Introduction

Gastropleural fistulae form in a variety of pathological conditions. Markowitz and Herter (₁) described three situations that result in GPF formation: 1) when the intrathoracic portion of the stomach is perforated in the setting of esophageal hiatal hernia; 2) as a direct result of trauma, or after diaphragmatic hernia formation; and 3) when the stomach in normal intraabdominal position is perforated, and a resultant subphrenic abscess erodes and eventually perforates the diaphragm. Reports have also documented GPF formation in association with intractable postoperative nausea and vomiting, esophageal surgery, congenital diaphragmatic hernia, gastric operation for obesity, and pulmonary resection (₂,₃,₄,₅).

Case Report

A 36-year-old male presented with foul-smelling drainage and leakage of food debris from and around a chest catheter that had been inserted in a previous procedure 4 years eralier. He also complained of weakness, weight loss, and progressive shortness of breath. The patient's medical history included investigation for progressive dyspnea at age 32, when he had been diagnosed with diaphragmatic perforation and hernia. The perforation was suspected to have originated from a childhood injury, as the patient had sustained blunt abdominal trauma when he was hit by a vehicle at 8 years of age. After diagnosis, the defect was repaired with a grafting operation. One month after this surgery, the patient had been re-hospitalized due to high fever and weakness. Graft infection and empyema were detected. He was treated with systemic antibiotics, and an intrapleural catheter was inserted to drain the chest cavity.

For the 4 years prior to presentation at our center, there had been intractable drainage from the catheter and the patient had been re-checked multiple times at different hospitals. On physical examination at our hospital, the patient's vital signs were normal. Chest inspection revealed a thoracic catheter inserted through the ninth intercostal space in the left posterior chest wall. There was also hyperemia and drainage around the chest tube, and a scar from a previous thoracotomy nearby. On auscultation, bowel sounds were heard beneath the left scapula. Routine blood test results were normal. Gram staining of the chest drainage material showed gram-negative bacilli and neutrophils, and a culture of the material grew Group A beta-hemolytic Streptococcus. Culture of a skin swab from the catheter site grew Pseudomonas aeruginosa and Klebsiella pneumoniae. A posteroanterior chest x-ray showed homogeneous infiltrate in the left lower lung field near the costophrenic angle (Figure 1). Thoracic computerized tomography (CT) showed that part of the stomach was located within the thorax (Figure 2). Esophagogastroscopy demonstrated a defect in the gastric fundus that was compatible with fistulous communication. Esophagogastroduodenal radiography after barium swallow demonstrated a GPF and volvulus (Figure 3). Evaluation of the same region with ultrasonography confirmed the presence of a fistulous tract.

Figure 1

Figure 1: Posteroanterior chest x-ray showed homogeneous infiltrate in the left lower lung field near the costophrenic angle.

Figure 2

Figure 2: Thoracic computerized tomography showed that part of the stomach was located within the thorax.

Figure 3

Figure 3: Esophagogastroduodenal radiography after barium swallow demonstrated gastropleural fistula and volvulus.

The patient was admitted to the ward and oral nutrition was discontinued. Nasogastric decompression and total parenteral nutrition were initiated, and empirical antibiotic therapy was administered for the empyema. The chest tube drainage continued for 2 more days, but it stopped on day 3 and the tube was then removed.

Repeat barium-swallow study performed after 1 week of hospitalization showed no leakage from the fistula site. At this stage, parenteral nutrition was discontinued and the patient returned to oral feeding. He was discharged after 17 days in hospital, and the thoracic surgery division at our center planned a future diaphragmatic hernia operation.

A re-check at 1 week post-discharge confirmed that the fistula was completely closed. Six months later, the patient remained asymptomatic and there was still no leakage from the GPF, indicating sustained complete closure. Unfortunately, the patient refused to undergo surgical repair of the diaphragm defect, and he was later lost to follow-up.

Discussion

In the present case, the history included diaphragmatic perforation caused by blunt abdominal trauma, as well as unsuccessful surgical repair of the diaphragm that led to postoperative empyema and graft infection. After the patient sustained the initial trauma that we suspect caused the diaphragmatic perforation, more than two decades passed without any serious medical problems. At 24 years post-trauma, he complained of progressive dyspnea, but there was no evidence of GPF when the diaphragmatic hernia was diagnosed. After the initial attempt at surgical repair of the diaphragm, the patient developed serious infection, with a high fever and stupor. The diagnosis of empyema and the chest tube drainage that occurred suggested that a fistula had formed between the stomach and the pleura postoperatively. Four years later, thoracic CT performed at our center showed that part of the stomach had shifted to an intrathoracic location. This indicated that the initial surgical correction of the diaphragmatic perforation had failed. As in the case reported by Mussi and co-workers, we believe that our patient's GPF may have formed from erosion of the gastric wall due to empyema (₆).

Although GPF is known to cause severe complications such as tension pneumothorax in some patients (₇), our case shows that this type of lesion can also manifest with insidious onset and chronic complaints. This case also demonstrates that discontinuation of oral feeding may relieve symptoms and promote spontaneous closure of the fistulous tract prior to surgery. Close monitoring of our patient and the fact that he was in stable condition enabled us to take a more conservative management approach. Nevertheless, to prevent fatal complications, surgical repair should always be performed as soon as possible in patients with GPF due to perforated diaphragm.

Correspondence to

Özgür Karacan MD, Baskent University Faculty of Medicine, Department of Pulmonary Medicine. Fevzi Çakmak Caddesi, 10. sokak, No:45 06490, Bahçelievler, Ankara, Turkey. Phone: + 90 312 212 04 34/125 Fax: +90 312 223 73 33 E-mail: ozgurkaracan@yahoo.com