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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 9  |  Issue : 4  |  Page : 295-299

Pneumatoceles due to Mechanical Ventilation in two preterm babies: A practical diagnostic approach


1 Department of Neonatology, Women wellness and Research Centre, Hamad Medical Corporation; Department of Pediatrics, Weill Cornell Medical College, Doha, State of Qatar
2 Department of Neonatology, Women wellness and Research Centre, Hamad Medical Corporation, Doha, State of Qatar

Date of Submission09-May-2020
Date of Decision15-Aug-2020
Date of Acceptance20-Aug-2020
Date of Web Publication01-Oct-2020

Correspondence Address:
Dr. Hussain Parappil
Department of Neonatology, Women's Wellness and Research Centre, Hamad Medical Corporation, PB. No 3050, Doha
State of Qatar
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jcn.JCN_66_20

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  Abstract 


Pneumatoceles are thin walled gas-filled cysts that develop within the lung parenchyma. Pneumatoceles are commonly seen after pneumonia, but rarely can also be seen after barotrauma from mechanical ventilation. Occasionally, it can rupture leading to air leak syndrome. We describe two interesting cases of large pneumatoceles in preterm babies due to ventilator associated injury and its management with review the literatures. We also propose a simplified practical approach for evaluation and management of these cases.

Keywords: Neonatal pneumatocele, prematurity, ventilator-induced lung injury


How to cite this article:
Parappil H, Ahmed SN, Kunhiabdullah S, Gaffari M. Pneumatoceles due to Mechanical Ventilation in two preterm babies: A practical diagnostic approach. J Clin Neonatol 2020;9:295-9

How to cite this URL:
Parappil H, Ahmed SN, Kunhiabdullah S, Gaffari M. Pneumatoceles due to Mechanical Ventilation in two preterm babies: A practical diagnostic approach. J Clin Neonatol [serial online] 2020 [cited 2020 Oct 19];9:295-9. Available from: https://www.jcnonweb.com/text.asp?2020/9/4/295/297006




  Introduction Top


Pneumatoceles (also known as pulmonary pseudocysts) are thin walled gas-filled cysts that develop within the lung parenchyma.[1] Pneumatoceles can occur at all ages and may coexist with pneumothorax, pulmonary interstitial emphysema, secondary infection, or pneumomediastinum which may require urgent treatment.[2],[3]

The etiology of pneumatoceles may be infectious, traumatic, congenital, or positive pressure ventilation induced. In older age groups, postinfectious pneumatocele is common in contrast to neonates where ventilator-induced pneumatocele[3],[4] is the most common etiology.

The incidence of pneumatoceles in babies born at ≤30 weeks gestation is 1.8%.[3] The mechanism of formation of pneumatoceles is controversial. Prematurity, respiratory distress syndrome (RDS), and use of positive pressure ventilation results in barotrauma and or volutrauma and a local increase in pro-inflammatory mediators and cytokines.[5] This can damage the epithelial lining of the lung parenchyma causing a focal area of inflammation, which in some cases may lead to a check valve type airway obstruction, air-trapping, and pseudo cyst formation.[6]

Here, we present two cases of pneumatoceles due to ventilator-associated injury in preterm babies, which got completely resolved with conservative management. We also propose a simplified practical approach for the evaluation of these cases.


  Case Reports Top


Case 1

Baby born, at 26 weeks' gestation to mother with unremarkable pregnancy, delivered by emergency cesarean section for nonreassuring fetal status with good Apgar Score 3 and 8 and weight of 1 kg. The baby required Mechanical ventilation and two doses of surfactant for severe RDS within 24 h of admission. Ventilatory support was escalated to high-frequency oscillatory ventilation (HFOV) (mean airway pressure [MAP] of 16) at 48 h of life for worsening respiratory acidosis. Initial X-ray showed moderate RDS [Figure 1] and the follow-up X-rays [Figure 2] and [Figure 3] showed the development of large pneumatocele on the left lower lobe (24 mm × 15 mm) on day 3. Infant sepsis parameter (complete blood cell [CBC] and C-reactive protein[CRP]) including blood culture and tracheal secretion cultures were negative and first line antibiotic was stopped at 48 h. The possibility of ventilator induced lung injury was thought and MAP was gradually weaned as tolerated over period of next 10 days. Follow-up X ray showed gradual reduction in the size of pneumatoceles corresponding to reduction in MAP. Subsequent X-ray [Figure 4] showed disappearance of pneumatocele on day 10 of life and infant could be weaned to nasal continuous positive airway pressure (CPAP) on day 16 of life. There were no further pneumatoceles on follow up till discharge.
Figure 1: Chest X-ray at 2 h of age Shows moderate respiratory distress syndrome and no evidence of pneumatocele

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Figure 2: Chest X-ray at day 3 of age shows a well demarcated air filled cavity (pneumatocele) on the left lower lobe of size 24 mm × 15.7 mm

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Figure 3: Chest X-ray Lateral view at day 3 of age shows a air filled cavity (pneumatocele) of size 16.5 mm × 16.4 mm

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Figure 4: Chest X-ray at 10 days of age shows resolution of the pneumatocele

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Case 2

A 34-week infant of diabetic mother required mechanical ventilation and surfactant replacement for severe RDS. HFOV was needed for worsening respiratory acidosis (maximum MAP 20) on day 2 of life. As infant sepsis workup (CBC and CRP along with blood culture) were negative, first-line antibiotic was stopped at 48 h. Initial X-ray showed moderate RDS [Figure 5] and the followed up X-rays [Figure 6] was noted to have developed right middle lobe pneumatocele (19.6 mm × 15.6 mm) on day 3 of life. A trial of weaning MAP at this junction was unsuccessful, and infant continue to require high MAP. She developed pneumothorax [Figure 7] the next day with rupture of pneumatocele and subsequently required tube thoracotomy. Her lung showed better expansion, after drainage of pneumothorax and subsequent resolution of pneumatocele. MAP was be gradually weaned and infant could be extubated on day 12 of life. Follow-up X-ray [Figure 8] after extubation showed no residual pneumatocele which was also confirmed with computed tomography scan before discharge.
Figure 5: Chest X-ray at 1 h of age shows moderate respiratory distress syndrome and no evidence of pneumatocele

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Figure 6: Chest X ray at day 3 of age shows a well demarcated air filled cavity (pneumatocele) on the right middle lobe of size 19.6 mm × 15 mm

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Figure 7: Chest X-ray at day 4 of age Shows right sided pneumothorax

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Figure 8: Chest X-ray at 2 weeks of age shows resolution of pneumatocele

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  Discussion Top


Studies have demonstrated that low birth weight and low gestational age infants have increased incidence of pneumatocele formation. Furthermore, the use of positive pressure ventilation had increased the risk of pneumatocele.[3] Both of our cases were preterm and required mechanical ventilation. de Bie et al. had reported a case of pneumatocele in a preterm baby due to pressure effects from CPAP.[7]

Pneumatoceles are described in neonates as a complication of pneumonia with Staphylococcus aureus, Streptococcus pneumonia, Escherichia coli, Klebsiella pneumonia, or Pseudomonas aeruginosa[8] and due to ventilator associated injury which is more common.[3] Manzar et al.[9] and Dembinski et al.[10] described pneumatocele must be differentiated from congenital anomalies such as congenital pulmonary adenomatous malformation, congenital lobar emphysema, pulmonary sequestration, and bronchogenic cyst[11] which can have similar appearance of air filled cavities on X-rays. These anomalies are usually present in the initial radiographs soon after birth.

In our cases, sepsis screening was negative and the initial X-rays have ruled out congenital anomalies. Hence, mechanical ventilation associated injury was the cause for pneumatoceles. [Table 1] shows a flow diagram for practical approach to evaluate pneumatoceles in a neonate.[9-11]
Table 1: Flow diagram for practical approach for Pneumatoceles in a neonate[9],[10,[11]

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The majority of pneumatoceles (more than 85%) will resolve spontaneously, partially, or completely over weeks to months without clinical or radiographic sequelae.[12] Several methods described for the management of pneumatoceles, including decubitus positioning with the affected side down, selective intubation of the uninvolved side, high frequency ventilation, chest tube drainage, or surgical resection.[12]

In the absence of infections and congenital anomalies, conservative management with reduction of MAPs or extubation is shown to be effective in improving these cases.[3] Hussain et al.[3] in 2010, reported pneumatoceles in 19 preterm babies with RDS requiring positive pressure ventilation, of which five infants died and the rest improved with conservative management. Harris[13] in 1977, Clarke and Edwards[14] in 1979, Williams et al.[15] in 1988, described many cases with ventilator-associated pneumatoceles in preterm and near tem babies which showed improvement with conservative management [Table 2].
Table 2: Series of cases of pnematoceles in neonates due to mechanical ventilation

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Both our cases improved with conservative management with reduction of MAPs.

Christopher et al.[16] in 2019, reported a case requiring lobectomy in a child as the weaning of ventilation was unsuccessful and Kumar et al.[17] in 2018 reported a successful percutaneous drainage of pneumatoceles in an extremely low-birth weight infant as lobectomy was contraindicated due to diffuse pathology and extremely low weight.

Park and Kim.[12] in 2015, reported a case treated successfully by a nonsurgical method in an enlarging, pneumatocele complicated with cardiorespiratory instability by the injection of fibrin sealant through a pigtail catheter. Both our babies did not require any surgical intervention.


  Conclusion Top


Pneumatocele formation is a rare complication due to mechanical ventilation especially in preterm babies. Due to lack of consistent association with infection or congenital anomalies, mechanical ventilator-related injury must be considered as a cause of this condition. Majority of these cases can improve with conservative management with a reduction in MAP and should be the primary modality of management. The need for surgical intervention should be restricted only to long standing cases or that show signs of mass effect unresponsive to reductions in MAP.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Quigley MJ, Fraser RS. Pulmonary pneumatocele: Pathology and pathogenesis. AJR Am J Roentgenol 1988;150:1275-7.  Back to cited text no. 1
    
2.
DiBardino DJ, Espada R, Seu P, Goss JA, Houston TX. Management of complicated pneumatocele. J Thorac Cardiovasc Surg 2003;126:859-61.  Back to cited text no. 2
    
3.
Hussain N, Noce T, Sharma P, Jagjivan B, Hegde P, Pappagallo M, et al. Pneumatoceles in preterm infants-incidence and outcome in the post-surfactant era. J Perinatol 2010;30:330-6.  Back to cited text no. 3
    
4.
Al-Ghafri M, Al-Hanshi S, Al-Ismaily S. Two cases of pneumatoceles in mechanically ventilated infants. Oman Med J 2015;30:299-302.  Back to cited text no. 4
    
5.
Clark RH, Slutsky AS, Gerstmann DR. Lung protective strategies of ventilation in the neonate: What are they? Pediatrics 2000;105:112-4.  Back to cited text no. 5
    
6.
Contreras M, Hariharan N, Lewandoski JR, Ciesielski W, Koscik R, Zimmerman JJ. Bronchoalveolar oxyradical inflammatory elements herald bronchopulmonary dysplasia. Crit Care Med 1996;24:29-37.  Back to cited text no. 6
    
7.
de Bie HM, van Toledo-Eppinga L, Verbeke JI, van Elburg RM. Neonatal pneumatocele as a complication of nasal continuous positive airway pressure. Arch Dis Child Fetal Neonatal Ed 2002;86:F202-3.  Back to cited text no. 7
    
8.
Bermejo Velasco E, González Molina E, Martinez Ayucar M, Zubia Aguillo A, Nieves Gil A, Salado Marin C, et al. Pneumatocele as a complication of Escherichia coli pneumonia in a newborn infant. Apropos of a case. An Esp Pediatr 1992;37:526-8.  Back to cited text no. 8
    
9.
Manzar S, Khan LH. A newborn with a cyst in the lung. Neoreviews 2017;18:e564.  Back to cited text no. 9
    
10.
Dembinski J, Kaminski M, Schild R, Kuhl C, Hansmann M, Bartmann P. Congenital intrapulmonary bronchogenic cyst in the neonate--perinatal management. Am J Perinatol 1999;16:509-14.  Back to cited text no. 10
    
11.
Kunyoshi V, Cataneo DC, Cataneo AJ. Complicated pneumonias with empyema and/or pneumatocele in children. Pediatr Surg Int 2006;22:186-90.  Back to cited text no. 11
    
12.
Park TH, Kim JK. Nonsurgical management of an enlarging pneumatocele by fibrin sealant injection via pigtail catheter. Pediatr Pulmonol 2015;51:E5-7.  Back to cited text no. 12
    
13.
Harris H. Pulmonary pseudocysts in the newborn infant. Pediatrics 1977;59:199-204.  Back to cited text no. 13
    
14.
Clarke TA, Edwards DK. Pulmonary pseudocysts in newborn infants with respiratory distress syndrome. AJR Am J Roentgenol 1979;133:417-21.  Back to cited text no. 14
    
15.
Williams DW, Merten DF, Effmann EL, Scatliff JH. Ventilator-induced pulmonary pseudocysts in preterm neonates. AJR Am J Roentgenol 1988;150:885-7.  Back to cited text no. 15
    
16.
Gerdung CA, Ross BC, Dicken BJ, Bjornson CL. Pneumonectomy in a child with multilobar pneumatocele secondary to necrotizing pneumonia: Case report and review of the literature. Case Rep Pediatr 2019;2019:2464390.  Back to cited text no. 16
    
17.
Kumar J, Mukhopadhyay K, Bhatia A. Successful percutaneous drainage of pneumatoceles in an extremely low-birthweight infant. BMJ Case Rep 2018;2018:bcr2017222630.  Back to cited text no. 17
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8]
 
 
    Tables

  [Table 1], [Table 2]



 

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