Ann Thorac Surg 2008;85:2114-2116. doi:10.1016/j.athoracsur.2007.12.051
© 2008 The Society of Thoracic Surgeons
Case Reports
Clinicopathologic Demonstration of Complex Bronchopulmonary Foregut Malformation
Hakan Kiral, MDa,
Cagatay Saim Tezel, MDb,*,
Altug Kosar, MDa,
Murat Keles, MDa
a Department of Thoracic Surgery, Sureyyapasa Chest Disease and Thoracic Surgery Teaching and Research Hospital, Istanbul, Turkey
b Department of Thoracic Surgery, Haydarpasa Numune Teaching and Research Hospital, Istanbul, Turkey
Accepted for publication December 18, 2007.
* Address correspondence to Dr Tezel, Yazmac
Tahir sok 51/9 A Blok, Bostanc-Istanbul, 34744, Turkey (Email: mdcagatay{at}hotmail.com).
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Abstract
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Bronchopulmonary foregut malformations are rare and often prove to be extremely difficult to diagnose. The presented case is a 44-year-old man complaining about cough and hemoptysis. These symptoms continued despite medical treatment. A computed thorax tomographic scan depicted a paravertebral cystic lesion. The patient underwent surgical exploration, and communication between the cyst and the esophagus was found. A left lower lobectomy was performed. The pathologic result confirmed a duplication cyst of mixed bronchogenic and esophageal type with bronchiectasis of the lower lobe. We present this unusual malformation complex especially in an adult with the review of the literature.
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Introduction
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The trachea and esophagus embryologically originate as a common epithelial tube. The term bronchopulmonary foregut malformation was coined by Gerle and colleagues [1] to describe pulmonary sequestration having patent communications with the upper gastrointestinal tract. The aim of the article is to expose the complex malformation with highly interesting and demonstrative pictures of the lesion, which were discussed in light of the literature.
A 44-year-old man was referred to our clinic with a history of hemoptysis, chest pain, and cough. His symptoms had developed within the last 5 months. He had been treated with several antibiotic regimes for lung abscess in different hospitals. Despite regression with the treatment, his symptoms were persistent. In the past medical history he had had tuberculosis in 1973, received anti-tuberculous treatment, and was cured. Figure 1
shows both chest roentgenograms and a thoracic computed tomographic scan.
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Fig 1. Chest roentgenogram shows (a) enlarged left hilum and elevated left diaphragm. Lateral chest roentgenogram shows (b) a cystic mass with air-fluid level located in the posterior mediastinum (white arrow). Computed thorax tomographic scan demonstrates (c) a 3-cm cystic lesion with a thin wall in the left paravertebral region.
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A further investigation with fiberoptic bronchoscope was done. Purulent high-density sputum was aspirated from the left lower lobe bronchi. A culture performed for mycobacterium tuberculosis was negative. Bronchial lavage was negative for malignant cells.
The patient underwent surgical resection through a left posterolateral thoracotomy incision for both diagnosis and treatment. During the mobilization of the lung, a fistula between the cystic lesion and the esophagus was seen. This mucous fistula tract was dissected, cut and oversewn by 3-0 Vicryl (Ethicon Inc, Somerville, NJ) with two layers, without opening the esophageal mucosa. Because of the extensive destruction of the lung parenchyma, the cystic mass was excised along with the left lower lobe.
Pathologic anatomy indicated an esophageal duplication cyst and a bronchogenic cyst communicating with each other. When the duct, named as a mucosal fistula, was dissected, a 3 x 2 cm diameter cavitary lesion was detected (Fig 2a). The wall of the lesion was lined with ciliated columnar epithelium (Fig 2b) and the presence of cartilage, which demonstrated a bronchogenic cyst. A highly interesting finding in this cavity was a fruit seed located in one of the compartments of the cavity (Fig 2c). While continuing the dissection, another 2 x 2 cm cavitary lesion was found, which was filled with fruit seeds. The second cavity wall was covered with squamous epithelium, which had several metaplastic focuses.
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Fig 2. (a) *Mucosal fistula, **left lower lobe superior segment bronchus, and ***common basal segment bronchus were marked. (b) Fruit seeds are located in one of the compartments of the cavity. (c) Ciliated columnar epithelium cells originated from the cavitary lesion wall was seen (Hematoxylin & eosin stain, x100).
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There was no complication in the postoperative period. After the definitive surgical pathology, a barium swallow esophagogram was performed. The sutured fistula was detected on the 29th cm of the esophagus accompanying a sliding hiatal hernia. On follow-up at 3 years, the patient was free of respiratory and gastrointestinal symptoms.
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Comment
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Esophageal duplication cysts are rare abnormalities, generally congenital, and they form at approximately week 7 of embryogenesis. A portion of this embryonic structure can become isolated from the rest and results in a congenital epithelial cyst. When the lining of the cyst is ciliated columnar epithelium, it is referred to as a bronchogenic cyst; when the lining is compassed of squamous epithelium, it is referred to as an esophageal duplication cyst [2]. Nearly 20% of primary mediastinal lesions are non-neoplastic mediastinal cysts and approximately 10% of them are enteric or duplication cysts [3].
Infection of benign cysts may cause symptoms in adults, although at present such inflammatory complications are noted infrequently. The diagnosis of the cysts were often incidental. Dysphagia was noted as the most frequent symptom of the esophageal cysts, whereas chest pain was noted for bronchogenic cysts [3]. In our patient, chest pain and hemoptysis were foremost, which was thus revealed as bronchogenic cysts.
Most bronchogenic and esophageal duplication cysts were located on the right posterior mediastinum. The distal portion of the esophagus was one of the most common sites of communication (83%) [3]. The esophagogram is the most useful diagnostic procedure. When the cyst gets infected or there is communication with the tracheobronchial tree, an air bubble is seen in the cyst.
Carcinomas arising from esophageal duplication cysts have been reported [4]. The preoperative diagnosis is not always possible, especially in patients without upper gastrointestinal symptoms. Although minimal invasive diagnostic techniques have been suggested for biopsies from the mediastinal masses, they are far from providing definite diagnosis; hence they usually complicate the surgical removal. Surgical findings and histologic criteria allow a precise definition.
Complete surgical excision is required for both diagnosis and treatment. The prognosis after complete resection is good [3, 5]. In the present case, the accurate diagnosis could not be preoperatively achieved. The indication for thoracotomy was the parenchyma abscess with localized bronchiectasis that was resistant to medical treatment. Contrary to this, a detailed pathologic examination showed a communication between cysts that was defined as both esophageal duplication and bronchogenic cysts.
Parenchymal bronchogenic cysts associated with communication between esophageal duplication cysts are rare. In this report, we will be able to see the esophageal duplication cyst filled with fruit seeds, as well as the communication between bronchogenic cysts. There are few case reports highlighting the coexistence of these complex anomalies [3, 6, 7]. From the surgical point of view, as Cioffi and colleagues [8] stated, esophageal duplication cysts should be excised by preserving the muscle layer. Nasogastric tube should be placed before the surgical excision and checked intraoperatively.
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References
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- Gerle RD, Jaretzki A, Ashley CA, Berne AS. Congenital bronchopulmonary foregut malformation N Engl J Med 1968;278:1413-1419.[Medline]
- Ribet ME, Copin MC, Gosselin BH. Bronchogenic cysts of the lung Ann Thorac Surg 1996;61:1636-1640.[Abstract/Free Full Text]
- Strollo DC, Rosado-de-Christenson ML, Jett JR. Primary mediastinal tumors: part II. Tumors of the middle and posterior mediastinum Chest 1997;112:1344-1357.[Medline]
- Lee MY, Jensen E, Kwak S, Larson RA. Metastatic adenocarcinoma arising in a congenital foregut cyst of the esophagus: a case report with the review of the literature Am J Clin Oncol 1998;21:64-66.[Medline]
- Bolton JWR, Shahian DM. Asymptomatic bronchogenic cysts: what is the best management? Ann Thorac Surg 1992;53:1134-1137.[Abstract]
- Kitano Y, Iwanaka T, Tsuchida Y, Oka T. Esophageal duplication cyst associated with pulmonary cystic malformations Pediatr Surg 1995;30:1724-1727.
- Kim KW, Kim WS, Cheon JE, et al. Complex bronchopulmonary foregut malformation: extralobar pulmonary sequestration associated with a duplication cyst of mixed bronchogenic and oesophageal type Pediatr Radiol 2001;31:265-268.[Medline]
- Cioffi U, Bonavina L, De Simone M, et al. presentation and surgical management of bronchogenic and esophageal duplication cysts in adult Chest 1998;113:1492-1496.[Medline]
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Abstract
Introduction
