Syncope and Facial Blushing Due to Giant Intrapulmonary Bronchogenic Cyst
Ricard Ramos-Izquierdo, PhD,
Ignacio Escobar-Campuzano, MD,
Roger Llatjos-Sanuy, MD1,
Juan Moya-Amorós, PhD
Department of Thoracic Surgery
1 Department of Pathology, Bellvitge University Hospital, Barcelona, Spain
Ricard Ramos-Izquierdo, PhD, Tel: + 34 93 335 7011, Fax: + 34 93 260 7983, Email: ricardramos{at}ub.edu, Cirugía Torácica, Hospital Universitari de Bellvitge, Feixa Llarga s/n, 08907 – L’Hospitalet de Llobregat (Barcelona), Spain.
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ABSTRACT
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A 43-year-old man presented with dizziness, head instability, and facial reddening, always in relation to body posture and without fever or systemic manifestations. Chest radiography revealed a large cavity with an air-fluid level in the right upper hemithorax. A right upper lobectomy was performed to remove a large bronchogenic cyst. The presentation with cardiac but no respiratory symptoms is uncommon but should be considered in the differential diagnosis of patients with intrathoracic cysts.
Key Words: Bronchogenic Cyst • Blushing • Pneumonectomy • Syncope
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INTRODUCTION
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Bronchogenic cysts are congenital anomalies with a prevalence of 1 in 42,000 to 1 in 68,000, located mainly in the mediastinum or lung parenchyma. Embryologically, the tracheobronchial tree originates from an anterior diverticulum of the primitive digestive tube. Abnormal division of this diverticulum may give rise to congenital anomalies affecting the entire tracheobronchial tree. Although the symptoms and presentation are highly diverse, most patients exhibit recurring clinical manifestations, but the disorder may constitute an incidental radiological finding.1,2
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CASE REPORT
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A 43-year-old man reported to the emergency service due to 4 self-limiting episodes of dizziness, head instability, syncope, and facial blushing, always in relation to his body posture and without associated fever or systemic manifestations. He had smoked 2 packets of cigarettes per day since 16 years of age, and had suffered hepatitis A in childhood. The physical examination revealed blood pressure of 120/80 mm Hg, heart rate 70 beats · min–1 and baseline O2 saturation of 98%. His axillary temperature was 36.7°C. Cardiopulmonary auscultation revealed only slightly diminished tones on the right side. The electrocardiogram showed sinus rhythm without alterations in repolarization, while the posteroanterior chest radiograph revealed a large cavity with well-defined walls and an air-fluid level, located in the right upper hemithorax, without pleural effusion or significant contralateral lesions (Figure 1
). Complete blood count, biochemistry, and coagulation findings were normal, except for minimal elevations of glutamic pyruvate transaminase and gamma glutamyl transferase. Bronchoscopy proved normal, and chest computed tomography confirmed the presence of a right upper lobe lesion measuring 10 cm in diameter with well-defined walls and associated multiple bullae in the apical segment, without any detectable mediastinal adenopathies (Figure 2). Lung function tests showed: tidal flow, 3,610 mL (86.8% of predicted); forced expiratory volume in 1st sec, 2,560 mL (74.3%); and forced expiratory volume in 1st sec/tidal flow, 70.9% (83.7%). With the diagnostic sign of an air-fluid level in the right upper lobe, a right lateral thoracotomy was performed, revealing abundant vascularized pleuropulmonary adhesions affecting the entire lung apex, and an enlarged right upper lobe with a cystic lesion measuring 14 cm in diameter in the lower portion of the anterior segment, in intimate contact with the superior vena cava. Following careful pneumolysis, a homolateral mediastinal retraction was observed, and a right upper lobectomy was performed after freeing the superior vena cava and right phrenic nerve, both of which were partially enveloped by the cyst. Careful hemostasia was carried out, with the placement of human fibrinogen and thrombin in the extrapleural zones with the most damaged surfaces, and in areas of lung parenchyma with air loss. The postoperative course was uneventful, with good oral tolerance and removal of the chest drain 72 hours after the operation. Discharge was decided on the following day after confirming normal radiological findings. Histology of the resected material revealed a right upper lobe measuring 22 x 12 x 9 cm, with apical bullae and a large thin-walled cyst with a smooth internal surface, containing a gelatinous substance of mucoid appearance. The maximum diameter of the lesion was 15 cm, at its lowermost portion. The findings were compatible with a giant bronchogenic lung cyst.
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Figure 1. Posteroanterior radiograph showing a large cavity with well-defined walls and an air-fluid level, with no pleural effusion or significant contralateral lesions.
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Figure 2. Computed tomography confirming a right upper lobe lesion with well-defined walls and multiple bullae in the apical segment.
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DISCUSSION
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Bronchogenic cysts are infrequent congenital lesions that constitute incidental radiological findings in 50%–80% of cases. The most common manifestations are fever, cough and pleuritic pain, although there have been reports of massive hemoptysis and pneumothorax.3,4 In our case, recurrent dizziness and head instability, associated with facial reddening, could have been related to superior vena caval compression by the cyst, according to the body posture of the patient.
Although such lesions are usually located in the mediastinum or lung parenchyma, they can also be found at the cervical, pericardial or even abdominal level, depending on the embryological period in which the cyst develops.5 Radiologically, intrapulmonary bronchogenic cysts can manifest in different ways, depending on whether there is communication with the tracheobronchial tree. In the absence of such communication, typical findings are a homogeneous nodule or mass, and the differential diagnosis includes granulomas, hamartomas, and even congenital vascular alterations. In the presence of a communication with the central airways, as in our patient, we typically observe air-fluid levels, requiring a differential diagnosis with hydatid cysts, infected bullae, and even cavitary malignancies. In rare instances, associated calcifications may be observed, suggesting adenopathies, endothoracic goiter, or aneurysms.
Plain chest radiographs and computed tomography typically suggest the diagnosis, but confirmation must be provided by surgery. Transbronchial puncture and/or mediastinoscopy may be useful as decompressive management and diagnostic options, although they are not systematically used.6 Surgical treatment is mandatory in the presence of associated clinical manifestations, and it is normally complicated by pericystic adhesions (as in our patient); as a result, extrapleural dissection is often necessary. Endoscopic surgery can be used with good lung collapse and minimal adhesions, although such circumstances are rarely found in disorders of this kind.7 Lobar or segmental resection is carried out, depending on the size of the lesion. In our case, the abundant pericystic fibrosis and large size of the lesion made it necessary to perform a lobectomy. Intrapulmonary bronchogenic cysts are infrequent and mostly asymptomatic congenital lesions that require careful surgical management in view of the risk of relapse when only partial resection is carried out.
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REFERENCES
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- St-Georges R, Deslauriers J, Duranceau A, Vaillancourt R, Deschamps C, Beauchamp G, et al. Clinical spectrum of bronchogenic cysts of the mediastinum and lung in the adult. Ann Thorac Surg 1991;52:6–13.[Abstract]
- Patel SR, Meeker DP, Biscotti CV, Kirby TJ, Rice TW. Presentations and management of bronchogenic cysts in the adult. Chest 1994;106:79–85.[Medline]
- Lardinois D, Gugger M, Ris HB. Bronchogenic cyst of the lower lobe associated with severe hemoptysis. Eur J Cardiothorac Surg 1999;16:382–3.[Abstract/Free Full Text]
- Matzinger MA, Matzinger FR, Sachs HJ. Intrapulmonary bronchogenic cyst: spontaneous pneumothorax as the presenting symptom. AJR 1992;58:987–8.
- Akto
u S, Yuncu G, Halilçolar H, Ermete S, Buduneli T. Bronchogenic cysts: clinicopathological presentation and treatment. Eur Respir J 1996;9:2017–21.[Abstract]
- Sanli A, Önen A, Ceylan E, Yilmaz E, Silistreli E, Acikel U. A case of bronchogenic cyst in a rare location. Ann Thorac Surg 2004;77:1093–4.[Abstract/Free Full Text]
- Yim AP, Lam HC, Ho JK. Thoracoscopic lobectomy for an infected intrapulmonary bronchogenic cyst. Surg Endosc 1996; 10:439–40.[Medline]
Asian Cardiovasc Thorac Ann 2009;
17:73-75
© 2009 by SAGE Publications
DOI: 10.1177/0218492309102509
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ABSTRACT
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