PMC7658569_01
Preoperative chest x-ray radiograph.. Diffuse infiltration of the left lung.
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A 17-month-old Japanese girl presented with a four-day history of fever (39.1  C) and persistent cough; WBC (41600/muL) and CRP (12.9 mg/dL) suggested severe inflammation, and a chest x-ray (CXR) revealed a diffuse infiltration of the left lung (Fig. 1) consistent with a diagnosis of pneumonia.
Preoperative computerized tomography (lt) and upper gastrointestinal series (rt).. Poorly aerated abnormal left lung with the fistula between the esophagus and the lung (arrow), and atrophic/hypoplastic left pulmonary artery (arrowhead).. A guidewire has been passed through the fistula, from the esophagus to the left lung.
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CXR on presentation. Computed tomography (CT) demonstrated a poorly aerated abnormal left lung with suspicion of a bronchopulmonary fistula (Fig. 2). An upper gastrointestinal series identified a fistula between the oesophagus and the left lung that was considered most likely be our case's left main bronchus embryologically (Fig. 2). Because of severe coexisting left pulmonary artery hypoplasia, there was no indication for preserving the left lung, and pneumonectomy was planned (Fig. 2).
Bronchoscopy and 3-D image from the computerized tomography.. Normal trachea, normal right main stem bronchus, and normal right distal airways, but no bifurcation of the left main stem bronchus (dotted circle). Arrow: Fistula.
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On bronchoscopy, the trachea, right main stem bronchus, and right distal airways were normal; however, there was no evidence of bifurcation of the trachea and no left main stem bronchus (Fig. 3).
Computed tomography pre and post conservative management.. Shrunken left lung after conservative management (lt) compared with its initial appearance (rt).
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After two weeks, CT showed improvement in her pneumonia (Fig. 4), WBC was 9300/muL, and CRP was 0.3 mg/dL, and thoracoscopic surgery was considered feasible.
Trocar positions for thoracoscopy.. All 4 trocars (5-mm) have been placed between the border of the erector spinae muscle and the posterior axillary line. Rt: Trocar in the 7th intercostal space for the surgeon's right hand. Scp: Trocar in the 9th intercostal space for the scope. Lt: Trocar in the 10th intercostal space for the surgeon's left hand. Assist: Trocar in the 6th intercostal space for the assistant. Note: The incision for the Lt trocar has been partially extended to remove the excised lung.
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All trocars were placed between the border of the erector spinae and the posterior axillary line (Fig. 5).
Fistula between the esophagus and left lung (intraoperative findings).. The fistula was identified and dissected circumferentially (lt).. The fistula was clamped and transected with a stapling device (rt).
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The oesophagus was identified and dissected free from the pleura, and the oesophageal fistula was identified and dissected circumferentially (Fig. 6). The trocar in the 10th intercostal space was removed and a surgical stapling device (ENDOPATH(R) ETS stapler, Ethicon Endo-surgery; Johnson & Johnson, Cincinnati, OH, USA) was introduced to transect the fistula directly (Fig. 6).
Pulmonary vein and artery (intraoperative findings).. The inferior pulmonary vein was dissected free then transected (lt).. The pulmonary artery was clamped and transected with a stapling device (rt).
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A 3-4 mm inferior pulmonary vein was identified and dissected free circumferentially, then transected with a vessel sealing device (LigaSure , Covidien, Mansfield, MA, USA) (Fig. 7). The pulmonary artery was identified and dissected circumferentially, noted to be 7-8 mm in size, clamped and transected with a different surgical stapling device (Echelon FLEX  stapler, Johnson & Johnson, Cincinnati, OH, USA) (Fig. 7).
Histopathology of Stoker type-3 congenital pulmonary airway malformation.. High power magnification showed cuboidal epithelium lining spaces (a). Histopathology of necrotizing acute bronchopneumonia (aspiration pneumonia). Low power magnification showing severe neutrophils accumulation and necrosis in bronchus and alveolar spaces (b: arrowheads). High power magnification showing acute inflammation surrounding foreign body giant cells (c: arrows).
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Histopathology showed Stoker type-3 congenital pulmonary airway malformation (Fig. 8).
Postoperative esophagogram.. Postoperative esophagogram showing no signs of leakage (rt).
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The patient tolerated surgery well, and an oesophagogram performed on postoperative day 5 showed no evidence of leakage (Fig. 9).