Eur J Cardiothorac Surg 1998;13:612-614
© 1998 Elsevier Science NL
Cervical bronchogenic cyst mimicking thyroid adenoma
Pierre Barsotti,
Antonis Chatzimichalis,
Gilbert Massard,
Jean-Marie Wihlm
Service de Chirurgie Thoracique, Hôpitaux Universitaires de Strasbourg, F-67091 Strasbourg, France
Received 28 July 1997;
received in revised form 26 January 1998;
accepted 24 February 1998.
Corresponding author. Tel.: +33 3 88116202; fax: +33 3 88116077.
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Abstract
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Bronchogenic cysts are congenital malformations of the foregut which are generally encountered within the mediastinum. We explored a patient presenting with a cystic, partially calcified tumor in a cervical and retrotracheal location. This lesion was interpreted as a thyroid adenoma preoperatively, but identified as a bronchogenic cyst at pathology.
Key Words: Bronchogenic cyst • Ectopia
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Introduction
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Bronchogenic cysts, which account for 6 to 15% of mediastinal tumors, are malformations arising at the time of organogenesis of the respiratory system [1]. During the third week of gestation, the trachea develops from a ventral diverticulum budding from the foregut
[1]
[2]. At the fourth week, the distal end of this tube separates to form the stem bronchi; after further budding, the lobar bronchi appear at 35 days of gestation. Abnormal budding during this period leads to formation of bronchogenic cysts, which are lined by a ciliated epithelium of the respiratory type
[1]
[2]. As expected from embryology, bronchogenic cysts are typically located within the middle compartment of the mediastinum along the trachea or stem bronchi, or within the posterior mediastinum in close vicinity to the esophagus. They are encountered with increased prevalence in the male, and on the right side of the midline. True bronchogenic cysts respond to four main locations: inferior paratracheal area, bronchial bifurcation, hilar area, and inferior pulmonary ligaments
[1]. Cysts arising later during gestation are located distally on the bronchial tree and are imbedded within the parenchyma. Paraesophageal cysts may also be lined with an esophageal mucosa and are then classified as esophageal duplications
[2].
The present case report illustrates the possibility of ectopic extrathoracic locations.
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Case report
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A 49-year-old male patient presented at the emergency room with recent onset of hemoptysis. His medical history was negative. General health status was normal. Functional signs were limited to an exertional dyspnea quoted 3/5, which had installed 6 weeks anteriorly. Physical examination disclosed a tracheal-type dyspnea with audible wheezing. Chest roentgenogram was normal. Fiberoptic bronchoscopy disclosed a major extrinsic compression of the membranous trachea at the cervical level; the lumen was reduced by 80%, and no distal exploration was performed. Computed tomographic scan of the cervical and mediastinal area showed a cystic cervical tumor in a retrotracheal location (
Fig. 1
Fig. 2
). The lesion appeared to be part of the body of the left lobe of the thyroid gland, and partial calcification suggested the diagnosis of a thyroid adenoma.
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Fig. 1. Computed tomographic scan demonstrates a cystic, partially calcified cervical mass suggestive of thyroid adenoma.
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The patient underwent surgical exploration of the neck through a collar incision. The calcified cystic tumor was clearly separated from the thyroid gland; the left thyroid lobe was merely reflected anteriorly by the tumor. The dissection proceeded easily, except along the membranous part of the trachea, where the adhesions were particularly dense. A part of the cystic wall was common to the trachea and the cyst, and was left in situ; the mucosa of this portion was abrased.
Histology showed that the lesion was lined with a bronchial-type mucosa and the diagnosis of bronchogenic cyst was therefore established (
Fig. 3
).
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Fig. 3. Histology (hematoxylin stain, x100) obviates a fibro-inflammatory wall lined with a ciliated mucosa and containing seromucous glands.
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Post-operative endoscopy revealed a normal tracheal lumen with an intact posterior membranous wall.
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Discussion
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Although rare, several ectopic locations of bronchogenic cysts have been reported. Intra-abdominal migration originating from the first branchial arch occurs early during organogenesis, prior to the fusion of the pleuroperitoneal membranes which are the precursors of the diaphragm. Isolated case reports describe parapancreatic and hiatal cysts
[3]. Various other exceptional ectopias have been reported within the diaphragm, the pericardium, the presternal area, or elsewhere subcutaneously
[1]
[4]
[5]. Ectopic cervical location seems to be rather exceptional, since to our knowledge a single previous case has been reported
[6].
Diagnosis of bronchogenic cysts is usually simple, although St Georges et al.
[1] demonstrated that diagnosis was made at operation in up to 57% of their series including 86 patients
[1]. Computed tomographic scan clearly sets the diagnosis in most events when a cystic mass is evidenced in vicinity of the respiratory tree. In a surgical perspective, anatomic relation to adjacent structures is adequately defined. Thoracoscopy appears as an invaluable diagnostic tool for differential diagnosis of mediastinal tumors
[7]
[8], although complete excision of a bronchogenic cyst by minimally invasive techniques may be challenging.
Treatment of bronchogenic cysts is well standardized in children, whereas different options may be discussed in adults. In a pediatric population, surgical excision of any bronchogenic cyst is recommended to prevent progressive growth and subsequent mediastinal compression
[2]
[9]. In adults, resection of intrapulmonary cysts is mandatory because the spontaneous complication rate is high owing to a frequent bronchocystic fistula
[9]. On the opposite, various options are to be discussed in case of an asymptomatic extrapulmonary lesion. Simple survey may be instituted for small and asymptomatic cysts
[9]. A conservative option with transbronchial or mediastinoscopic needle decompression has been described
[10]. However, most authors recommend routine surgical excision because of the risk for complications, and exceptional malignant change
[1]
[2]. Video-assisted thoracic surgery should be used with modesty, and conversion ought to be performed in case of dense adhesions with the bronchial wall, the esophagus or the pericardium
[2].
In ectopic locations, surgical resection and pathologic expertise are required to ascertain the diagnosis.
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References
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- St Georges R., Deslauriers J., Duranceau A., Brisson J. Clinical spectrum of bronchogenic cyst of the mediastinum and lung in the adult. Ann Thorac Surg 1991;52:6-13.[Abstract]
- Ribet M., Copin M., Gosselin B. Bronchogenic cysts of the mediastinum. J thorac Cardiovasc Surg 1995;109:1003-1010.[Abstract]
- Coselli M., De Ipolyi P., Bloss R., Diaz R., Fitzerald J. Bronchogenic cyst above and below the diaphragm: report of eight cases. Ann Thorac Surg 1987;44:491-494.[Abstract]
- Buddington T.W. Intradiaphramatic cyst, ninth reported case. N Engl J Med 1957;257:613.[Medline]
- Gomes M.N., Hufnagel C.A. Intrapericardial bronchogenic cyst. Am J Cardiol 1975;36:817.[Medline]
- Dubois P., Belanger R., Wellington J.L. Bronchogenic cyst presenting as a supraclavicular mass. Can J Surg 1981;24:530-531.[Medline]
- Kern J., Daniel T., Tribble C., Silen M., Rodgers B. Thoracoscopic diagnosis and treatment of mediastinal masses. Ann Thorac Surg 1993;56:92-96.[Abstract]
- Bolten J.R., Shahian D. Asymptomatic bronchogenic cysts: what is the best management?. Ann Thorac Surg 1992;53:1134-1137.[Abstract]
- Ginsberg R.J., Atkins R.W., Paulson D.L. A bronchogenic cyst successfully treated by mediastinoscopy. Ann Thorac Surg 1972;13:266-268.[Medline]
- Naunheim K., Andrus Ch. Thoracoscopic drainage and resection of giant mediastinal cyst. Ann Thorac Surg 1993;55:156-158.[Abstract]
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Abstract
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