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Bronchogenic cyst presenting as superior vena caval syndrome: A case report Article in Indian Journal of Thoracic and Cardiovascular Surgery · May 2004 DOI: 10.1007/s12055-004-0056-1
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Bronchogenic cyst presenting as superior vena caval syndrome: A case report Lalit Mohan Joshi, MS, Sushil Kumar Singh, M.Ch., Mohd. Salman Siddiqui, M.Ch., Sanjay Pandey, MS, Girish Chandra, M.Ch., Anita Mehrotra MD, Shekhar Tandon M.Ch.
Department of Thoracic & Cardiovascular Surgery, King George Medical University (Upgraded King George's Medical College, Lucknow) Superior vena cava (SVC) obstruction most often is c a u s e d by i n t r a t h o r a c i c m a l i g n a n t process e.g. Bronchogenic c a r c i n o m a a n d l y m p h o m a s . The Commonest cause of benign superior vena cava (SVC) obstruction is fibrosing mediastinitis6, which accounts for 10% of overall cases. Here we report an unusual presentation of right apical bronchogenic cyst4 presenting as a superior vena cava syndrome, which was successfully operated at our institution.
of upper lobe compressing superior vena cava. (Fig.2). Sputum test for Mycobacterium, and ELISA test for Echinococcus were negative, and Fine Needle Aspiration Cytology (FNAC) was inconclusive.
Case Report A 50-year-male was referred to our department from TB and Chest Hospital of the same institution (K.G's. Medical University, Lucknow, India). Patient presented to us with signs and symptoms of SVC obstruction. It was of gradual onset and had a progressive course. He h a d taken a n t i t u b e r c u l a r regimen for one year in the past, and was referred to us with the provisional diagnosis of post tubercular loculated empyema with SVC syndrome. At the time of admission, he was dyspnoeic but hemodynamically stable. There was no history of trauma, or exposure to radiation in the past. On examination, the patient had diffuse swelling of the face, neck, upper limbs and anterior chest wall, with dilated superficial veins over the chest and neck. Harvey's sign indicated blood flow from cranial to caudal direction, above the umbilicus. Chest R a d i o g r a p h s h o w e d r o u n d e d r a d i o o p a q u e shadow at right upper zone (Fig. 1). Computerised axial tomography demonstrated encysted hyperdense mass of the size of 8 x 10 cm. at right apicoposterior segment Address for correspondence: Dr. LM Joshi Sr. Resident, Room No 110, Resident Hostel Dept. of CTVS K.G.M.U., Lucknow Email:
[email protected] @ IJTCVS 097091 3420 20604/16 R e c e i v e d - 6/2/04; R e v i e w
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Fig. 1. Chest X-ray PA view
Fig. 2. CT Thorax
Surgical Approach P a t i e n t was i n t u b a t e d w i t h a d o u b l e l u m e n endotracheal tube. A Right posterolateral thoracotomy was done and the pleural cavity was entered through the bed of 4th rib. Mass was present at the apex and was densely adherent to superior mediastinal structures producing compression of superior vena cava. On aspiration, the contents of the cyst were mucoid. Its wall was incised and contents were evacuated. Cyst wall was separated by blunt and sharp dissection, relieving compression from mediastinal structures and Central venous pressure immediately came down to 11 mm Hg from 40 m m Hg. After complete excision of the cyst and proper hemostasis, chest was closed with an intercostal drainage tube. Postoperative course was uneventful, producing marked reduction in facial oedema and regression of d i s t e n d e d veins over the chest wall. Final histopathological report revealed it, as a bronchogenic cyst. (Fig. 3) and the patient is symptom free in the 8 months of follow up.
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Fig. 3. H i s t o p a t h o l o g i c a l Slide
Discussion
Superior v e n a cava obstruction is not a c o m m o n clinical p r e s e n t a t i o n . The clinical f e a t u r e s of this s y n d r o m e have been discussed b y O c h s n e r and others. These patients usually have swelling of the face, neck and u p p e r extremities, dyspnoea, orthopnea and cough. The c o m m o n physical finding constitutes distended and tortuous veins over the chest wall, neck and head along with plethora and cyanosis over face and lips. Other s y m p t o m s m a y be headache, changes in mental status, vertigo, seizure, dysphagia, somnolence and dysphorea. Patients with s y m p t o m s suggesting of cerebral o d e m a or laryngeal o d e m a (hoarseness, stridor) h a v e p o o r prognosis. P a r i s h et al 7 found an average survival time of o n l y 6 weeks, if these patients d i d not u n d e r g o d e c o m p r e s s i o n . Most of the r e p o r t e d cases of SVC s y n d r o m e h a v e b e e n d e s c r i b e d in p a t i e n t s w i t h malignant disease, although benign S conditions have also been mentioned. H u n t e r 1 (1757), first reported SVC compression b y aortic aneurysm. Various reported cases of benign SVC obstructions w e r e r e v i e w e d f r o m English literature. T h e y w e r e c l a s s i f i e d in t e r m s of I M e d i a s t i n i t i s , IIMediastinal tumours, IIIVascular pathology, IVCardiac l e s i o n s , V p u l m o n a r y l e s i o n s , VITraumatic a n d VIIMiscellaneous c a u s e s ( B e h c e t ' s s y n d r o m e , Retroperitoneal fluid, Bilateral clavicular osteomyelitis, Silicosis a n d Sarcoidosis). Mediastinitis c a u s e d b y various granulomatous processes is most c o m m o n cause of the benign SVC syndrome. One case, of bronchogenic cyst 4 causing SVC s y n d r o m e has also been described in
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the literature. A n d therefore our case is probably the second in the literature. On clinical b a c k g r o u n d it is v e r y difficult to rule out benign nature of this syndrome; but some minor features can help, e.g. insidious onset and slow progression of s y m p t o m s in m o n t h s and years c o m p a r e d to that of malignant type, w h e r e the progress of the disease is rapid in weeks. Only sometimes the s y m p t o m s m a y appear rapidly, even in benign type, due to s u p e r a d d e d infection and enlargement of the lesion, requiring urgent intervention. The treatment of SVC s y n d r o m e depends on cause and prognosis. Supportive measures such as elevation of the head, supplemental oxygen, diuretics and steroids m a y i m p r o v e s y m p t o m s . S t r a t e g i e s for r e l i e f of malignant obstruction m a y involve radiation therapy, c h e m o t h e r a p y or surgery. Surgical a p p r o a c h to SVC obstruction, includes decompression, t h r o m b e c t o m y and long term anticoagulation, caval replacement and bypass. The use of percutaneously placed endovascular stents has been described for limited indications. Considering all above facts surgical decompression was the best possible option for this case. References 1. H u n t e r W: The history of a n a n e u r y s m of the aorta w i t h s o m e r e m a r k s o n a n e u r y s m in general. Med Obs Sac Phys Cand 1: 323,
1757. 2. BalloonHC, FrancisBF:Consequencesof variation in mediasthlal pressure, mediasthlal and subcutaneous emphysema. Arch Surg 1929; 19: 1627-59. 3. McIntire FT, Syko EM Jr. Obstruction of the superior vena cava. A review of the literature and report of two personal cases. Ann Intern Med 1949;30: 92540. 4. Gtunpeni Rairunohan,Herbert W. Berger,Fouad Lajam,Wilfrido J. Buhain : Superior Vena Cava Syndrome caused by bronchogeixiccyst. Chest 1975;68: 599-601. 5. Mahajan V, Strhnlan V, Van Ordstran HC, Loop FD. Superior vena cava syndrome secondary to aortic disease. Chest 68: 1975; 32: 32-35. 6. Dhlis DE, Payne WS, Bernatz PE et al. Mediastinal granttlomas and fibroshlg mediastinitis. Chest 1975; 75: 320. 7. Parish JM, Marschke RF, Dinis DE, Lee RE : Etiologic considerations in superior vena cava syndrome. Mayo Clin Proc 1981; 56: 407-13. 8. Abner A : Approach to the patient who presents with superior venacava obstruction. Chest 1993;103: 3945.
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