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Journal of Indian Association of Pediatric Surgeons
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Table of Contents   
CASE REPORT
Year : 2021  |  Volume : 26  |  Issue : 5  |  Page : 348-350
 

One-stage bilateral lobectomy in an infant with bilateral congenital lobar emphysema


1 Department of Pediatric Surgery, Healthway Hospital, Kadamba Plateau, Old Goa, Goa
2 Pediatric Intensive care Unit, Healthway Hospital, Kadamba Plateau, Old Goa, Goa
3 Department of Anesthesia, Healthway Hospital, Kadamba Plateau, Old Goa, Goa
4 Department of Pediatric Surgery, Healthway Hopsital, Kadamba Plateau, Old Goa, Goa and Aster Hospital, Margao, Goa, India, Goa

Date of Submission18-May-2020
Date of Decision06-Jul-2020
Date of Acceptance15-Aug-2020
Date of Web Publication16-Sep-2021

Correspondence Address:
Dr. Sumant Prabhudesai
Pediatric Intensive Care Unit, Healthway Hospital, Plot No. 132/1 (Part), Kadamba Plateau - 403 402, Old Goa, Goa
Goa
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaps.JIAPS_168_20

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   Abstract 


We report a 4-month-old baby presenting with bilateral congenital lobar emphysema. A two-staged bilateral lobectomy was planned, but bilateral lobectomy had to be performed as a single-staged procedure. Data are scarce on the appropriate approach to children with bilateral involvement. Both single-staged and two-staged procedures have shown variable success.


Keywords: Bilateral lobectomy, congenital lobar emphysema, left upper lobe


How to cite this article:
Sawant V, Prabhudesai S, Sawant B, Das S. One-stage bilateral lobectomy in an infant with bilateral congenital lobar emphysema. J Indian Assoc Pediatr Surg 2021;26:348-50

How to cite this URL:
Sawant V, Prabhudesai S, Sawant B, Das S. One-stage bilateral lobectomy in an infant with bilateral congenital lobar emphysema. J Indian Assoc Pediatr Surg [serial online] 2021 [cited 2022 Jul 6];26:348-50. Available from: https://www.jiaps.com/text.asp?2021/26/5/348/326064





   Introduction Top


Congenital lobar emphysema (CLE) is a rare disorder in infants.[1] While asymptomatic children can often be managed conservatively, surgical intervention is recommended for symptomatic ones. Literature related to bilateral disease is limited to case series and isolated case reports, and as such, there are no guidelines on the approach to bilateral disease.[2],[3],[4],[5] We report a 4-month-old infant with bilateral CLE who successfully underwent a one-staged bilateral lobar resection.


   Case Report Top


A 4-month-old male infant presented with a history of intermittent cough and breathlessness since the 1st month of age. His symptoms had failed to respond to multiple courses of oral antibiotics and nebulized bronchodilators. He was born at term to nonconsanguineous parents and weighed 2.5 kg at birth. His antenatal and perinatal course was uneventful.

At presentation, the child was mildly tachypneic with subcostal retractions. He weighed 6.1 kg, and general examination was unremarkable. Air entry was good bilaterally, and breath sounds were vesicular. Bilateral scattered wheeze was noted. His saturation was 95% in air. Cardiovascular, abdominal, and neurological examinations were normal.

Complete blood counts and renal and liver function tests were normal. His chest X-ray showed localized hyperinflation in the left upper zone. Contrast-enhanced computerized tomography (CT) of the thorax was suggestive of CLE of the left upper lobe and right middle lobe [Figure 1]. Two-dimensional echocardiography was normal. An elective thoracotomy was scheduled with a plan to perform a left upper lobectomy followed by an interval right middle lobectomy.
Figure 1: (a) Chest X-ray showing lobar emphysema involving the left upper lobe. (b) Chest computed tomography showing emphysematous right middle (striped arrow) and left upper lobes with adjacent normal lung segments (black arrow)

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He was premedicated with triclofos, glycopyrrolate, and ondansetron. General anesthesia was induced after preoxygenation with thiopentone and fentanyl. Oral intubation was performed with a 4.0 uncuffed endotracheal tube. Anesthesia was maintained by spontaneous with assisted ventilation with oxygen, air, and sevoflurane. Epidural bupivacaine was administered through an 18G epidural catheter, which was inserted caudally and directed cephalad to reach the T5–T6 level. Anesthesia was supplemented with intravenous midazolam and fentanyl and rectal paracetamol.

Left upper lobectomy was performed via left thoracotomy. Hyperinflation of the emphysematous right middle lobe was noted immediately after the lobectomy, resulting in persistent desaturation up to 80%. After ruling out endotracheal tube displacement on C-arm, a decision was made to proceed with right middle lobectomy via right thoracotomy. The saturation promptly improved upon right thoracotomy. The procedure was completed with no further complications.

Postoperatively, the child was electively ventilated in the intensive care unit for 48 h (synchronized intermittent mandatory ventilation with pressure control/pressure support – positive end-expiratory pressure 5 cmH2O, peak inspiratory pressure adjusted to achieve a tidal volume of 6–8 ml/kg, rate 30/min, Ti 0.6 s, and FiO2 0.4). He was sedated with fentanyl and midazolam infusions, and his ventilation was stable. He was successfully extubated on the 2nd postoperative day (POD) after a T-piece spontaneous breathing trial. Antibiotics were started preoperatively and continued for 5 days.

The pre- and post-extubation chest X-rays showed good inflation of the intact lung areas. The right and left chest drains were removed on the 8th and 9th PODs, respectively. He was discharged on the 10th POD.

The child was asymptomatic at 1-, 6-, and 12-month and 2-year follow-ups. Respiratory examination and oxygen saturation were normal. Growth parameters were within acceptable centiles. His Chest X-ray showed compensatory hyperinflation of the intact adjacent lobes with good overall lung aeration [Figure 2].
Figure 2: Postoperative chest X-ray showing good lung aeration

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   Discussion Top


CLE occurs due to overinflation of a lobe as a result of intrinsic deficiency of bronchial cartilage and elastic tissue. The overinflation results in partial obstruction of the lobar bronchus. CLE has an incidence of about 1:20000–1:30000 live births.[6] The left upper lobe, right upper lobe, and left middle lobe are most commonly involved in that order. About 20% cases have bilateral involvement.[1] Children usually present with symptoms in the newborn period although diagnosis may often be delayed till late infancy.

Although our initial plan was to perform a two-staged procedure, the intraoperative deterioration demanded a change of plan. While some authors suggest a two-staged approach to surgical resection with a variable interval between both resections, others have described a single-staged bilateral lobectomy.[2],[3],[4] In some of the cases reported earlier, the diagnosis of bilateral involvement was evident only intraoperatively or on postoperative imaging.[3] With the availability of modalities such as high resolution CT and magnetic resonance imaging, bilateral CLE can be diagnosed preoperatively making it possible to plan bilateral lobectomies as a single-staged procedure.[3]

Great variability exists in the anesthetic and ventilation techniques in infants with CLE. As we had bilateral involvement and therefore a greater risk of hyperinflation-related mediastinal shift and hemodynamic deterioration, we chose not to paralyze our patient. Although inhalational induction and maintenance of spontaneous ventilation is a common approach, intravenous induction, paralysis, and gentle positive pressure ventilation have been used safely in these children with unilateral CLE.[1],[7]


   Conclusion Top


One-stage bilateral lobectomy appears not only safe but sometimes becomes necessary in infants with bilateral CLE.

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.
Prabhu M, Joseph TT. Congenital lobar emphysema: Challenges in diagnosis and ventilation. Anesth Essays Res 2012;6:203-6.  Back to cited text no. 1
  [Full text]  
2.
Perea L, Blinman T, Piccione J, Laje P. Bilateral congenital lobar emphysema: Staged management. J Pediatr Surg 2017;52:1442-5.  Back to cited text no. 2
    
3.
Sathish Kumar T, Simon A, Sen S. Bilateral congenital emphysema: Unusual cause for recurrent respiratory tract infection. J Indian Assoc Pediatr Surg 2006;11:151-2.  Back to cited text no. 3
    
4.
Maiya S, Clarke JR, More B, Desai M, Parikh D. Bilateral congenital lobar emphysema: How should we proceed? Pediatr Surg Int 2005;21:659-61.  Back to cited text no. 4
    
5.
Abushahin AM, Tuffaha AS, Khalil NK, Ismeal AM. Bilateral congenital lobar emphysema: A rare cause for respiratory distress in infancy. Ann Thorac Med 2012;7:250-2.  Back to cited text no. 5
  [Full text]  
6.
Latif I, Shamim S, Ali S. Congenital lobar emphysema. J Pak Med Assoc 2016;66:210-2.  Back to cited text no. 6
    
7.
Kamal YA. Management of congenital lobar emphysema: The current challenges. ARC J Sur 2018;4:20-5.  Back to cited text no. 7
    


    Figures

  [Figure 1], [Figure 2]



 

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