Case report

Concurrent cysts of the mediastinum, pleura and neck

Wael I. Awada, Andrew G. Nicholsonb, Peter Goldstrawa,*

aDepartment of Thoracic Surgery, Royal Brompton Hospital, Sydney Street, London SW3 6NP, UKbDepartment of Histopathology, Royal Brompton Hospital, Sydney Street, London SW3 6NP, UK

Received 14 May 2001; received in revised form 9 July 2001; accepted 12 July 2001

Abstract

A 14-year-old male was found to have a mediastinal mass on chest radiograph. Chest computed tomography scans showed a cystic lesion

behind the left main bronchus. Magnetic resonance imaging revealed additional cystic lesions in the left chest and root of the neck. He

underwent excision of mediastinal mass and a pleural cyst. The neck lesion was presumed to be a cystic hygroma. Histological examination

of the two lesions resected showed them to be a foregut cyst and a benign mesothelial cyst. We know of no other report of concurrent

multicystic lesions in the chest and neck and hypothesize that these cysts may have a common embryonic origin. q 2001 Elsevier Science

B.V. All rights reserved.

Keywords: Mediastinum; Foregut cyst; Mesothelial cyst

1. Introduction

Benign cystic lesions in the mediastinum can be conge-

nital or acquired. Primary cysts of the mediastinum include

bronchogenic cysts, enteric or duplication cysts, mesothelial

cysts, thymic cysts and thoracic duct cysts. Bronchogenic

and enteric cysts are closely related as the respiratory tree

and oesophagus arise at a common origin from the primitive

foregut. We report a case of multiple cysts in the chest and

neck and hypothesize that these cysts may have a common

embryonic origin.

2. Case report

A 14-year-old, asymptomatic, male non-smoker was

found to have a large cystic mass in the posterior mediasti-

num on routine chest radiograph. Chest computed tomogra-

phy (CT) scan revealed a well-circumscribed lesion of low

attenuation measuring approximately 6.5 cm in diameter

posterior to the left main bronchus. The lesion did not

enhance with intra-venous contrast and was compatible

with a bronchogenic cyst. The rest of the lung fields

appeared clear.

Magnetic resonance imaging (MRI) showed the large

mediastinal mass seen on the CT scan and another smaller

cystic lesion involving the left pleura (Fig. 1). In addition,

there was a lobulated polycystic mass in the suprasternal

notch consistent with cystic hygroma (Fig. 2).

It was felt that the mediastinal lesions should be excised,

to provide a definitive histological diagnosis and to prevent

future complications by compression or infection. The neck

lesion was not considered a threat and the patient was

advised to seek medical help if the situation changed.

Rigid bronchoscopy revealed compression of the poster-

ior wall of the left main bronchus. We judged that VATS

would not have allowed us to adequately inspect the left

hemithorax, and so the patient underwent a left postero-

lateral, muscle-sparing thoracotomy. There was a 7 £ 7

cm, thin-walled tense cyst arising in the posterior mediasti-

num. The cyst was adherent to the posterior surface of the

lower lobe, the inferior pulmonary vein and the pulmonary

artery. It appeared to be part of the posterior wall of the main

bronchus and herniated into the mediastinum. The lower

oesophagus was stretched across its posterior surface.

The main cyst was mobilized from the oesophagus, the

posterior aspect of the lung and pulmonary vessels, and

dissected from the bronchus without breaching its lumen.

A further thin-walled, uniloculated cyst was attached to the

main cyst. The lung appeared normal. Smaller cystic lesions

were excised from the surface of the lingula, as seen on

MRI, and the inferior pulmonary ligament. All cysts

contained thin clear fluid.

The post-operative course was uncomplicated. The

patient was discharged on day 5 and was well and asympto-

European Journal of Cardio-thoracic Surgery 20 (2001) 861–863

1010-7940/01/$ - see front matter q 2001 Elsevier Science B.V. All rights reserved.

PII: S1010-7940(01)00918-6

www.elsevier.com/locate/ejcts

* Corresponding author. Tel.: 144-2073-518559; fax: 144-2073-

518560.

E-mail address: p.goldstraw@rbh.nthames.nhs.uk (P. Goldstraw).

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matic at the 4 month follow-up. Histological examination of

the mediastinal cyst wall showed it to be composed of

connective tissue and smooth muscle, lined by benign

respiratory epithelium. There were mucus glands within

the wall, but no cartilage. The features were those of a

simple congenital foregut cyst. The small cysts excised

from the lingula and inferior pulmonary ligament had thin

fibrous walls lined by cytologically bland cells that stained

for calretinin, indicating benign mesothelial cells.

3. Discussion

A CT scan may suggest a specific diagnosis of mediast-

inal cysts by way of careful evaluation of anatomic relation-

ships and convincing evidence that the mass is cystic.

Occasionally, bronchogenic cysts may be a source of confu-

sion because the fluid density may be relatively high due to a

high protein content. In situations where the CT scans are

equivocal, MRI can be used as a problem-solving tool.

Enteric cysts are discovered more commonly in child-

hood. Generally, they lie in the middle or posterior medias-

tinum adjacent to the oesophagus, or even embedded in its

muscular wall. They are lined by stratified squamous epithe-

lium or by gastric or intestinal mucosa which distinguishes

them from bronchogenic cysts, which are lined with ciliated,

columnar epithelium and may contain mucus glands or

cartilage in their walls. Bronchogenic cysts account for

approximately 60% of mediastinal cysts. They are often

located posterior to the carina but may be located more

distally in association with the bronchi. Rarely, they may

communicate with the tracheobronchial tree. They are

usually asymptomatic incidental findings, unless they

compress adjacent structures. In children, bronchogenic

cysts may present with severe respiratory distress and

even respiratory failure [1]. Overall, complications are

reported to occur in up to 26% of patients [2]. Surgery is

indicated because of the unpredictable risk of haemorrhage,

infection, enlargement with associated pressure symptoms

or malignant change [2].

Mesothelial cysts comprise a variety of cysts that have

been reported as pleuropericardial, pleural and simple

mesothelial cysts. They are unilocular cysts whose walls

consist of a single layer of flattened endothelial cells with

an underlying connective tissue stroma, and are most often

incidental radiological findings, as in our case.

Cervical cystic hygromas are believed to occur as a result

of the failure of establishment of appropriate connection to

the normally present lymphatic channels. They are usually

encountered at birth or in early infancy. Very few hygromas

extend into the mediastinum [3]. The cervical cyst in our

case, which was small and found incidentally, was consis-

tent with a cystic hygroma, although it is possible that it

W.I. Awad et al. / European Journal of Cardio-thoracic Surgery 20 (2001) 861–863862

Fig. 1. MRI scan of the chest demonstrating a large multilocular mediastinal

cyst and a pleural cyst (shown by an arrow) in the left hemithorax.

Fig. 2. MRI scan of the neck demonstrating a lobulated polycystic mass in

the suprasternal notch.

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represented a thymic cyst. These can present as either cervi-

cal or anterior mediastinal masses, and are frequently symp-

tomatic [4]. Hendrickson and colleagues [4] encountered 14

patients with congenital thymic cysts, ranging in age from 2

weeks to 16 years, and reported that seven patients had

cervical masses, five had mediastinal masses and two had

both sites involved. We elected not to surgically pursue the

cervical cysts in our patient in view of the high probability

that this was a cystic hygroma.

Yamauchi and colleagues [5] describe a rare case of

double mediastinal cysts (one in the anterior and one in

the posterior mediastinum) in a 60-year-old male, which

were confirmed as a thymic cyst and a thoracic duct cyst,

respectively. Their patient was managed successfully by

video-assisted thoracic surgery. Indeed, selected patients

with mediastinal cysts can be managed safely and effec-

tively by thoracoscopic means [6]. We elected to perform

thoracotomy in our case and this decision seemed to be

indicated by the difficulty in resecting the lesion from the

back of the left main bronchus, oesophagus and pulmonary

vessels.

We have reported a unique case of multiple cysts in a 14-

year-old boy. The concurrent occurrence of multiple cysts in

the neck and mediastinum may reflect a common embryonic

origin. We recommend complete excision in most instances

to confirm the diagnosis, relieve any symptoms and to

prevent complications.

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