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J Thorac Cardiovasc Surg 2006;132:972-974
© 2006 The American Association for Thoracic Surgery
Brief Communication |
a Pathology Institute, IRCCS Policlinico San Matteo, University of Pavia, Pavia, Italy.
b Cardiology, IRCCS Policlinico San Matteo, University of Pavia, Pavia, Italy.
c C Dubost Transplant Centre, IRCCS Policlinico San Matteo, University of Pavia, Pavia, Italy.
d Transplant Research Area, Centre for Inherited Cardiovascular Diseases, IRCCS Policlinico San Matteo, University of Pavia, Pavia, Italy.
Received for publication May 31, 2006; accepted for publication June 6, 2006. * Address for reprints: Eloisa Arbustini, MD, Transplant Research Area, IRCCS Policlinico San Matteo, Piazzale Golgi 2, 27100 Pavia, Italy. (Email: e.arbustini{at}smatteo.pv.it).
Intracardiac bronchogenic cysts are rare, with only 5 cases reported to date: 2 in the atrial septum,1,2
1 in the left atrium,3 and 2 in the right ventricle.4,5 They are identified by asymptomatic occasional findings or nonspecific symptoms, mostly in the second to fourth decades.3
Bronchogenic cysts are believed to represent a localized portion of the tracheobronchial tree that separates from the normal airways during the branching process and does not undergo further development. They develop between the 26th and the 40th day of intrauterine life, during the most active period of airway development.3 The timing of the abnormal budding may determine the location: earlier in the mediastinum and later within lung tissue, which are the 2 most common locations.3 Intracardiac location can be explained with a more precocious abnormal budding, approximately 21 days after fertilization, when the cardiac primordial tube is near the foregut or primitive tracheobronchial tree.2
Other noncardiac, atypical locations are cutaneous, retroperitoneal, cervical, intradiaphragmatic, intrapericardial, intraspinal, and intrapleural.2
We report a case of a bronchogenic cyst located in the wall of a large aneurysm of the pars membranacea septi triggering premature ventricular complex (PVC) and sinus tachycardia.
A 5-year-old girl was referred to us for the occasional finding of PVC first recorded in 2001 in the third month of life, and confirmed in further controls. Two years later, a Holter monitoring documented sinus tachycardia with several PVCs. An echocardiographic study indicated a possible aneurysm of the left Valsalva sinus. The left atrium and ventricle were moderately enlarged. In 2004, a further echocardiogram described the lesion as a "subaortic aneurysm of the left ventricle."
On November 2005 the girl presented with normal cardiac tones and apparently free pauses; the chest roentgenogram showed a slightly enlarged cardiac shadow. The electrocardiogram confirmed sinus rhythm with respiratory arrhythmia and several PVCs. Transthoracic echocardiography documented an "aneurysm of the pars membranacea septi" with internal diameter of 2 cm (Figure 1, A). Tricuspid, mitral, aortic, and pulmonary valves showed normal function. The need for an angio-computed tomography scan was discussed, but it was decided that echocardiography was sufficient for surgery. Indication for surgical excision was based on the potential link between the aneurysm and the PVC, and on the potential risk of embolization. The girl underwent successful surgery and is doing well 1 year after the resection. Sinus tachycardia and PVC were no longer recorded after surgery, suggesting a cause and effect relationship between the aneurysm and the arrhythmias.
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The bronchogenic cyst was a non-expected finding at the pathologic study of the removed aneurysm that constituted the major lesion. This case documents the possible presence of a bronchogenic cyst within the wall of an aneurysm of the pars membranacea septi.
Prior reported cases are different from the present one because the bronchogenic cysts were large enough to be detected as cysts at the echocardiography or computed tomography scan study.1-5 In our patient the lesion diagnosed with echocardiography was the aneurysm. A small cystic space was suspected (Figure 1, A) but did not influence the echocardiography diagnosis and the surgical decision.
A bronchogenic cyst is a benign lesion. Surgical treatment is controversial,2 especially in patients without clinical symptoms. However, the benignant nature of the cyst is unknown before pathologic examination. Moreover, the cyst may progressively enlarge1 because of mucus accumulation and inflammation of the wall. Analogously to other cardiac masses, it may trigger complications such as arrhythmias, conduction disturbances, obstruction of blood flow, or embolization.2-4
Footnotes
Supported by research grants "Ricerche Finalizzate e Correnti" IRCCS Policlinico San Matteo of Pavia, Italy.
References
This article has been cited by other articles:
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  V. Martinez-Mateo, M. A. Arias, R. Juarez-Tosina, and L. Rodriguez-Padial Permanent third-degree atrioventricular block as clinical presentation of an intracardiac bronchogenic cyst Europace, May 1, 2008; 10(5): 638 - 640. [Abstract] [Full Text] [PDF]
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