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CASE REPORT
Year : 2016  |  Volume : 4  |  Issue : 1  |  Page : 37-40

Tubercular pleuropericardial cyst presenting with symptomatic pulmonary stenosis


1 Department of Cardiothoracic and Vascular Surgery, JN Medical College, Aligarh Muslim University, Aligarh, Uttar Pradesh, India
2 Department of Obstetrics and Gynaecology, JN Medical College, Aligarh Muslim University, Aligarh, Uttar Pradesh, India

Date of Web Publication5-May-2016

Correspondence Address:
Sayyed Ehtesham Hussain Naqvi
Department of Cardiothoracic and Vascular Surgery, JN Medical College, Aligarh Muslim University, Aligarh, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2320-3846.181814

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  Abstract 

A 24-year-old male presented to us with complaints of dyspnea on exertion and cyanosis. On examination, he had systolic murmur over pulmonary area. X-ray and contrast-enhanced computed tomography chest confirmed a large cystic pleuropericardial mass compressing the pulmonary artery which was excised successfully by midline sternotomy. Microscopic examination of purulent content of cyst revealed acid-fast Mycobacterium bacilli. This report is unusual on account that symptomatic and clinically evident pulmonary stenosis from pleuropericardial cyst is rare. Second, it is one of the rare presentations of tuberculosis.

Keywords: Cyanosis, pulmonary stenosis, systolic murmur, tubercular pleuropericardial cyst


How to cite this article:
Naqvi SE, Beg MH, Ali E. Tubercular pleuropericardial cyst presenting with symptomatic pulmonary stenosis. Saudi Surg J 2016;4:37-40

How to cite this URL:
Naqvi SE, Beg MH, Ali E. Tubercular pleuropericardial cyst presenting with symptomatic pulmonary stenosis. Saudi Surg J [serial online] 2016 [cited 2022 Jan 28];4:37-40. Available from: https://www.saudisurgj.org/text.asp?2016/4/1/37/181814


  Introduction Top


Differential diagnosis of cystic mediastinal masses is varied and includes bronchogenic cysts, neurenteric cysts, esophageal duplication cysts, inflammatory pleuropericardial cysts, thymic cysts, cystic teratomas, lymphangiomas, meningoceles, Thoracic duct cysts and cystic degeneration of mediastinal tumours. [1] Clinical presentation of these mediastinal cysts is as varied as their etiology and largely depend on their etiology and location. Tuberculosis is still a rampant disease in developing South East Asian countries. Manifestation of tuberculosis is many a times nonclassical causing diagnostic dilemmas. [2] We hereby report a case of large tubercular pleuropericardial cyst presenting with features of pulmonary stenosis in a young male.


  Case Report Top


A 24-year-old male presented to us with complaints of dyspnea on exertion and occasional cyanosis observed over the lips. Dyspnea was exertional and grade NYHA II. It was not associated with history of chest pain, palpitations, syncope, and cough or sputum production. Cyanosis was observed by patient's attendants when patient used to arise in the morning after sleeping in the supine position. There was no cyanosis or clubbing at the time of examination. In the examination of cardiovascular system, P2 was loud with Grade 4/5 murmur in early and midpart of systole best heard in the left second intercoastal space lateral to sternal border.

A provisional diagnosis of pulmonary stenosis was made on the basis of history and clinical examination. Patient was planned for investigations including X-ray chest and transthoracic echocardiography. X-ray chest of the patient revealed an abnormal convexity of the left heart border overlying the aortic and pulmonary artery markings with suspicion of an anterior mediastinal mass over the great vessel area. There were reduced vascular markings over the left lung field suggestive of pulmonary oligemia [Figure 1]. Transthoracic echocardiography confirmed the diagnosis of pulmonary stenosis. Color Doppler echocardiography revealed a turbulent flow across the pulmonary artery. Peak gradients of 54 mm Hg and a mean gradient of 32 mm Hg were observed across left pulmonary artery origin. Right pulmonary artery had a peak gradient of 22 mm Hg and mean of 12 mm Hg at its origin.
Figure 1: X-ray chest posteroanterior view with abnormal convexity of the left heart border overlying aorta and pulmonary artery

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Patient was taken for contrast-enhanced computed tomography (CT) scan of the chest for evaluation of mediastinal mass. CT scan revealed a large pleuropericardial cyst lying in the anterior mediastinum with significant stenosis of main pulmonary artery. There was no evidence of septations, calcification, or daughter cyst within the cystic mass. There was no evidence of contrast leak. Few subcentrimetric mediastinal lymph nodes were observed. There was no evidence of pleural thickening [Figure 2] and [Figure 3]. Routine blood investigations revealed normal total and differential cell counts with raised erythrocyte sedimentation rate of 57 mm/h.
Figure 2: Contrast-enhanced computed tomography chest coronal section showing large cystic mass with compression of main and left pulmonary artery

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Figure 3: Contrast-enhanced computed tomography chest transverse section showing large cystic mass occupying anterior and apical mediastinum overlying pulmonary artery

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After routine preoperative preparations, the patient was taken for excision of the pleuropericardial cyst. Midline sternotomy approach was used. Sternotomy revealed a large tense pericardium-based cyst which got ruptured during sternotomy. Contents were thick and purulent. There was no evidence of daughter cysts [Figure 4]. Cystic cavity had no connections with lung, airway, or any major vessel. Base of the cyst was adhered to main pulmonary and left pulmonary artery which could be dissected out with difficulty [Figure 5] and [Figure 6].
Figure 4: Showing ruptured cyst with purulent contents poststernotomy

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Figure 5: Peroperative photograph showing walls of the cyst and its relation to pulmonary artery

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Figure 6: Peroperative photograph showing pericardial defect after cyst excision with visualization of pulmonary artery

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Examination of the purulent cystic content revealed the presence of acid-fast Mycobacterium bacilli. Microscopic histopathological specimen revealed features of chronic inflammation along with multiple granulomas. There were no atypical cells suggestive of malignancy.

Patient had a smooth postoperative course and was discharged on the 4 th day following surgery on antitubercular therapy. Postoperative chest X-ray depicted clear heart and lung shadows.


  Discussion Top


Cystic lesions of the mediastinum comprise around 12-18% of all primary mediastinal tumors. [3] Cystic mediastinal masses may have their origin from pleura, pericardium, tracheobronchial tree, aberrant gastrointestinal tissue, neurogenic tissue, lymphoid tissue, or thymic glandular tissue. [4] Esme et al. in his study of 32 mediastinal cysts reported 12 cysts to be bronchogenic, 9 pericardial, 7 thymic, and 2 enteric cysts, together with 2 cystic teratomas. [5] Cystic mediastinal masses are particularly significant because of the difficulty in reaching particular diagnosis. They simulate multiple lesions, both benign and malignant.

Congenital pleuropericardial cysts are benign cyst of mesothelial origin. They arise as a result of persistence of mesenchymal lacunae that normally fuse to form the pericardial sac. [6] These cysts are usually unilocular with clear fluid content. They typically present in anterior cardiophrenic angle, more often on the right side than the left. [7] Inflammatory cysts can be caused by rheumatic pericarditis, pyogenic and tubercular pericardial infections, and trauma including surgery. [8]

Majority of patients with pleuropericardial cyst are free of symptoms, and these cysts are incidentally detected during X-ray or CT chest. Symptoms if present may be in the form of chest pain, fever, cough, breathlessness, or hoarseness of voice in case of recurrent laryngeal compression. [5]

However, some of the rare presentations of pleuropericardial cyst have been cardiac tamponade, [9] rupture of the cyst, [10] obstruction of the right ventricular outflow, [7] superior vena cava syndrome, [11] erosion of the cyst into right ventricular wall, [12] congestive heart failures, [13] atrial fibrillation, pericarditis, obstruction of the bronchi, and sudden death. [14] Ng et al. have reported a case similar to our with pleuropericardial cyst causing right ventricular outflow tract (RVOT) stenosis; however, the patient did not have clinically evident RVOT stenosis with complaints of cyanosis and pulmonary systolic murmur as was present in our case. [7]

Transthoracic echocardiography, CT, and magnetic resonance imaging (MRI) are the methods of choice for the diagnosis of pericardial cysts. [15] Echocardiography has a role being noninvasive and being accurate in diagnosing cardiac compression. Its accuracy is limited as compared to CT and MRI for diagnosing the nature of cyst preoperatively. [16]

Management of a pleuropericardial cyst is mainly decided by its nature, size, and predominant complaints of the patient. Asymptomatic patients can be followed with clinical and radiological monitoring with possibility of spontaneous resolution. [17] Large cyst with symptoms of local compression, cysts with systemic manifestations in case of infective origin, complicated cysts, and cysts with suspicion of malignant changes require prompt surgical intervention. [5] The various treatment modalities include percutaneous aspiration of cyst, ethanol sclerosis, surgical resection, or video-assisted thoracoscopic surgery. Surgical excision becomes mandatory when pericardial cyst cause ventilator and/or hemodynamic impairment. [18]


  Conclusion Top


Pleuropericardial cyst is one of the rare presentations of tuberculosis. Cystic mediastinal masses may have varied presentations. They may present with features of pulmonary stenosis simulating congenital cardiac diseases and causing diagnostic dilemma as was in our case. If preoperatively diagnosis of tubercular pleuropericardial cyst is confirmed one can start antitubercular therapy followed by surgical excision after percutaneous aspiration of cyst. In cases where cyst morphology appears complex on CT or when diagnosis of infective etiology is not confirmed surgical excision of the cyst is required for same.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Oldham HN Jr. Mediastinal tumors and cysts. Ann Thorac Surg 1971;11:246-75.  Back to cited text no. 1
    
2.
Dinesh K, Kothari P, Adivarekar P, Ravikumar, Dipali R, Kulkarni B. Atypical presentation of tuberculosis. Indian J Tuberc 2006;53:223-6.  Back to cited text no. 2
    
3.
Takeda S, Miyoshi S, Minami M, Ohta M, Masaoka A, Matsuda H. Clinical spectrum of mediastinal cysts. Chest 2003;124:125-32.  Back to cited text no. 3
    
4.
McAllister HA Jr. Primary tumors and cysts of the heart and pericardium. Curr Probl Cardiol 1979;4:1-51.  Back to cited text no. 4
    
5.
Esme H, Eren S, Sezer M, Solak O. Primary mediastinal cysts: Clinical evaluation and surgical results of 32 cases. Tex Heart Inst J 2011;38:371-4.  Back to cited text no. 5
    
6.
Lillie WI, McDonald JR, Clagett OT. Pericardial celomic cysts and pericardial diverticula; a concept of etiology and report of cases. J Thorac Surg 1950;20:494-504.  Back to cited text no. 6
    
7.
Ng AF, Olak J. Pericardial cyst causing right ventricular outflow tract obstruction. Ann Thorac Surg 1997;63:1147-8.  Back to cited text no. 7
    
8.
Sharifi-Mood B, Alavi Naini R, Eazadi M. Cystic tubercular pericarditis a rare form. J Res Med Sci 2005;10:236-8.  Back to cited text no. 8
    
9.
Shiraishi I, Yamagishi M, Kawakita A, Yamamoto Y, Hamaoka K. Acute cardiac tamponade caused by massive hemorrhage from pericardial cyst. Circulation 2000;101:E196-7.  Back to cited text no. 9
    
10.
King JF, Crosby I, Pugh D, Reed W. Rupture of pericardial cyst. Chest 1971;60:611-2.  Back to cited text no. 10
    
11.
Kaul P, Javangula K, Farook SA. Massive benign pericardial cyst presenting with simultaneous superior vena cava and middle lobe syndromes. J Cardiothorac Surg 2008;3:32.  Back to cited text no. 11
    
12.
Chopra PS, Duke DJ, Pellett JR, Rahko PS. Pericardial cyst with partial erosion of the right ventricular wall. Ann Thorac Surg 1991;51:840-1.  Back to cited text no. 12
    
13.
Borges AC, Gellert K, Dietel M, Baumann G, Witt C. Acute right-sided heart failure due to hemorrhage into a pericardial cyst. Ann Thorac Surg 1997;63:845-7.  Back to cited text no. 13
    
14.
Komodromos T, Lieb D, Baraboutis J. Unusual presentation of a pericardial cyst. Heart Vessels 2004;19:49-51.  Back to cited text no. 14
    
15.
Abad C, Rey A, Feijóo J, Gonzalez G, Martín-Suarez J. Pericardial cyst. Surgical resection in two symptomatic cases. J Cardiovasc Surg (Torino) 1996;37:199-202.  Back to cited text no. 15
    
16.
Pepi M, Muratori M. Echocardiography in the diagnosis and management of pericardial disease. J Cardiovasc Med (Hagerstown) 2006;7:533-44.  Back to cited text no. 16
    
17.
Abbey AM, Flores RM. Spontaneous resolution of a pericardial cyst. Ann Thorac Cardiovasc Surg 2010;16:55-6.  Back to cited text no. 17
    
18.
Michelotto E, Tarantino N, Ostuni V, Pedote P, Colonna P, Guglielmi R. An uncommon pericardial cyst in the central mediastinum: Incremental diagnosis with contrast-enhanced three-dimensional transesophageal echocardiography. J Cardiovasc Echography 2013;23:106-10.  Back to cited text no. 18
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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]



 

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