. An abdominal X-ray was obtained, which showed pneumoperitoneum [Figure 1]. There were no radiological features suggestive of NEC. Chest X-ray did not show air leak. On clinical examination, the baby's abdomen was tense, distended, and nontender. There was no discoloration of the overlying skin and no palpable mass. The nasogastric aspirates were nonbilious. The baby's heart rate and blood pressure were within the normal range and remained stable on CPAP without any respiratory deterioration. Arterial blood gases did not show acidosis. Her full blood counts showed normal platelet counts with unremarkable inflammatory markers, and the renal panel was within the normal limits. Based on these observations, we stopped her enteral feeds and started total parenteral nutrition and intravenous antibiotics with added anaerobic coverage. CPAP support was replaced with low-flow nasal cannula. Abdominal ultrasound showed no evidence of echogenic free fluid. In view of the persevering clinical stability, laparotomy was deferred with continued close vigil for any clinical downturn. She was monitored for lactic acidosis, hyponatremia, and thrombocytopenia. Her blood cultures were sterile. Serial abdominal X-rays showed complete resolution of pneumoperitoneum by day 17 of life [Figure 2]. Enteral feeds were re-introduced 1 week after the resolution of pneumoperitoneum and tolerated well. She remained stable and discharged home on full enteral feeds. The baby was well on follow-up with appropriate growth and development.
Figure 1: (a) Anteroposterior abdominal X-ray showing free air under diagram (arrow heads) and (b) Lateral decubitus abdominal X-ray showing pneumoperitoneum

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Figure 2: Resolution of pneumoperitoneum on follow-up abdominal X-ray

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  Discussion Top


Pneumoperitoneum is an ordeal finding for neonatologists and pediatric surgeons. The supportive management in pneumoperitoneum includes cessation of enteral feeds, bowel rest with nasogastric tube insertion, intravenous fluid resuscitation, and parenteral antibiotics with anaerobic coverage. In most neonatal units, urgent surgical exploration is the standard of care for the fear of intestinal perforation with or without NEC.

However, a distinctive group of neonates completely recover from pneumoperitoneum with conservative management alone. The true nature, incidence, and mechanism behind spontaneous recovery in these cases are still obscure.

The aim of this article is to explore the enigma surrounding the nonsurgical pneumoperitoneum and practicality of such approach in extreme low-birth-weight infants.

Spontaneous (idiopathic/benign) pneumoperitoneum is an entity with a nonsurgical etiology. These babies have radiological evidence of pneumoperitoneum but remain stable and reported to recover completely without surgical intervention.[4],[5] The probable mechanism is the passage of free air under tension in the mediastinum along the vascular planes into the peritoneum. The normal anatomical openings in the diaphragmatic, pulmonary lymphatic channels; congenital diaphragmatic defects; or pleuroperitoneal fistulous connections may act as passage for the free air to enter the peritoneum.[6] In a survey of 92 patients receiving CPAP therapy, Altman and Johnson found evidence of pneumoperitoneum in four (4.3%) cases, with none requiring operative management.[7] The reported time for resolution of pneumoperitoneum ranges from 48 h to 10 days.[8]

Zerella and Mc Collough reported two babies with birth weight of 1000 grams who had respiratory distress and pneumoperitoneum without evidence of GI perforation, both babies did not survive.[5] Sammut et al. published the case of a 420-g baby on high-frequency oscillation with recurrent pneumoperitoneum requiring two laparotomies with no evidence of intestinal perforation; finally, pneumoperitoneum spontaneously resolved with survival of the baby.[9] Both Bakal et al. and Gummalla et al. reported pneumoperitoneum secondary to pneumothorax in extreme premature babies.[10],[11] All the above-mentioned babies had significant respiratory support/obvious pneumothorax which could substantiate the mechanism explained for idiopathic pneumoperitoneum. Our case had minimal respiratory support, and we found no evidence of pneumothorax on chest X-ray.

Sealed spontaneous intestinal perforation (SIP) is considered a distinct entity from NEC. The solitary, less commonly multiple intestinal perforations occurring most commonly in terminal ileum are characterized histopathologically by a mucosal thinning, small perforation without ischemic necrosis, or neutrophil infiltrates. The median age of perforation is 7 days. Abdominal distension with bluish discoloration of the overlying skin is classical. The echogenic free fluid on abdominal ultrasound is highly sensitive.[12] The spontaneous healing of SIP in preterms is reported.[13]

Reported cases of conservatively managed SIP in extreme preterm neonates are very sparse. Chioukh et al. reported the management of a 27-week-old premature infant with pneumoperitoneum by needle suction alone.[14] There was a dramatic improvement following needle suction of peritoneal air. Michel et al. reported six infants with a mean gestation of 27 weeks and birth weight of 1000 g with a diagnosis of pneumoperitoneum due to SIP. Five of the six babies showed complete resolution of pneumoperitoneum with needle aspiration alone.[15] None of the babies had evidence of concomitant air leaks or significant lung disease.

With the available evidence, it is reasonable to conclude that pneumoperitoneum even in extreme premature neonates can occur without intestinal perforation, and those due to isolated SIP can be managed with or without minimally invasive surgery, but the burning question is how to identify the one who can be spared of operative intervention? The following features may help in identifying the benign nature of pneumoperitoneum: incidental, asymptomatic pneumoperitoneum with persevering clinical status; absence of acidosis; thrombocytopenia; abnormal electrolytes and raised inflammatory markers on serial blood tests; and evidence of concomitant pneumothorax.

Finally, one cannot ignore the tremendous physiological stress of surgery on preterm infants and concerns of impact of general anesthesia on the developing brain.[16],[17],[18]

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 initials 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.



 
  References Top

1.
Calisti A, Perrelli L, Nanni L, Vallasciani S, D'Urzo C, Molle P, et al. Surgical approach to neonatal intestinal perforation. An analysis on 85 cases (1991-2001). Minerva Pediatr 2004;56:335-9.  Back to cited text no. 1
    
2.
Mularski RA, Sippel JM, Osborne ML. Pneumoperitoneum: A review of nonsurgical causes. Crit Care Med 2000;28:2638-44.  Back to cited text no. 2
    
3.
Abdelmohsen SM, Osman MA. Idiopathic neonatal pneumoperitoneum, a case report. Int J Surg Case Rep 2017;31:250-3.  Back to cited text no. 3
    
4.
Gupta R, Bihari Sharma S, Golash P, Yadav R, Gandhi D. Pneumoperitoneum in the newborn: Is surgical intervention always indicated? J Neonatal Surg 2014;3:32.  Back to cited text no. 4
    
5.
Zerella JT, McCullough JY. Pneumoperitoneum in infants without gastrointestinal perforation. Surgery 1981;89:163-7.  Back to cited text no. 5
    
6.
Macklin CC. Transport of air along sheaths of pulmonic blood vessels from alveoli to mediastinum: Clinical implications. Arch Intern Med 1939;64:913-6.  Back to cited text no. 6
    
7.
Altman AR, Johnson TH. Pneumoperitoneum and pneumoretroperitoneum. Consequences of positive end-expiratory pressure therapy. Arch Surg 1979;114:208-11.  Back to cited text no. 7
    
8.
Gupta R. Spontaneous pneumoperitoneum in paediatrics patients: Dilemmas in management. J Indian Assoc Pediatr Surg 2018;23:115-22.  Back to cited text no. 8
[PUBMED]  [Full text]  
9.
Sammut A, Soares Oliveira M, Jackson C, Sortica da Costa C. Pneumoperitoneum in a neonate weighing less than 500 g. What do we really know about it? BMJ Case Rep 2018;2018. pii: bcr-2018-224398.  Back to cited text no. 9
    
10.
Bakal U, Aydin M, Orman A, Taskin E, Kazez A. A non-surgical condition of neonatal pneumoperitoneum: Retroperitoneal free air secondary to massive tension pneumothorax. J Med Cases 2016;7:13-4.  Back to cited text no. 10
    
11.
Gummalla P, Mundakel G, Agaronov M, Lee H. Pneumoperitoneum without intestinal perforation in a neonate: Case report and literature review. Case Rep Pediatr 2017;2017. doi: 10.1155/2017/6907329.  Back to cited text no. 11
    
12.
Fischer A, Vachon L, Durand M, Cayabyab RG. Ultrasound to diagnose spontaneous intestinal perforation in infants weighing ≤ 1000 g at birth. J Perinatol 2015;35:104-9.  Back to cited text no. 12
    
13.
Khan RA, Mahajan JK, Rao KL. Spontaneous intestinal perforation in neonates: Is surgery always indicated? Afr J Paediatr Surg 2011;8:249-51.  Back to cited text no. 13
[PUBMED]  [Full text]  
14.
Chioukh FZ, Ben Ameur K, Laamiri R, Ben Hmida H, Nouri A, Monastiri K. Spontaneous intestinal perforation in a very low birth weight infant: Successful management by peritoneal needle suction. J Neonatal Surg 2016;5:39.  Back to cited text no. 14
    
15.
Michel JL, Harper L, Alessandri JL, Jacquemot L, De Napoli-Cocci S, Pilorget H, et al. Peritoneal needle suction for intestinal perforation in the preterm neonate. Eur J Pediatr Surg 2004;14:85-8.  Back to cited text no. 15
    
16.
Anand KJ, Brown MJ, Bloom SR, Aynsley-Green A. Studies on the hormonal regulation of fuel metabolism in the human newborn infant undergoing anaesthesia and surgery. Horm Res 1985;22:115-28.  Back to cited text no. 16
    
17.
Vutskits L. Anesthetic-related neurotoxicity and the developing brain: Shall we change practice? Paediatr Drugs 2012;14:13-21.  Back to cited text no. 17
    
18.
Nestor KA, Zeidan M, Boncore E, Richardson A, Alex G, Weiss M, et al. Neurodevelopmental outcomes in infants undergoing general anesthesia. J Pediatr Surg 2017;52:895-900.  Back to cited text no. 18
    


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  [Figure 1], [Figure 2]



 

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