Bilateral Giant Pulmonary Bronchogenic Cysts
Ligang Liu, MD,
Tiecheng Pan, MD,
Xiang Wei, MD
Department of Cardiothoracic Surgery, Tong Ji Hospital, Tong Ji Medical College, Hua Zhong University of Science and Technology, Wu Han, China
Ligang Liu, MD, Tel: +86 27 8366 3671, Fax: +86 27 8366 3671, Email: liuligang{at}hotmail.com, Department of Cardiothoracic Surgery, Tong Ji Hospital, Tong Ji Medical College, Hua Zhong University of Science and Technology, Wu Han 430030, China.
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ABSTRACT
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Bilateral giant tension bronchogenic cysts were detected by computed tomography in a 13-year-old girl who presented with fever and severe cough. One was located in the right upper lobe, the other in the left lower lobe. The cysts, both measuring 10 cm in diameter, were removed in 2 operations 2 months apart.
Key Words: Bronchogenic Cyst • Cough • Respiratory Sounds
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INTRODUCTION
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Pulmonary bronchogenic cyst (PBC) has a wide range of clinical and radiological manifestations. Most cysts are asymptomatic and only recognized incidentally at radiography, but they occasionally lead to serious life-threatening complications such as infection, hemothorax, tension pneumothorax, and even malignant tumor formation. Therefore, surgical resection is recommended as early as possible, once diagnosis is confirmed, especially for the giant tension type. Pulmonary bronchogenic cyst is not uncommon in the cardiothoracic department, but a case of bilateral giant tension bronchogenic cysts is very rare.
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CASE REPORT
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A 13-year-old girl with no disease history, presented with cough (60–70 mL sputum per day) and fever (38.1°C) for 1 week. On chest auscultation, the breath sounds of the right upper lobe became weaker than the left, as did the left lower lobe. Computed tomography demonstrated 2 giant well-defined cystic lesions in bilateral lung fields, with thickened walls and an obvious air-fluid level inside. One was located in the right upper lobe and the other in the left lower lobe, each 8 x 8 cm in size (Figure 1
). The white blood cell count was 11 x 109/L, with a high proportion of neutrophilic granulocytes (80%). Thus the patient was preliminarily diagnosed with bilateral infected PBCs. Initial treatment was to control cystic infection with antibiotics, according to the sputum culture results. One week after her body temperature and white blood cell count returned to normal, chest radiography demonstrated elimination of the fluid in both cysts, but they were still the same size (Figure 1). Because this patient was too young to tolerate bilateral thoracotomies in one operation, the pulmonary cysts were resected in stages. Compression by the tension cyst was more severe in the right lung than the left, thus the first procedure dealt with the right pulmonary cyst. Through a right posterolateral thoracotomy, a giant cystic mass was found in the right upper lobe, tensile and 10 cm in diameter. The remnant of normal lung tissue in this lobe was very small. Lobectomy was carried out to eliminate the possibility of cyst recurrence and malignant degeneration of the residual epithelium. Oxygen saturation increased postoperatively from 90% to 99% in the extremities. The patient was given the same antibiotic therapy as before and recovered well. Two months later, she underwent a left thoracotomy. The intrapulmonary cyst was giant, 10 cm in diameter, located in the left lower lobe, with a small amount of surrounding normal lung tissue. This lobe was also resected to eliminate the possibility of cyst recurrence. Both pulmonary cysts were similar in gross appearance, with a thickened cystic wall and a little clear mucinous fluid inside (Figure 2). Histology revealed that both cystic luminal surfaces were lined by ciliated epithelium and the walls contained serous-mucinous glands and submucosal lymphocytic proliferation (Figure 2). The postoperative course was uneventful, and the patient was discharged on the 20th day after the last operation.
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Figure 1. Computed tomography showing 2 well-defined tension cystic lesions in the right upper lobe and left lower lobe, measuring 8 cm in diameter, with an obvious air-fluid level inside and severe compression of the surrounding normal lung tissues. After antibiotic chemotherapy, a standard posteroanterior chest radiograph demonstrated giant cystic lesions in both lungs, 10 cm in diameter, without an obvious air-fluid level, shaped like 2 balloons.
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Figure 2. In gross appearance, both giant cysts had a thickened cystic wall and a little clear mucinous fluid inside. Microscopically, the cysts were lined by ciliated epithelium. The walls contained chronic inflammatory infiltrates, necrosis, and connective tissue, bronchial glands, and smooth muscles.
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DISCUSSION
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Bronchogenic cysts are uncommon congenital anomalies of foregut origin and usually located within the mediastinum (two-thirds) or the lung (one-third).1,2 The frequency of bronchogenic cysts is unknown, presumably because most patients are asymptomatic. Histologically, the cyst wall is lined with pseudostratified ciliated epithelium, often with a focal area of metaplastic squamous or attenuated cuboidal epithelium. Bronchogenic cysts do not initially communicate with the tracheobronchial tree. Instrumentation of the cyst or infection may lead to an air-filled cyst or an air-fluid level.3 In this case, due to cystic infection, both cysts communicated with the normal bronchial tree through a single-direction valve, with and air-fluid level inside.
Clinical symptoms are due to the size and position of the cyst. Generally, PBCs present in 1 of 3 ways: symptomatic compression of adjacent intrathoracic structures in infants, asymptomatic radiographic findings, or infectious complications in adults.4 Symptoms are most often caused by compression of the trachea or bronchi, leading to cough, dyspnea, and pneumonia. Infection occurs in 20% of patients with intraparenchymal cysts.5 Our patient had symptoms of pulmonary infection on admission, and computed tomography indicated pulmonary cystic infection. To avoid uncontrolled life-threatening infection, it was important to control the infection before surgery.
Chest radiography can provide elementary diagnostic information, but computed tomography can locate an intrathoracic cyst, define its extent and relationship to key structures, and characterize the intrinsic density.6 Lobectomy is the best choice to treat an intrapulmonary cyst because it is often surrounded by areas of atelectasis and pneumonia; anatomic segmentectomy is reasonable when the cyst is small in size.7 Although fine-needle aspiration is useful and less traumatic for diagnosis and management, recurrences or complications may be observed during or after fine-needle aspiration.8 If staged surgery is needed for bilateral PBCs, care must be taken in choosing the optimal side for the initial operation, based on the degree of compression of surrounding lung tissue and the patient’s condition. Considering the possibility of rupture of the contralateral cyst, double-lumen endobronchial intubation and high-frequency ventilation with a low tidal volume should be guaranteed. Breath sounds of the contralateral lung should be monitored intraoperatively. The extent of resection depends on the size of the cyst and condition of the surrounding lung tissue. Lesions must be resected radically while preserving as much as possible of the surrounding healthy tissue. In this case, the cysts were both intrapulmonary and occupied most of the lobe, so lobectomy was necessary to eliminate the possibility of recurrence.
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REFERENCES
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Asian Cardiovasc Thorac Ann 2009;
17:64-66
© 2009 by SAGE Publications
DOI: 10.1177/0218492309102482
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