Interactive Cardiovascular and Thoracic Surgery 2:685-687(2003)
© 2003 European Association of Cardio-Thoracic Surgery
Negative results - Thoracic general |
Recurrent bronchogenic cyst 15 years after incomplete excision
Tsuyoshi Hasegawa*,
Fumio Murayama,
Shunsuke Endo and
Yasunori Sohara
Division of General Thoracic Surgery, Department of Surgery, Jichi Medical School, 3311-1 Yakushiji, Minami-kawachi, Kawachi, Thochigi, Japan
* Corresponding author. Tel.: +81-285-58-7368; fax: +81-285-44-6271
tcvgo{at}jichi.ac.jp
Received May 4, 2003;
received in revised form August 8, 2003;
accepted August 26, 2003
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Abstract
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Objectives: Bronchogenic cysts of the mediastinum are rare congenital anomalies that arise early in gestation from abnormal budding of the developing respiratory system. Recommended treatment is surgical resection and incomplete resection may lead to local recurrence more than 10 years later with or without various symptoms. Methods: We reported a case and reviewed literatures. Results: A case of a recurrent bronchogenic cyst in a 42-year-old man 15 years after first resection is presented. The patient had a persistent high fever resistant to antibiotic therapy. Magnetic resonance imaging and subsequent thoracotomy revealed a recurrent bronchogenic cyst. This case illustrates that incompletely resected mediastinal bronchogenic cyst may recur with later symptoms. Conclusions: Recommended treatment for bronchogenic cysts is complete surgical excision.
Key Words: Bronchi; Cysts; Mediastinal tumor; Reoperation
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1. Introduction
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Bronchogenic cysts (BCs) of the mediastinum are rare congenital anomalies that arise early in gestation from abnormal budding of the developing respiratory system. The location of the BCs depends on the embryonic stage of development at which the anomaly occurs. Mediastinal BCs account for 10–15% of all primary mediastinal masses. Recommended treatment is surgical resection, and incomplete resection may lead to local recurrence more than 10 years later.
We report a patient with persistent high-grade fever and recurrent mediastinal BCs noted 15 years after incomplete resection.
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2. Case report
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A 42-year-old man was admitted to our hospital because of a mediastinal mass and persistent high-grade fever. Three months earlier, he had presented to another hospital with high fever and cervical lymph node swelling. He was treated with antibiotics and non-steroidal anti-inflammatory drugs, but his fever did not resolve. Chest X-ray revealed a double-density mass in the subcarinal region with slight widening of the carinal angle. Computed tomography of the chest showed a well defined isodensity subcarinal mass, and magnetic resonance imaging showed a strongly intensified mass in the same region on T2-accentuated film (Fig. 1). Past history showed a right thoracotomy performed 15 years earlier for incomplete removal of the BC because of tight adhesion surrounding it. Physical examination was normal except for the presence of persistent high fever, at around 40 °C, and a well-healed right thoracotomy scar. Cervical lymph node size was not significant on admission. Laboratory data revealed mild leukocytopenia, white blood cell count of 2500/mm3, and mild elevation of C-reactive protein, 1.9 mg/dl. Although antibiotics were administered, the high fever did not resolve. Results of blood, sputum and urine cultures were negative. Significant elevation in antibody titer by hemagglutination for parainfluenza virus was confirmed. We considered a diagnosis of recurrent BC with infection. Right thoracotomy was performed, and the BC, which was located in the subcarinal region and adhered tightly to the right main stem bronchus, was resected completely. The patient's persistent high fever continued on postoperative day 1, and 500 mg of methylpredenisolone was administered. The fever then resolved, and the patient remained afebrile during an uneventful postoperative course. Histologically, the specimens were composed of ciliated columnar epithelium, smooth muscle, and mucous glands, making these findings compatible with the diagnosis of BC. Fluid contained in the cyst showed high values for amylase and antibody titer for parainfluenza type 3 virus, but it was not cultured for identification of virus. Fluid in the cyst was not cultured. Follow-up 2 months later revealed complete resolution of his fever and respiratory symptoms.
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Fig. 1 Magnetic resonance imaging shows a strongly intensified, well circumscribed mass in the subcarinal region. (a) T1-accentuated film shows low intensity in the mass. (b) Significantly intensified mass is located in the same region on T2-accentuated film.
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3. Discussion
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Although many asymptomatic cases have been reported, a number of complications with BCs have been described. Infection is the most common; other reported complications include recurrent respiratory obstruction, fistulae to various structures in the chest, hemorrhage into the cyst cavity, superior vena caval syndrome, arrhythmias, pulmonary artery obstruction, cyst rupture, cyst recurrence following incomplete excision, and malignant transformation. Adenocarcinoma and sarcoma arising from the cyst wall have also been reported.
Few case reports of recurrent BCs associated with reoperation after incomplete excision exist. Residual growing BCs take more than 10 years to appear, and it is difficult to diagnose recurrent BCs; only eight cases have been reported until now [1–8]. Many BCs are asymptomatic, but recurrent BCs to tend to show symptoms. Although most BCs are located along the tracheobronchial tree, they have been reported in the neck, within the dura, or pericardium, in the presternal space, and below the diaphragm. BCs are most often seen in the subcarinal region.
Parainfluenza viruses are members of the Paramyxoviridae family, comprise the genus Paramyxovirus, and are distributed throughout the world. Infection is acquired in early childhood, so that by 8 years of age most children have antibodies to serotypes 1, 2 and 3. Type 3 infection has been detected at all times of the year. In adults, parainfluenza infections are generally mild and account for fewer than 5% of respiratory illnesses. In the present case, the patient had a persistent high fever suggestive of parainfluenza viral infection in the epithelium of the cyst wall, especially because hemagglutination of the parainfluenza virus revealed high titers in both the patient's serum and the fluid contained in the cyst. But fluid contained in the cyst regretfully could not be cultured. This virus, which tends to infect the respiratory tract, tends to infect the cyst wall preferentially, and our patient's persistent high fever might point to this. No previous reports were found concerning viral infected BCs.
Percutaneous or transtracheal needle aspiration has been suggested as an alternative to excision. These techniques have proven to be unsatisfactory, with cysts recurring in virtually all patients. Two investigators have reported symptomatic reaccumulation of mediastinal cysts within weeks to 2 years after simple aspiration [9,10].
Recommended treatment is complete resection [8], or if incomplete, residual mucosa should be treated by toxic agents or electrocautery, which destroy the epithelial layer. Some authors have reported the effectiveness of ethanol injection into the cyst wall, but further long-term follow-up is necessary. Complete surgical excision is the goal, and depending on cyst location, it can be performed through a right or left thoracotomy or video-assisted thoracic surgery.
doi:10.1016/S1569-9293(03)00204-4
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References
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Abstract
1. Introduction