Ann Thorac Surg 1996;62:276-278
© 1996 The Society of Thoracic Surgeons
Case Report
Giant Bronchogenic Cyst Presenting as a Lobar Emphysema in a Newborn
Hamit Okur, MD,
Mustafa Küçükaydin, MD,
Adnan Öztürk, MD,
Süleyman Balkanli, MD,
Ali Bozkurt, MD
Departments of Pediatric Surgery, Pediatrics, and Pathology, Erciyes University Faculty of Medicine, Kayseri, Turkey
Accepted for publication January 23, 1996.
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Abstract
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Bronchogenic cysts are relatively uncommon congenital lesions. Because of the variability in clinical presentation and the shortcomings of diagnostic procedures, bronchogenic cysts present a diagnostic problem. This report describes a giant bronchogenic cyst that presented as a lobar emphysema in a newborn.
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Introduction
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Bronchogenic cysts arise from abnormal budding of the primitive tracheobronchial tree during airway development. Depending on the timing and orientation of this process, cysts can be located either within the mediastinum, within the lung parenchyma, or, rarely, in the lower neck. They may be detected on routine radiography in asymptomatic patients or may present with various manifestations. Operation is considered the treatment of choice, even in the asymptomatic patients [1–5].
A 25-day-old male neonate presented with a 5-day history of respiratory distress and cyanosis. Clinical examination revealed diminished breath sounds on the right side. Chest roentgenography showed overinflation of the right lobe and shift of the mediastinum to the contralateral side (Fig 1
). Ultrasonography and computed tomography revealed a homogeneously overexpanded, hyperlucent right upper lobe, which was interpreted as a right bullous emphysema (Fig 2). At thoracotomy a giant cyst originating from the right upper lobe was detected (Fig 3). There was a tiny communication between the cyst and the bronchial lumen. Right upper lobectomy with excision of the cyst was performed. Histopathologic examination of the lesion showed a bronchogenic cyst that was surrounded by a wall containing bronchial cartilage and smooth muscle and lined by columnar epithelium. The postoperative course was uneventful, and the control chest roentgenography performed after 1 month showed expansion of the right lung.
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Fig 1. . Chest radiograph showing overinflation of the right lobe and displacement of the mediastinum to the contralateral side.
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Fig 2. . Computed tomographic scan of the chest showing homogeneously overexpanded, hyperlucent right upper lobe.
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Fig 3. . Intraoperative photograph demonstrating a giant bronchogenic cyst originating from the right upper lobe and bulging through the incision.
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Comment
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The clinical presentations of bronchogenic cysts are variable. These cysts frequently maintain some connection to the tracheobranchial tree and usually produce signs early in life, either by enlarging rapidly as a result of air-trapping with the cyst or, more frequently, by becoming infected. In infants the most common presenting symptom is respiratory distress, whereas in older children pulmonary infections are the more usual mode of presentation [1–4]. Chest radiography is the usual imaging method for initial detection of bronchogenic cysts, although the findings are not always diagnostic [2]. In one series chest films were diagnostic in 77% of cases [4]. On a chest radiograph, an intrapulmonary bronchogenic cyst typically appears as a sharply defined, thin-walled, round or oval lesion that may be air filled, have homogeneous water density, or contain an air-fluid level [3, 6]. Bronchogenic cysts are often not visible in plain roentgenograms except for compression of the trachea or bronchus. The obstructed lobe or lung may be either emphysematous or atelectatic, depending on the degree of obstruction [1, 4, 5, 6]. Some patients present with congenital lobar emphysema, either extraluminal airway obstruction by a bronchogenic cyst or air trapping into a cyst [2, 4, 7]. Most often they are unilocular rather than multilocular, although they may contain internal trabeculations [1, 2]. In this case there was a tiny connection with the bronchus, and trabeculations that, simulating a bullous emphysema, were visible. Computed tomography has proved to be helpful in the diagnosis of bronchogenic cysts and often demonstrates a cyst not visualized by conventional radiographic techniques. Computed tomographic scanning provides optimal demonstration of cyst location, morphology, and contents [3, 5, 6]. Although chest roentgenography and computed tomographic scanning are the most valuable diagnostic studies in the differential diagnosis of cysts and cystlike lesions of the lung, we failed to diagnose a bronchogenic cyst in this case. This case represents an unusual presentation of this relatively uncommon congenital lesion. The possibility of an unrecognized bronchogenic cyst must therefore be considered whenever congenital lobar emphysema is diagnosed.
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Footnotes
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Address reprint requests to Dr Okur, Department of Pediatric Surgery, Erciyes University Faculty of Medicine, 38039, Kayseri, Turkey.
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References
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Abstract
Introduction
