Clinical Profile:

A 27 year old man, came to the hospital with complaints of right sided chest pain since two months. •The pain was continuous and dull aching in nature. It was followed by breathlessness on exertion. There was increased respiratory activity with a dull note on percussion over the right hemithorax.

Radiological findings:

The frontal chest radiograph shows increased radiolucency in the right hemithorax with a fluid level. There is collapse of the underlying lung. There is no shift of mediastinum. The side hole of the intercostal drain is just within the chest wall (Figure A)

A diagnosis of right hydropneumothorax was made The intercostal drain tube had an output of two to three litres of frank pus per day. It was noticed that the drain had pus with an admixture on fluids consumed by the patient.

Hence a esophagogram was advised by the referring physician. (Figure B)

Meanwhile the patient was also started on anti tubercular treatment due to the exudative nature of the effusion.

The esophagram performed with barium demonstrates a linear fistulous tract from the right wall of the lower third of the esophagus extending into the right pleural cavity. This confirmed the diagnosis of esophageal -pleural fistula.

A gastroduodenoscopy (Figure C) confirmed the diagnosis made on the barium esophagogram. The esophageal- pleural fistula was seen at the lower end of the esophagus with pus extruding out of the lower end.

Final diagnosis :

Esophageal perforation with esophagopleural fistula .

At this time, endoscopy showed a diverticulum with a fistula opening 2 cm above the gastroesophageal junction. The edges of the fistula were ablated . The cardiac septal occluder device (LA size 26mm and RA size 22mm and of of width 4mm )was placed into the fistulous tract.

Post procedure , the patient complained of increased respiratory activity and dyspnea on walking .

The Intercostal tube drain output had decreased but was around 500 ml per day.

A CT scan (Figure E &F) the thorax was performed.

The CT scan showed a right pleural effusion with an intercostal drain in place. The cardiac septal occluder device was seen to have migrated into the right pleural cavity .

The patient is now being managed conservatively and a surgical procedure using VATS( video assisted thoracoscopic surgery) is being planned for retrieval of the device and closure of the fistula.

Discussion:

Esophagopleural fistula is an abnormal communication between the oesophagus and the pleura and is a rare complication following esophageal perforation . Patients with esophagopleural fistula form a very heterogenous group due to the varying etiologies involved. Esophageal injury may be caused by iatrogenic trauma, post pneumonectomy, diseases of the esophagus such as corrosive esophagitis , esophageal ulcers, chronic infections likely tuberculosis ,as a complication of carcinoma esophagus and rarely spontaneously.(1)

The exact site of fistula depends upon the cause of the perforation .

The radiological signs depend on the cause and location of the perforation but more importantly on the integrity of the pleura. If the pleura remains intact, esophageal perforation causes mediastinitis with subsequent rupture of the mediastinal pleura causing hydrothorax and pneumothorax. •If the pleural space is disrupted it will cause an esophagopleural fistula with decompression of the contents in the pleural space .(2)

The clinical diagnosis of esophagopleural fistula is difficult as the clinical symptoms are non specific. However it should be suspected when there is increased output from the intercostal drainage tube particularly if the patient gives an antecedent history of prior surgery or instrumentation of the esophagus.

The radiologist plays a very important role in the diagnosis of this condition as various imaging modalities can be used for diagnosis each modality having its own advantages.

Chest radiograph, barium studies , contrast CT are commonly used for diagnosis.

The chest radiograph can show findings of a pleural effusion or hydropneumothorax.

Barium esophagogram can demonstrate the presence of fistulous tract and help in making the diagnosis as in our case.

CT is often superior to esophagogram for making the diagnosis as it is fast, requires little patient cooperation, and can evaluate both the mediastinum and the pleura. It also can detect very minute quantities of air or contrast leak.(3) . It can also help to make a diagnosis of the underlying etiology.

A cardiac septal occluder device can be deployed for closure of a persistent esophageal fistula.(6) . Although the septal occluder has been conventionally used for closure of Atrial of ventricular septal defects it has many extra cardiac uses as well. It is a self expanding double disc closure device and is composed of nitinol and a polymer which promotes tissue in growth .

Complications include failure of fistula closure due to incomplete opening of the device or migration of the device as in our case. The overall success rate is 75%.

References:

1.Massard G, Ducrocq X, Hentz JG, Kessler R, Dumont P, Wihlm JM, Morand G. Esophagopleural fistula: an early and long-term complication after pneumonectomy. The Annals of thoracic surgery. 1994 Nov 1;58(5):1437-40.

2.Vyas S, Prakash M, Kaman L, Bhardwaj N, Khandelwal N. Spontaneous esophageal-pleural fistula. Lung India: Official Organ of Indian Chest Society. 2011 Oct;28(4):300.

3.Wechsler RJ. CT of esophageal-pleural fistulae. American Journal of Roentgenology. 1986 Nov 1;147(5):907-9.

4.Al-Haddad M, Craig CA, Odell J, Pajaro O, Wallace MB. The use of self-expandable plastic stents for non-malignant esophago-pleural fistulas. Diseases of the Esophagus. 2007 Dec 1;20(6):538-41.

5.Böhm G, Mossdorf A, Klink C, Klinge U, Jansen M, Schumpelick V, Truong S. Treatment algorithm for postoperative upper gastrointestinal fistulas and leaks using combined vicryl plug and fibrin glue. Endoscopy. 2010 Jul;42(07):599-602.

6.De Moura DT, Baptista A, Jirapinyo P, De Moura EG, Thompson C. Role of Cardiac Septal Occluders in the Treatment of Gastrointestinal FIstulas: A Systematic Review. Clinical Endoscopy. 2019 Jul 9.