Successful bronchotracheal reconstruction in esophageal bronchus: Two case reports

Successful Bronchotracheal By J.L. Michel,Y.

Revillon,

Reconstruction in Esophageal Bronchus: Two Case Reports

C. Salakos,

J. De Blic, D. Jan, A. Beringer, Paris, France

l Esophageal bronchus is the most common congenital bronchopulmonary foregut malformation. Current surgical treatment is resection of anomalous pulmonary tissue, which is often hypoplastic and destroyed by infection. The authors report two cases of bronchotracheal reconstruction. The diagnosis was early, before 15 days of age. The anomalous pulmonary tissue had a pulmonary arterial supply and venous drainage as assessed by angiography and a good functional capacity on selective ventilation. Bronchotracheal anastomosis was successful in both cases: a right main bronchus at 25 days of age and a left main bronchus at 13 days of age. One child underwent reoperation 1 year later for bronchomalacia of the reimplanted bronchus. Both children are well with normal growth 3 and 7 years after surgery. Chest roentgenograms showed normal and symmetrical lung aeration. Tracheal reimplantation may be prefered to pulmonary resection when the anomalous pulmonary tissue is not destroyed. The pulmonary functional capacity is increased and the complications of pneumonectomy avoided. Copyright o 1997 by W.B. Saunders Company INDEX WORDS: Bronchopulmonary esophageal bronchus, bronchotracheal

foregut malformation, reconstruction.

OMMUNICATING bronchopulmonary foregut malC formations (CBFM) are rare congenital anomalies characterized by a patent communication between the esophagus or stomach and an isolated portion of the respiratory system.’ Esophageal bronchus is the most common of these anomalies. We report two cases classified as group II according to the classification of CBFM proposed by Srikanth et al (one lung originates from the lower esophagus, and the ipsilateral mainstem bronchus is absent from the trachea).’ We performed two bronchotracheal anastomoses. We report our results and discuss the choice between reimplantation and pulmonary resection. CASE

REPORTS

Case 1 A 3.3-kg full-term girl, born after an uncomplicated pregnancy. presented at 13 days of age with respiratory symptoms and a nght lung opacity noted on plain chest roentgenogram (Fig 1). Bronchoscopy results showed the absence of the right main bronchus. Esophagoscopy results showed a fistulae at the lower third of the esophagus and the esophagogram showed a rtght bronchial tree connected to the esophagus (Fig 2). A digttal angrogram showed a normal pulmonary arterial supply and venous drainage. No assocrated anomaly was found. On the 25th day of life a reconstructive operation was performed by nght posterolateral thoracotomy. The right lung presented atelectasis but otherwise appeared normal macroscopically, and the mam bronchus Journal

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1997: pp 739-742

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was connected to the lower third of the esophagus. Selective ventilation of the transected bronchus showed reexpansion of the nght lung, leading us to implant the bronchus on the trachea. The right main bronchus was mobilized easily. The tracheal and right main bronchus diameter was normal for her age. A terminolateral bronchotracheal anastomosis with mterrupted suture of 6/O polydiaxonone (PDS) was performed. During the anastomosis a conventional mechamcal ventilation was maintamed. Extubation on the fifth postoperative day was well tolerated, with a nght lung aeration noted on chest roentgenogram (Fig 3). Scintigraphy showed good perfusion and ventilation of the lung and control angiography results showed satisfactory development of distal pulmonary vascularization (Fig 4). Bronchomalacia of the reimplanted bronchus with expiratory wheezing progressively developed. Bronchography examination showed a right main bronchus stenosis. She underwent reoperation 1 year later. The bronchus was incised longitudinally and reinforced with a pericardial and costal cartilage graft. The postoperative period was uneventful. Six years after the second operation, the patient was free of symptoms despite a moderate hyperventilation of the right lung on chest roentgenogram, and presented normal growth and development (Fig 5). The results of lung function studies were normal, and perfusion lung scintigraphy showed 28% on the operated side (right) and 72% on the left.

Case 2 A 2.8-kg full-term boy, born after an uncomplicated pregnancy, presented at birth with slight respiratory distress. A chest roentgenogram and a computed tomography (CT) scan showed a right lung distension with mediastmal shift and atelectasis of the left lung. A bat-mm esophagogram showed an esophageal bronchus. The main left bronchus was connected with the central thud of the esophagus. Angiography tests showed a normal pulmonary arterial supply and venous drainage, and a patent ductus arteriosus. During angiography a selective transesophageal ventilatron of the left bronchus showed opacification of the distal pulmonary arteriovenous circulation. No associated anomaly was found. On the 13th day of life a reconstructive operatton was performed by left posterolateral thoracotomy. The left lung appeared macroscopically normal although presenting atelectasis. The esophageal bronchus was transected and then ventilated resulting in reexpansion. Selective ventilation of the left lung maintained normal oxygen percutaneous blood saturation. The left main bronchus was mobilized easily. The tracheal and left main bronchus diameter was normal for hrs age We performed a terminolateral anastomosis with interrupted suture of 6/O polydmxonone (PDS) between the bronchus and the trachea, followed by ligation of the patent ductus arteriosus. During the anastomosis a conventional mechanical ventilation was mamtained. Postoperatively, left lung atelectasis persisted and an inflammatory anastomotic stenosis developed, which was cured by

From the Departments of Pediatric Surgery, Pediatric Pneumology, and Anesthesiology, Necker-Enfants Malades Hospital, Paris, France. Address reprint requests to Y Revillon, Clinique de Chirurgie Pddiatrique. Service de Chirurgie Visce’rale, Hdpital Necker-Enfaats Malades, 149 rue de Svres, 75743 Paris, Cedex 15, France. Copyright o 1997 by WB. Saunders Company 0022-3468/97/3205-0021$03.00/0 739

MICHEL

Fig 1. Preoperative chest right hemithorax opacity.

roentgenogram

of case

1, showing

a

suction bronchoscopy and administration of steroid (prednisone, 2 mg/kg/d). The patient was extubated on the 13th postoperative day. One year later, bronchoscopy tests showed a slight decrease of the anastomosis diameter, with a hypoperfusion of the left lung on scmtigraphy (26% to the left, 76% to the right). Three years after the bronchotracheal reconstruction, the patient was well, growth was normal, and the chest roentgenogram showed a normal symmetrical aerated lung.

Fig 3. Chest roentgenogram showing right lung aeration after

of case bronchial

ET AL

1 six days postoperative, reconstruction.

nonfunctioning pulmonary parenchyma destroyed by infections or hypoplasia.2~3~8-10 The vascularization can be pulmonary or systemic.1,3,6J1 The diagnosis is suspected by a parenchymal opacity

DISCUSSION

CBFM are rare malformations. In a review of the literature, Srikanth et al,’ reported only 57 cases including his own six. Communication with the lower esophagus is the most frequent presentation (66%), communication with stomach is less common (16%), and some cases of bilateral esophagus communication have been described.lm4Associated anomalies are frequent.3-7 Most of the reported cases present as communicating pulmonary sequestrations with systemic arterial supply, and with a

&-‘,<

_

za

Fig 2. Esophagogram of case 1 showing the opacification of the right main bronchus connected with the lower third of the esophagus (arrow).

Fig 4. (A) Preoperative angiography of case 1 showing the right pulmonary vascular supply and poor distal vascularization (arrow). (B) Postoperative angiography of case 1 showing the right pulmonary vascular supply and distal vascularization development (arrow).

BRONCHOTRACHEAL

741

RECONSTRUCTION

Fig 5. Chest roentgenogram showing a normal right lung sion, no mediastinal shift).

of case 1 seven years postoperative, (good aeration and pulmonary expan-

on chest roentgenogram. and confirmed by esophagogram and bronchoscopy. One case diagnosed by scintigraphy performed for gastroesophageal reflux has been reported. l2 The standard treatment is resection of anomalous pulmonary tissue.‘-3.5,10.‘1.‘3In group II CBFM as defined by Srikanth et al’ (one lung originates from the lower esophagus, and the ipsilateral mainstem bronchus is absent from the trachea), if the anomalous pulmonary tissue is hypoplastic or not completely destroyed, bronchial reconstruction could be functionally valuable avoiding pneumonectomy and its complications.‘~r” Early diagnosis is very important to preserve pulmonary parenchyma before destruction by infection, and a satisfactory pulmonary arterial blood supply and venous drainage

must be confirmed by angiography. In our two group II cases, the pulmonary vessels were normal without any systemic vascular supply. The pulmonary parenchyma was not destroyed. The assessment of the functional capacity of the lung can be performed by CT scan and oxygen percutaneous blood saturation measurements during selective ventilation of anomalous pulmonary tissue. The selective ventilation can be performed before surgery as in our second case, or during surgery as in our first case. In our second case the selective ventilation was performed at the same time of angiography and we observed a spectacular change in the distal pulmonary vascularization. The risks associated with tracheal reimplantation include leakage and stenosis. One of our patients suffered temporary stenosis cured by steroid treatment. In the first case a bronchomalachia of the reimplanted bronchus required bronchoplasty 1 year later. Our experience demonstrates that bronchial reconstruction is the best treatment when the anomalous pulmonary tissue is not destroyed. Only one case of bronchial reconstruction of CBFM has previously been published, and the patient died on postoperative day 5.6 After the diagnosis of CBFM, we suggest the best strategy is to assess the anomalous pulmonary tissue by CT scan. If it appears destroyed (atelectasis, bullae, abcess) or if only one lobe is involved (Srikanth groups I and III), no further investigation is necessary, and the treatment is the resection of the anomalous pulmonary tissue. If it does not appear destroyed, angiography should be performed. If the vascularization is pulmonary, selective ventilation of the anomalous pulmonary tissue should be performed to assessits oxygenation capacity. If the anomalous pulmonary tissue is functional, bronchial reconstruction is possible. Group II esophageal bronchus should be considered a particular form of CBFM that can be diagnosed early and easily. Angiography and selective ventilation of the bronchus are essential to help decide whether the corresponding parenchyma should be removed or conserved. If the anomalous pulmonary parenchyma is functional with a normal pulmonary vascular supply, conservative surgery may be appropriate.

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