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Large Bronchogenic Cyst: Diagnosis and Surgical Management
CASE REPORTS
Case Reports
Large Bronchogenic Cyst: Diagnosis and Surgical Management Igor E. Konstantinov, MD, PhD a , Pankaj Saxena, MCh, DNB a,∗ , Rohan vanden Driesen, FRANZCR b and Mark A.J. Newman, FRACS a a
Department of Cardiothoracic Surgery, Sir Charles Gairdner Hospital, Nedlands, WA 6009, Australia b Department of Radiology, Sir Charles Gairdner Hospital, Nedlands, WA, Australia
Herein we describe a patient with an unusually large bronchogenic cyst. The removal of the cyst was performed through a mini-thoracotomy and facilitated by video-assisted thoracic surgery (VATS). (Heart, Lung and Circulation 2008;17:146–166) © 2006 Australasian Society of Cardiac and Thoracic Surgeons and the Cardiac Society of Australia and New Zealand. Published by Elsevier Inc. All rights reserved. Keywords. VATS; Bronchogenic cyst
Introduction
C
ongenital cysts of the mediastinum are uncommon and thought to arise from the primitive foregut. Bronchogenic cysts are the most common of mediastinal cysts.1 Although bronchogenic cysts are mainly asymptomatic in adults, some may become complicated by infection, bronchial fistula, pneumothorax or undergo malignant transformation.2–4 Thus, surgery is generally recommended in asymptomatic patients to prevent these complications.2–5
Case Report A 39-year-old male bodybuilder using anabolic steroids presented with severe chest pain radiating to his back that started acutely during strenuous weight lifting. Computerised tomography (CT) scan was done (Figs. 1 and 2). There was a small aneurysm of the sixth posterior intercostal artery on the right side with a localised haematoma that was most likely responsible for the adjacent nerve compression and pain (Figs. 1A, C and 2). More impressively, however, there was an incidental finding of a large posterior mediastinal mass that measured 4.6 cm × 7.0 cm × 9 cm in dimension (Fig. 1). The mass was adjacent to the carina, left atrium, descending aorta and appeared cystic with a well-defined capsule (Fig. 1A and B). Three-dimensional CT reconstruction further delineated the precise anatomy of the cyst and the adjacent structures (Fig. 2A and B, for interactive CT reconstructed images visit http://www.rohansplace.com/cyst/cyst.htm).
On further questioning the patient recalled that he had a mediastinal cyst aspiration under radiological guidance 16 years ago. He also noted increasing shortness of breath over the past two months during training. Prompt pain relief was achieved, the patient was discharged home and scheduled for CT guided cyst aspiration. CT guided needle aspiration, however, failed to obtain any fluid despite appropriate needle placement inside the mass, presumably because the high viscosity of the fluid. The patient continued to experience shortness of breath with exercise that interfered with his strenuous training regime. In view of his symptoms, potential risk of infection and malignant transformation, surgery was recommended. The cyst was visualised by video-assisted thoracic surgery (VATS) approach. However, further dissection by VATS alone appeared hazardous due to dense peri-cystic adhesions to the posterior pericardium, left main bronchus and major vessels. A muscle sparing mini-thoracotomy was performed to facilitate further dissection. After initial dissection of the cyst from the lung, oesophagus and both caval veins, the cyst was opened and 200 ml of thick mucus was evacuated. This facilitated completion of the dissection from the roof of the left atrium, left main bronchus and descending aorta. The cyst was resected. There was no air leak and flexible bronchoscopy demonstrated normal bronchial tree. Two chest tubes were placed with one of them being introduced behind the left atrium into the left pleural cavity. Post-operative course was uneventful. Pathology examination of the specimen demonstrated benign bronchogenic cyst lined by pseudostratified, ciliated epithelium.
Received 14 May 2006; received in revised form 11 October 2006; accepted 13 October 2006; available online 8 March 2007
Comment
∗ Corresponding author. Tel.: +61 8 9346 3333; fax: +61 8 934 62 344. E-mail address: [email protected] (P. Saxena).
To the best of our knowledge, this is the largest uncomplicated bronchogenic cyst described in the English
© 2006 Australasian Society of Cardiac and Thoracic Surgeons and the Cardiac Society of Australia and New Zealand. Published by Elsevier Inc. All rights reserved.
1443-9506/04/$30.00 doi:10.1016/j.hlc.2006.10.010
Case Reports
Figure 1. A ruptured aneurysm of the intercostal artery (A and C) with a large cyst adjacent to the left atrium (A, B, D), carina (D), and descending aorta (A and C).
Figure 2. Three-dimensional reconstruction of the cyst demonstrates its proximity to the aorta, trachea, main bronchi, and intercostal artery aneurysm in postero-lateral (A) and posterior views (B). Interactive images are available at http://www.rohansplace.com/cyst/cyst.htm.
147 CASE REPORTS
Heart, Lung and Circulation 2008;17:146–166
148
Case Reports
CASE REPORTS
Heart, Lung and Circulation 2008;17:146–166
language literature. It is possible that the use of anabolic steroids contributed to significant growth of the cyst. Three-dimensional CT reconstruction of the cyst was helpful in precise delineation of the adjacent anatomy. Although bronchogenic cysts may produce symptoms in up to two thirds of patients,1–5 the patient described herein was asymptomatic at rest or with moderate exercise. It was of concern that the unusually large size of the cyst and the patient’s strenuous training program may predispose to the rupture of the cyst. The combination of VATS with muscle sparing mini-thoracotomy ensured safe surgery and provided good cosmetic result. We recommend that resection of any bronchogenic cyst following failed needle aspiration is mandatory to relieve symptoms and prevent potential complications especially infection.
References 1. Takeda S, Miyoshi S, Minami M, Ohta M, Masaoka A, Matsuda H. Clinical spectrum of mediastinal cysts. Chest 2003;124:125–32. 2. St George R, Deslauriers J, Duranceau A, Vaillancourt R, Deschamps C, Beauchamps G, et al. Clinical spectrum of bronchogenic cysts of the mediastinum and lung in the adult. Ann Thorac Surg 1991;52:6–13. 3. Cuypers P, De Leyn P, Capelle L, Verougstraete L, Demedts M, Deneffe G. Bronchogenic cysts: a review of 20 cases. Eur J Cardiothorac Surg 1996;10:393–6. 4. Jakopovic M, Slobodnjak Z, Krizanac S, Samarzija M. Large cell carcinoma arising in bronchogenic cyst. J Thorac Cardiovasc Surg 2005;130:610–2. 5. Kanemitsu Y, Nakayama H, Asamura H, Kondon H, Tsuchiya R, Naruke T. Clinical features and management of bronchogenic cysts: report of 17 cases. Surg Today 1999;29:1201–5.
Coronary Artery Fistula with Coronary Sinus Obstruction and Retrograde Drainage Neerod K. Jha, MD a,∗ , Fahad AlHabshan, MD b , Mansour AlMutairi, MD b , Michael Godman, MD b and Hani K. Najm, MD a b
a Department of Cardiac Surgery, King Fahad Hospital, Riyadh, Saudi Arabia Department of Pediatric Cardiology, King Fahad Hospital, Riyadh, Saudi Arabia
Coronary artery to coronary sinus fistula (CACSF) in association with stenosis of coronary sinus (CS) ostium and retrograde drainage via persistent left superior vena cava (LSVC) is an extremely rare anomaly presenting in the neonatal period. We report herein a 2-day-old boy with similar anomalies, who presented to us with congestive heart failure and underwent surgical repair. Echocardiography including Doppler ultrasound was a non-invasive, quick and accurate tool to diagnose each abnormality satisfactorily. This anomaly has clinical, physiological and surgical implications due to presence of altered flow pattern and variable anatomy. Therefore, such malformations need a careful assessment, preferably by non-invasive diagnostic tools, and appropriate management. (Heart, Lung and Circulation 2008;17:146–166) © 2006 Australasian Society of Cardiac and Thoracic Surgeons and the Cardiac Society of Australia and New Zealand. Published by Elsevier Inc. All rights reserved. Keywords. Coronary; Sinus; Fistula; Stenosis; Artery
C
oronary artery to coronary sinus fistula (CACSF) in association with stenosis of coronary sinus (CS) ostium and retrograde drainage via persistent left superior vena cava (LSVC) is an extremely rare cardiovascular anomaly and has not been reported in a neonate earlier. The recognition of this anomaly has clinical, physiological and surgical implications especially in the presence of altered flow pattern and variable anatomical features. Therefore, for successful outcome, each component of this malformation needs careful assessment and management.
Received 16 September 2006; received in revised form 10 October 2006; accepted 20 October 2006; available online 12 March 2007 ∗
Corresponding author at: Cardiac Sciences, King Fahad Hospital, Box-22490, Riyadh, Saudi Arabia. Tel.: +966 1 2379259. E-mail address: nk [email protected] (N.K. Jha).
Case Report A full-term, 2-day-old boy, presented with severe congestive heart failure (CHF) and required ventilatory support. His weight was 3 kg. There was a grade 3/6 continuous murmur at the left sternal border and hepatomegaly. Chest X-ray showed cardiomegaly. However, electrocardiogram (ECG) was unremarkable. A 2D echocardiogram revealed situs solitus, dilated right atrium, right ventricle and the CS (Fig. 1). There was a large fistulous communication and flow (left-toright shunt) between the circumflex coronary artery (CA) and the CS. However, we did not quantify the shunt. The CS ostium was narrow with a high velocity flow into the right atrium and a peak instantaneous gradient of 70 mmHg across it (Fig. 1). On colour flow Doppler ultrasound, there was turbulence at the junction of the fistula to
© 2006 Australasian Society of Cardiac and Thoracic Surgeons and the Cardiac Society of Australia and New Zealand. Published by Elsevier Inc. All rights reserved.
1443-9506/04/$30.00 doi:10.1016/j.hlc.2006.10.014
Case Reports
Large Bronchogenic Cyst: Diagnosis and Surgical Management Igor E. Konstantinov, MD, PhD a , Pankaj Saxena, MCh, DNB a,∗ , Rohan vanden Driesen, FRANZCR b and Mark A.J. Newman, FRACS a a
Department of Cardiothoracic Surgery, Sir Charles Gairdner Hospital, Nedlands, WA 6009, Australia b Department of Radiology, Sir Charles Gairdner Hospital, Nedlands, WA, Australia
Herein we describe a patient with an unusually large bronchogenic cyst. The removal of the cyst was performed through a mini-thoracotomy and facilitated by video-assisted thoracic surgery (VATS). (Heart, Lung and Circulation 2008;17:146–166) © 2006 Australasian Society of Cardiac and Thoracic Surgeons and the Cardiac Society of Australia and New Zealand. Published by Elsevier Inc. All rights reserved. Keywords. VATS; Bronchogenic cyst
Introduction
C
ongenital cysts of the mediastinum are uncommon and thought to arise from the primitive foregut. Bronchogenic cysts are the most common of mediastinal cysts.1 Although bronchogenic cysts are mainly asymptomatic in adults, some may become complicated by infection, bronchial fistula, pneumothorax or undergo malignant transformation.2–4 Thus, surgery is generally recommended in asymptomatic patients to prevent these complications.2–5
Case Report A 39-year-old male bodybuilder using anabolic steroids presented with severe chest pain radiating to his back that started acutely during strenuous weight lifting. Computerised tomography (CT) scan was done (Figs. 1 and 2). There was a small aneurysm of the sixth posterior intercostal artery on the right side with a localised haematoma that was most likely responsible for the adjacent nerve compression and pain (Figs. 1A, C and 2). More impressively, however, there was an incidental finding of a large posterior mediastinal mass that measured 4.6 cm × 7.0 cm × 9 cm in dimension (Fig. 1). The mass was adjacent to the carina, left atrium, descending aorta and appeared cystic with a well-defined capsule (Fig. 1A and B). Three-dimensional CT reconstruction further delineated the precise anatomy of the cyst and the adjacent structures (Fig. 2A and B, for interactive CT reconstructed images visit http://www.rohansplace.com/cyst/cyst.htm).
On further questioning the patient recalled that he had a mediastinal cyst aspiration under radiological guidance 16 years ago. He also noted increasing shortness of breath over the past two months during training. Prompt pain relief was achieved, the patient was discharged home and scheduled for CT guided cyst aspiration. CT guided needle aspiration, however, failed to obtain any fluid despite appropriate needle placement inside the mass, presumably because the high viscosity of the fluid. The patient continued to experience shortness of breath with exercise that interfered with his strenuous training regime. In view of his symptoms, potential risk of infection and malignant transformation, surgery was recommended. The cyst was visualised by video-assisted thoracic surgery (VATS) approach. However, further dissection by VATS alone appeared hazardous due to dense peri-cystic adhesions to the posterior pericardium, left main bronchus and major vessels. A muscle sparing mini-thoracotomy was performed to facilitate further dissection. After initial dissection of the cyst from the lung, oesophagus and both caval veins, the cyst was opened and 200 ml of thick mucus was evacuated. This facilitated completion of the dissection from the roof of the left atrium, left main bronchus and descending aorta. The cyst was resected. There was no air leak and flexible bronchoscopy demonstrated normal bronchial tree. Two chest tubes were placed with one of them being introduced behind the left atrium into the left pleural cavity. Post-operative course was uneventful. Pathology examination of the specimen demonstrated benign bronchogenic cyst lined by pseudostratified, ciliated epithelium.
Received 14 May 2006; received in revised form 11 October 2006; accepted 13 October 2006; available online 8 March 2007
Comment
∗ Corresponding author. Tel.: +61 8 9346 3333; fax: +61 8 934 62 344. E-mail address: [email protected] (P. Saxena).
To the best of our knowledge, this is the largest uncomplicated bronchogenic cyst described in the English
© 2006 Australasian Society of Cardiac and Thoracic Surgeons and the Cardiac Society of Australia and New Zealand. Published by Elsevier Inc. All rights reserved.
1443-9506/04/$30.00 doi:10.1016/j.hlc.2006.10.010
Case Reports
Figure 1. A ruptured aneurysm of the intercostal artery (A and C) with a large cyst adjacent to the left atrium (A, B, D), carina (D), and descending aorta (A and C).
Figure 2. Three-dimensional reconstruction of the cyst demonstrates its proximity to the aorta, trachea, main bronchi, and intercostal artery aneurysm in postero-lateral (A) and posterior views (B). Interactive images are available at http://www.rohansplace.com/cyst/cyst.htm.
147 CASE REPORTS
Heart, Lung and Circulation 2008;17:146–166
148
Case Reports
CASE REPORTS
Heart, Lung and Circulation 2008;17:146–166
language literature. It is possible that the use of anabolic steroids contributed to significant growth of the cyst. Three-dimensional CT reconstruction of the cyst was helpful in precise delineation of the adjacent anatomy. Although bronchogenic cysts may produce symptoms in up to two thirds of patients,1–5 the patient described herein was asymptomatic at rest or with moderate exercise. It was of concern that the unusually large size of the cyst and the patient’s strenuous training program may predispose to the rupture of the cyst. The combination of VATS with muscle sparing mini-thoracotomy ensured safe surgery and provided good cosmetic result. We recommend that resection of any bronchogenic cyst following failed needle aspiration is mandatory to relieve symptoms and prevent potential complications especially infection.
References 1. Takeda S, Miyoshi S, Minami M, Ohta M, Masaoka A, Matsuda H. Clinical spectrum of mediastinal cysts. Chest 2003;124:125–32. 2. St George R, Deslauriers J, Duranceau A, Vaillancourt R, Deschamps C, Beauchamps G, et al. Clinical spectrum of bronchogenic cysts of the mediastinum and lung in the adult. Ann Thorac Surg 1991;52:6–13. 3. Cuypers P, De Leyn P, Capelle L, Verougstraete L, Demedts M, Deneffe G. Bronchogenic cysts: a review of 20 cases. Eur J Cardiothorac Surg 1996;10:393–6. 4. Jakopovic M, Slobodnjak Z, Krizanac S, Samarzija M. Large cell carcinoma arising in bronchogenic cyst. J Thorac Cardiovasc Surg 2005;130:610–2. 5. Kanemitsu Y, Nakayama H, Asamura H, Kondon H, Tsuchiya R, Naruke T. Clinical features and management of bronchogenic cysts: report of 17 cases. Surg Today 1999;29:1201–5.
Coronary Artery Fistula with Coronary Sinus Obstruction and Retrograde Drainage Neerod K. Jha, MD a,∗ , Fahad AlHabshan, MD b , Mansour AlMutairi, MD b , Michael Godman, MD b and Hani K. Najm, MD a b
a Department of Cardiac Surgery, King Fahad Hospital, Riyadh, Saudi Arabia Department of Pediatric Cardiology, King Fahad Hospital, Riyadh, Saudi Arabia
Coronary artery to coronary sinus fistula (CACSF) in association with stenosis of coronary sinus (CS) ostium and retrograde drainage via persistent left superior vena cava (LSVC) is an extremely rare anomaly presenting in the neonatal period. We report herein a 2-day-old boy with similar anomalies, who presented to us with congestive heart failure and underwent surgical repair. Echocardiography including Doppler ultrasound was a non-invasive, quick and accurate tool to diagnose each abnormality satisfactorily. This anomaly has clinical, physiological and surgical implications due to presence of altered flow pattern and variable anatomy. Therefore, such malformations need a careful assessment, preferably by non-invasive diagnostic tools, and appropriate management. (Heart, Lung and Circulation 2008;17:146–166) © 2006 Australasian Society of Cardiac and Thoracic Surgeons and the Cardiac Society of Australia and New Zealand. Published by Elsevier Inc. All rights reserved. Keywords. Coronary; Sinus; Fistula; Stenosis; Artery
C
oronary artery to coronary sinus fistula (CACSF) in association with stenosis of coronary sinus (CS) ostium and retrograde drainage via persistent left superior vena cava (LSVC) is an extremely rare cardiovascular anomaly and has not been reported in a neonate earlier. The recognition of this anomaly has clinical, physiological and surgical implications especially in the presence of altered flow pattern and variable anatomical features. Therefore, for successful outcome, each component of this malformation needs careful assessment and management.
Received 16 September 2006; received in revised form 10 October 2006; accepted 20 October 2006; available online 12 March 2007 ∗
Corresponding author at: Cardiac Sciences, King Fahad Hospital, Box-22490, Riyadh, Saudi Arabia. Tel.: +966 1 2379259. E-mail address: nk [email protected] (N.K. Jha).
Case Report A full-term, 2-day-old boy, presented with severe congestive heart failure (CHF) and required ventilatory support. His weight was 3 kg. There was a grade 3/6 continuous murmur at the left sternal border and hepatomegaly. Chest X-ray showed cardiomegaly. However, electrocardiogram (ECG) was unremarkable. A 2D echocardiogram revealed situs solitus, dilated right atrium, right ventricle and the CS (Fig. 1). There was a large fistulous communication and flow (left-toright shunt) between the circumflex coronary artery (CA) and the CS. However, we did not quantify the shunt. The CS ostium was narrow with a high velocity flow into the right atrium and a peak instantaneous gradient of 70 mmHg across it (Fig. 1). On colour flow Doppler ultrasound, there was turbulence at the junction of the fistula to
© 2006 Australasian Society of Cardiac and Thoracic Surgeons and the Cardiac Society of Australia and New Zealand. Published by Elsevier Inc. All rights reserved.
1443-9506/04/$30.00 doi:10.1016/j.hlc.2006.10.014