HOME HELP FEEDBACK SUBSCRIPTIONS ARCHIVE SEARCH TABLE OF CONTENTS		QUICK SEARCH:   [advanced] Author: Keyword(s): Year:  Vol:  Page:

This Article Abstract Full Text (PDF) Alert me when this article is cited Alert me if a correction is posted Citation Map Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Add to Personal Folders Download to citation manager Author home page(s): Erdem Silistreli Ünal Açikel Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Sanli, A. Articles by Açikel, U. Search for Related Content PubMed PubMed Citation Articles by Sanli, A. Articles by Açikel, U. Related Collections Trachea and bronchi

Ann Thorac Surg 2004;77:1093-1094
© 2004 The Society of Thoracic Surgeons

---

Case report

A case of a bronchogenic cyst in a rare location

Aydn anli, MD^a*, Ahmet Önen, MD^a, Emel Ceylan, MD^b, Erkan Yilmaz, MD^c, Erdem Silistreli, MD^d, Ünal Açikel, MD^a

^a Thoracic Surgery, Dokuz Eylül University Medical School, Izmir, Turkey
^b Department of Pulmonary Medicine, Dokuz Eylül University Medical School, Izmir, Turkey
^c Department of Radiodiagnostics, Dokuz Eylül University Medical School, Izmir, Turkey
^d Department of Cardiovascular Surgery, Dokuz Eylül University Medical School, Izmir, Turkey

Accepted for publication April 8, 2003.

^* Address reprint requests to Dr anli, Gogus Cerrahisi AD, Dokuz Eylül Tip Fak Hastanesi, 35340 Balcova, Izmir, Turkey
e-mail: aydin.sanli{at}deu.edu.tr

	Abstract

Top Abstract Introduction Comment References

 
Bronchogenic cysts occur as a result of a developmental fault during the division and budding of the tracheobronchial tree in the embryonic period. They are usually located in the mediastinum and intrapulmonary regions. They may cause serious respiratory distress in childhood; they present less commonly in adults and often present with a variety of symptoms, including recurrent infections. We report the case of an adult female patient who presented with recurrent pulmonary infections and swelling of the neck because of a bronchogenic cyst that was unusually located in the cervical region.

	Introduction

Top Abstract Introduction Comment References

 
During the third week of gestation, the trachea develops from a ventral diverticulum budding from the foregt. Abnormal division during this period and developmental faults of the budding tracheobronchial tree can lead to the formation of bronchogenic cysts [1].

The localization of cysts may be either in the lungs or in the mediastinum. Mediastinal bronchogenic cysts may result from anomalies in the fifth week of development, whereas intrapulmonary bronchogenic cysts result from anomalies in the sixth week. Mediastinal bronchogenic cysts are seen in the inferior paratracheal, subcarinal, hilar, and paraesophageal locations. They are encountered with increased prevalence in males and on the right side of the midline. Cysts that arise later during gestation are located distally on the bronchial tree and are imbedded within the parenchyma. Histologically, the wall is a layered structure; this relates to its origin from the tracheobronchial tract [1].

Bronchogenic cysts are most often symptomatic in childhood. Although they are rarely observed in adults, they do show themselves with chest pain in the retrosternal area, dysphagia, cough, fever, hemoptysis, and recurrent infections [2, 3]. We report the case of a bronchogenic cyst in an unusual localization in an adult.

A 48-year-old woman had a cervical air cyst detected by computerized tomography (CT) while undergoing investigation for symptoms including swelling of the right side of the neck, a feeling of "hang out" during swallowing, and frequent pulmonary infections. The CT examination revealed a 2-cm cyst in the right paratracheal region beginning at the level of C7 and ending at the level of T1 (Fig 1). The cyst consisted of multiple components. On bronchoscopy there was no fistula between the trachea and the cyst.

View larger version (113K): [in this window] [in a new window]   Fig 1. (Left) Preoperative computed tomographic (CT) image showing the bronchogenic cyst. (Right) Postoperative CT image showing no residual cyst.

View larger version (132K): [in this window] [in a new window]   Fig 2. Resected bronchogenic cyst.

On pathologic examination, the cyst measured 2.8 x 1.5 x 0.9 cm and was divided into 2 sections. The wall was layered with pseudostratified ciliated columnar epithelium. Dense mononuclear cell infiltration and mucous gland formation in the walls of the cyst were also seen. The diagnosis was bronchial cyst.

	Comment

Top Abstract Introduction Comment References

 
Bronchogenic cysts are rare congenital lesions with a reported prevalence of 1 in 42,000 to 1 in 68,000 [4]. In a series of 2,163 mediastinal lesions, 72 (3.3%) were found to be due to bronchogenic cysts [5].

Fifty percent of cysts are located in the posterior mediastinum, 14% in the superior mediastinum, and 35% in the pericarinal area [6]. Isolated case reports describe parapancreatic, hiatal, and diaphragmatic pericardial cysts [1]. Localization in the cervical area, as in our case, is rare and suggests a developmental anomaly that had taken place before the fifth week of development. The differential diagnosis includes tracheal diverticular formations, lymphoepithelial cyst, laryngocele, pharyngocele, Zenker diverticula, apical pulmonary hernia, thyroid adenoma, and thyroid cyst [1, 7, 8].

Bronchogenic cysts are observed on chest roentgenogram and CT as smoothly bordered, oval, or round lesions with a homogeneous density or air-liquid level. They are generally not connected to the airways [1]. Connections between bronchogenic cysts and the tracheobronchial system or esophagus are rarely observed and usually occur with intrapulmonary cysts. When an air-liquid level is observed, these patients may present with recurrent infections [6, 9]. Although recurrent infection was one of the presenting symptoms in our patient and, furthermore, on cross-sectional CT imaging there was a suggestion of a connection of the cyst and tracheobronchial system, no connection was found during operation. CT imaging can be misleading when it suggests a connection between the cyst and the tracheobronchial system. For this reason, it is important to use thin intervals between scans.

In the presence of these symptoms and with the risk of malignant transformation, complete excision is the preferred surgical management. This is usually achieved through a thoracotomy [4–6]. Transbronchial or mediastinoscopic needle aspiration can be helpful for diagnosis and decompression of the cysts; however, complete resection cannot be implied, and requires reexcision. Video-assisted thoracic surgery should be used with caution, and conversion to open operation should be performed in case of dense adhesions with the bronchial wall, the esophagus, or the pericardium. In our case, the cyst was carefully resected from the paratracheal region and completely removed with a cervical approach. Great care was taken to avoid recurrent nerve trauma, and no complication such as dysphonia was observed after the operation.

	References

Top Abstract Introduction Comment References

 

1. Barsotti P., Chatzimichalis A., Massard G., Wihlm J.M. Cervical bronchogenic cyst mimicking thyroid adenoma. Eur J Cardiothorac Surg 1998;13:612-614.
2. St Georges R., Deslauriers J., Duranceau A., et al. Clinical spectrum of bronchogenic cysts of the mediastinum and lung in the adult. Ann Thorac Surg 1991;52:6-13.[Abstract]
3. Pujary K., Pujary P., Shetty R., Hazarika P., Rao L. Congenital cervical bronchogenic cyst. Int J Pediatr Otorhinolaryngol 2001;57:145-148.[Medline]
4. Coselli M.P., de polyi P., Bloss R.S., Diaz R.F., Fitzgerald J.B. Bronchogenic cysts above and below the diaphragm: report of eight cases. Ann Thorac Surg 1987;44:491-494.[Abstract]
5. Lyerly H.K., Sabiston D.C., Jr Primary neoplasms and cysts of the mediastinum. In: Fishman A.P., ed. . Pulmonary diseases and disorders, 2nd ed. New York: McGraw-Hill, 1988:2090-2105.
6. Durancerau A.C.H., Deslauriers J. Foregut cysts of the mediastinum in the adult. In: Shields T.W., ed. Mediastinal surgery. Philadelphia: Lea & Febiger, 1991:305-313.
7. Mo Goo J., Gi Im J., Mo Ahn J., et al. Right paratracheal air cysts in the thoracic inlet: clinic and radiologic significance. AJR Am J Roentgenol 1999;173:65-70.[Abstract/Free Full Text]
8. Shimizu J., Kawaura Y., Tatsuzawa Y., Maeda K., Suzuki S. Cervical bronchogenic cyst that presented as a thyroid cyst. Eur J Cardiothorac Surg 2000;166:659-661.
9. Djamouri F., Le Pimpec Barthes F., Pheulpin G., Grignet J.P., Carnot F., Riquet M. Air-filled cysts of tracheal origin: nosological problems and actual frequency. Rev Mal Respir 2002;19:523-526.[Medline]

This article has been cited by other articles:

				R. Ramos-Izquierdo, I. Escobar-Campuzano, R. Llatjos-Sanuy, and J. Moya-Amoros Syncope and Facial Blushing Due to Giant Intrapulmonary Bronchogenic Cyst Asian Cardiovasc Thorac Ann, January 1, 2009; 17(1): 73 - 75. [Abstract] [Full Text] [PDF]

				Cystic Swelling Overlying the Upper Sternum in a Teenager--Diagnosis Arch Dermatol, September 1, 2006; 142(9): 1221 - 1226. [Full Text] [PDF]

				X. Wei, A. Omo, T. Pan, J. Li, L. Liu, and M. Hu Left Ventricular Bronchogenic Cyst Ann. Thorac. Surg., April 1, 2006; 81(4): e13 - e15. [Abstract] [Full Text] [PDF]

This Article Abstract Full Text (PDF) Alert me when this article is cited Alert me if a correction is posted Citation Map Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Add to Personal Folders Download to citation manager Author home page(s): Erdem Silistreli Ünal Açikel Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Sanli, A. Articles by Açikel, U. Search for Related Content PubMed PubMed Citation Articles by Sanli, A. Articles by Açikel, U. Related Collections Trachea and bronchi