Volume 4, Issue 2 , Pages 56-58, March 2009
An unusual localization of a bronchogenic cyst: Cervical region—A case report
Article Outline
Summary
Objective: In that report, we present an unusual localization of a bronchogenic cyst which was the low anterior cervical region. Methods: A 4 years old girl admitted to our clinic with a mass located in the low anterior cervical region. In physical examination, there was a 3
cm×3cm, painless, semi-mobile mass in cervical region. Results: Surgical intervention revealed a 3cm×3cm, multilocular infectious fluid filled cyst that was appeared as an abscess. Histopathological diagnosis was bronchogenic cyst. Conclusions: Although the incidence of a bronchogenic cyst is rare, it must be considered in the differential diagnosis of the cervical masses.
Keywords: Bronchogenic cyst, Cervical region, Child
1. Introduction
Bronchogenic cysts are abnormal masses that develop during intrauterine life. The respiratory diverticulum (lung bud) appears as an outgrowth from the foregut at approximately the 4th week of gestation. It then extends caudally to develop into trachea, bronchi, and lungs. Abnormal division during this period and developmental flaws of the budding tracheobronchial tree can lead to the formation of a bronchogenic cyst [1]. Those cysts were filled with a mucoid material lined with a cylindrical cuboidal epithelium. Central or peripherical locations of bronchogenic cysts may be possible. Most common locations of those cysts are cervical and thoracal regions, hilus of bronchi and distal portion of parenchymal bronchi. Seventy percent of those cysts were located in lung parenchyma and rest was located in mediastinum [2]. Remote locations such as cervical region were extremely rare.
Clinical presentation of those cysts differs with age. Bronchogenic cysts are oftenly symptomatic in childhood, initially causing dyspnea or respiratory distress, cough, and bronchopulmonary infections due to compression of the tracheobronchial tree and only 15% present with an inflamed mass. In adults, they are usually asymptomatic and detected by a routine chest X-ray [3].
Preoperative diagnosis of those cysts is very rare. Postoperative diagnosis is made by histopathological examination. Incorrect histopathological diagnosis of those cysts may sometimes be possible.
2. Case report
A 4 years old girl who had a mass in anterior part of lower cervical region since birth was admitted to our clinic because of the mass beginned to enlarge since the last week. The patient had not any other complaint. In physical examination a semimobile, painless mass in 3cm×3cm dimensions was detected. Since the mass had local inflammatory signs, redness and fluctuation, it was considered as an abcess. So it was drained and purulent material was seen. After 1 week antibiotherapy the patient was reevaluated and it was seen that the mass had been persisted even the signs of infection disappeared. All routine laboratory analysis and chest X-ray were normal. In USG examination the mass was reported as an infected lenfadenopathy. All those findings made us to think that the mass was an infected cystic hygroma or an epidermoid cyst. After the diagnostic approaches we operated the patient. Surgical intervention was performed through a low transverse cervical small incision, which revealed a 3cm×3cm, multilocular infectious fluid filled cyst that was appeared as an abscess. The cyst was completely excised from the cervical region. The postoperative period of the patient was uneventful. After 3h operation, the patient was in full oral feeding and she was discharged. In the histopathological examination of the excised cyst with H&E section it was seen that, bronchial epithelium lined the cyst wall which was establishing the diagnosis of a bronchogenic cyst.
3. Discussion
During embryogenesis, the primitive foregut arises. The laryngotracheal groove develops from the ventral wall of the primitive foregut and then forms the trachea and the bilateral bronchial buds. Further budding results in the development of the bronchial trees. It has been hypothesized that bronchogenic cysts originate from abnormal budding [4].
In consideration of embryogenesis, it is very unusual for a bronchial cyst to develop out of the thorax such as in the lower neck. For that reason the preoperative diagnosis of those cysts was nearly impossible. In our case we did not think that the mass was a bronchogenic cyst. We prediagnosed the mass as an infected cystic hygroma or an epidermoid cyst. In literature it was seen that those cysts were misdiagnosed as a branchial cyst, dermoid cyst, teratoma, thyroglossal cyst, epithelial cyst, thymic cyst, neurogenic tumor or retention cyst [5], [8].
Maier classified bronchogenic cysts according to their site of origin: para-tracheal, carinal, hilar, para-esophageal, and atypical (such as diaphragmatic, abdominal, intracutaneous, or subcutaneous, or in the supraclavicular neck area) [6]. In our case unusual localization for a bronchogenic cyst that was in the lower cervical area, suggested a developmental anomaly which occured before the 4th week of gestation.
Clinical presentation of a bronchogenic cyst depends upon the age of patient. In infants and children, they are mostly asymptomatic. However, dyspnea or respiratory distress, cough, and bronchopulmonary infections may be the initial symptoms which are caused by compression on the tracheobronchial tree. In adults the cyst is detected coincidentally by a routine chest X-ray examination since the cyst mostly causes no symptoms. In our case the bronchogenic cyst was infected which was not an expected finding for those cysts. The infected bronchogenic cyst made us to prediagnose mistakenly it as an abcess.
There was no specific diagnostic method for the bronchogenic cyst. Routine laboratory analysis methods are useless for the diagnosis. Routine chest X-ray helps to diagnose most of the bronchogenic cysts, but when the cyst is localized in neck, plain films of the neck appear to have little diagnostic value [7].
Diagnostic methods of USG, CT and MR may be helpful for the localization and relation of bronchogenic cyst with the adjacent tissues. For the definite diagnosis of bronchogenic cyst histopathological examination is necessary. In our case preoperative diagnostic procedures were insufficient and postoperatively histopathological examination of the excised tissue made us to diagnose the bronchogenic cyst. Histopathologic examination of the cyst wall highlighted tissues normally found in the respiratory tract; including cartilage, smooth muscle, mucous glands, and fibrous connective tissue. The epithelium is usually a pseudostratified ciliated columnar type, unless there was previous infection, in which case squamous epithelium may be found [8]. In our case, histopathologic examination confirmed the diagnosis of bronchial cyst. Histology of our excised specimen revealed that the cyst wall was lined by ciliated pseudostratified columnar epithelium with patchy chronic inflammation within the wall (Fig. 1), together with fibrous tissue and occasional microscopic focus of smooth muscle.
Fig. 1.
Ciliated pseudostratified columnar epithelium, lining bronchial cyst wall with mild chronic inflammation in adjacent fibrous tissue. H&E, X200.
The main complications of bronchogenic cysts reported in current literature were infection, symptoms developed due to pressure of the cyst, malignant transformation of the cyst and very rarely lethal air embolism [9], [10], [11]. In our case the bronchogenic cyst was infected.
Surgical excision is both diagnostic and curative, and also eliminates the risk of infection or recurrence. Aspiration of the cyst is an inadequate treatment, because recurrence after this procedure is possible [12].
In conclusion, in differential diagnosis of cervical masses we must consider the bronchogenic cysts. For the diagnosis of bronchogenic cysts rare localizations such as lower cervical region, difficult preoperative diagnosis, mistakes in histopathological examination and possible malignant transformation should not be overlooked.
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PII: S1871-4048(08)00046-4
doi:10.1016/j.pedex.2008.06.003
© 2008 Elsevier Ireland Ltd. All rights reserved.
Volume 4, Issue 2 , Pages 56-58, March 2009
