Empyema resulting from a true colopleural fistula complicating a perforated sigmoid diverticulum

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CASE REPORT PAPAGIANNOPOULOS ET AL EMPYEMAS FROM COLOPLEURAL FISTULAS

Ann Thorac Surg 2004;77:324 – 6

References

CASE REPORTS

1. Lukens JN. The thalassemias and related disorders. In Wintrobe’s clinical hematology. 10th edition. Baltimore: Williams and Wilkins, 1999:1405– 48. 2. Smith PR, Manjoney DL, Teitcher JB, Choi KN, Braverman AS. Massive hemothorax due to intrathoracic extramedullary hematopoiesis in a patient with thalassemia intermedia. Chest 1988;94:658 –60. 3. Kupferschmid JP, Shahian DM, Villanueva AG. Massive hemothorax associated with intrathoracic extramedullary hematopoiesis involving the pleura. Chest 1993;103:974 –5. 4. Bartlett RP, Greipp PR, Tefferi A, Cupps RE, Mullan BP, Trastek VF. Extramedullary hematopoiesis manifesting as a symptomatic pleural effusion. Mayo Clin Proc 1995;70:1161–4. 5. Chu KA, Lai RS, Lee CH, Lu JY, Chang HC, Chiang HT. Intrathoracic extramedullary haematopoiesis complicated by massive haemothorax in alpha-thalassaemia. Thorax 1999;54: 466 –8. Fig 2. Thoracic computed tomographic scan; hyperdense areas within the pleural fluid and haziness of borders of the largest extramedullary mass (arrow), indicative of intrapleural bleeding.

The extramedullary foci consist of hemopoietic cells and adipose elements. They are not circumscribed by a capsule. The mass is extremely vascular and contains little fibrous tissue. This structure renders the mass prone to hemorrage. Our patient represents a case of spontaneous massive hemothorax. Only four similar cases are reported in the literature due to ␤-thalassemia [2], myelofibrosis [3], agnogenic myeloid metaplasia [4], and ␣-thalassemia [5], which were subjected to different treatment. In our patient, despite the rapid pleural fluid reproduction, there was some indication of self-cessation of bleeding (regression of pleural fluid hematocrit values in successive punctures), permitting a more conservative approach. Our report illustrates a rare complication of extramedullary erythropoiesis and, hopefully, will bring about a better understanding of the disease. Namely: (1) Spontaneous bleeding may be a complication of extramedullary erythropoiesis. (2) Most reports face extramedullary erythropoiesis as a benign complication; however, the possibility of such a complication necessitates a reconsideration of this view. (3) In such patients where the hematocrit values of pleural fluid progressively decrease, surgical intervention may not be necessary; 2 of 4 reports in the literature described surgical intervention, whereas the other two did not. (4) All thus far published cases of extramedullary foci bleeding deal with the pleural cavity; the reason is not yet clear, but it may be related to preferential localization or specific tissue structure(s), rendering them vulnerable to rupture due to thoracic respiratory movements. (5) Conservative treatment (transfusions and hydroxyurea) may suffice for the regression of extramedullary erythropoiesis. (6) Whether extramedullary foci represent a contraindication to anticoagulants is a matter of speculation. © 2004 by The Society of Thoracic Surgeons Published by Elsevier Inc

Empyema Resulting From a True Colopleural Fistula Complicating a Perforated Sigmoid Diverticulum Kostas Papagiannopoulos, MMED, Dimitrios Gialvalis, MD, Ibrahim Dodo, MRCS, and Mike J. Darby, MD Department of Cardiothoracic Surgery and Radiology, Leeds General Infirmary, Leeds, United Kingdom

Empyemas developing after traumatic rupture of intraabdominal organs have been previously reported. We report a case of a true nontraumatic colopleural fistula following surgery for spontaneous rupture of a sigmoid diverticulum. The diagnosis was suspected by the presence of an air-containing tract seen in a computerized tomogram of chest and abdomen and was established with a contrast study. The empyema cavity was initially drained, followed by a laparotomy and fistulectomy with primary large bowel anastomosis and loop ileostomy. Although rare, colopleural fistulas present a diagnostic challenge and delayed management can lead to increased morbidity. (Ann Thorac Surg 2004;77:324 – 6) © 2004 by The Society of Thoracic Surgeons

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mpyemas following inflammatory bowel diseases or intestinal trauma could indicate a fistulous connection of bowel with the pleural cavity. Careful evaluation of the pleural fluid, computerized tomograms of chest and abdomen, and contrast studies could provide early, prompt diagnosis leading to appropriate management. A 46-year-old male smoker, with no significant past medical history, presented in the Accident and Emergency Department with an acute abdomen. An emergency laparotomy and Hartmann’s procedure was under-

Accepted for publication April 8, 2003. Address reprint requests to Mr Papagiannopoulos, Leeds General Infirmary, Jubilee Building, Level D, Great George St, Leeds LS1 3EX, UK; e-mail: [email protected].

0003-4975/04/$30.00 doi:10.1016/S0003-4975(03)01378-X

taken for a perforated sigmoid diverticulum, complicated with abscess formation and fecal peritonitis. His postoperative recovery was slow but uneventful. Two months later he was readmitted with fever, left sided upper abdominal and thoracic pain, and significant weight loss. On examination he looked pale and malnourished. The chest film revealed left sided pleural effusion, which was initially drained with insertion of an intercostal drain. Computerized tomography (CT) of the chest and abdomen showed a basal empyema pocket with a trapped left lower lobe and no intraabdominal collection. He was transferred across to our department. Due to his general condition, a rib resection was performed with evacuation of the empyema, curettage of the cavity, and placement of a thick bore drain. His condition improved dramatically and he was discharged 48 hours later. He was readmitted though with left upper abdominal pain and fever. The chest drain fluid had now become severely malodorous and turbid. Cultures revealed coliforms and streptococcus group D growth. An urgent CT of chest and abdomen showed a small residual effusion and a suspicious looking air-containing tract which could be followed in the peritoneal cavity close to the colostomy area (Fig 1). A colostomy enema was performed with 400 mL of Niapam 300 contrast. The sinogram revealed a network of true colopleural fistulas, the longest extending from the colostomy site to the pleural space, and a communicating channel with the distal large bowel stamp (Fig 2). The patient received an urgent laparotomy with adhesiolysis, further en bloc resection of the fistula and colon, with primary large bowel anastomosis and loop ileostomy. He was finally discharged 10 days later in very good clinical condition. Two months later his empyema cavity had resolved with full reexpansion of the left lung. No further surgical management was necessary.

Comment In our case, the empyema was the result of a colopleural fistula. Although visceropleural fistulas are rare entities,

Fig 1. Suspicious (circle) air-containing tract, showing in computed tomography of the chest and abdomen.

CASE REPORT PAPAGIANNOPOULOS ET AL EMPYEMAS FROM COLOPLEURAL FISTULAS

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CASE REPORTS

Ann Thorac Surg 2004;77:324 – 6

Fig 2. Contrast study through colostomy (1) revealing a long fistulous connection (2) with the left pleural space.

their presentation can be dramatic upon failure of early diagnosis. Our literature review revealed four similar cases. Olesen and associates reported a nontraumatic colopleural fistula causing empyema with no definitive etiology [1]. Radin and associates, as well as Sinha and associates [2, 3], have reported cases of colopleural fistulas, after undiagnosed diaphragmatic ruptures or congenital diaphragmatic hernias, leading to colon herniation and necrosis with rupture and empyema. Roberts and associates [4] reported a similar case of empyema due to gastric wall erosion and rupture into the pleural cavity with associated gastro colonic fistula. Our case presented as a true colopleural fistula with tract formation and no direct continuity of the colonic lumen with the diaphragm. How can anyone suspect such a rare pathology? There is no specific protocol other than high suspicion in cases of combined upper abdominal and chest pain with associated unexplained pleural effusion. The microbiology and biochemical analysis of the pleural effusion might give further information combined with previous medical history, detailed clinical examination, and CT of the chest and abdomen. We found, nevertheless, that the contrast study of the gastrointestinal tract not only demonstrates effectively the pathology, but provides useful information for further treatment planning. There is no universally accepted management for colopleural fistulas. Conservative treatment with total parenteral nutrition can be considered in selected uncomplicated cases, or where surgery is contraindicated with associated comorbidity [1, 5]. Successful conservative management might be compromised, due to the pressure gradient between the abdominal and pleural end of such fistulas, since the intrapleural pressure is negative or when the fistulous tract is covered with mucosa. Surgical management is essential when there is associated complex pleural pathology, with or without colonic pathology such as distal obstruction, ischemic bowel

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CASE REPORT LIN AND IANNETTONI ADHESIVE CLOSURE OF BRONCHIAL FISTULAS

necrosis, malignancy, or a complex network of established true fistulas. In conclusion, colopleural fistulas are rare and present, almost always, a diagnostic challenge. They could be classified as traumatic or nontraumatic and false or true when an established sinus is present. Diagnosis should be suspected by the presence of colonic flora in the pleural cavity following colonic injuries or benign and malignant abdominal pathology, or ongoing unexplained pleural effusion with abdominal pain. Delayed recognition might lead to complications requiring complex management with escalation of hospitalization cost and increased morbidity.

Ann Thorac Surg 2004;77:326 – 8

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CASE REPORTS

ronchopleural fistulas can be a severe complication of pulmonary resection with a mortality between 29% and 71% [1] and occur in 0.8% to 15% of pneumonectomies according to a recent series [2]. Large, refractory fistulas may require operative interventions including bronchial stump revision with omental or muscular reinforcement, thoracoplasty, or open window thoracostomy. However, minimally-invasive techniques using tissue sealants, including fibrin, have been recommended for initial therapy [3]. In this report the application of a bovine albumin-glutaraldehyde tissue adhesive (BioGlue; Cryolife Inc., Kennesaw, GA) is described as an alternative agent that may be effective in the closure of bronchopleural fistulas.

References 1. Olesen LL, Pedersen JT. Colo-pleural fistula. Eur Respir J 1989;2(8):792–3. 2. Radin DR, Ray MJ, Halls JM. Strangulated diaphragmatic hernia with pneumothorax due to colopleural fistula. AJR 1986;146:321–322. 3. Sinha M, Gibbons P, Kennedy SC, Matthews HR. Colopleural fistula due to strangulated Bochdalek hernia in an adult. Thorax 1989;44:762–763. 4. Roberts CM, Gelder CM, Goldstraw P, Spiro SG. Tension pneumothorax and empyema as a consequence of gastro– pleural fistulae. Resp Med 1990;84(3):253–4. 5. Graham JA. Conservative treatment of gastrointestinal fistulas. Surg Gynaecol Obstet 1977;144:512–514.

Closure of Bronchopleural Fistulas Using Albumin-Glutaraldehyde Tissue Adhesive Jules Lin, MD, and Mark D. Iannettoni, MD Section of Thoracic Surgery, University of Michigan Medical Center, Ann Arbor, Michigan

Bronchopleural fistulas are a life-threatening complication of pulmonary resection. A 21-year-old woman developed a large bronchopleural fistula after undergoing a pneumonectomy for carcinoid tumor. Despite bronchial stump revision and omental coverage, the fistula recurred. The second patient is a 42-year-old woman with a history of multiple thoracotomies who developed a bronchopleural fistula following aortic root replacement. Using either rigid bronchoscopy or thoracoscopy, these fistulas were evaluated and sealed with an albuminglutaraldehyde tissue adhesive that may have improved strength and biocompatibility compared with other tissue sealants. This approach may be an effective alternative in the treatment of bronchopleural fistulas. (Ann Thorac Surg 2004;77:326 – 8) © 2004 by The Society of Thoracic Surgeons Accepted for publication April 29, 2003. Address reprint requests to Dr Iannettoni, Department of Surgery, University of Michigan Medical Center, 2120 Taubman Center, 1500 E. Medical Center Dr, Box 0344, Ann Arbor, MI 48109; e-mail: [email protected].

© 2004 by The Society of Thoracic Surgeons Published by Elsevier Inc

Case Reports Patient 1 A 21-year-old woman underwent a right pneumonectomy for a carcinoid tumor involving the right mainstem bronchus, which was transected just above the origin of the upper lobe. One-month later, she developed an empyema and underwent tube thoracostomy. Bronchoscopy revealed a large fistula at the medial aspect of the bronchial stump. Although the air leak initially subsided with the application of fibrin, it recurred the following day. On final pathology, the pneumonectomy margin was positive for focal carcinoid. After transfer to the University of Michigan for further treatment, she was found on bronchoscopy to have a 1-cm opening at the bronchial stump. She underwent reexcision of the bronchial cuff, with negative frozen sections, using a stapled closure reinforced with Vicryl sutures and an omental flap. One-week later, a Clagett procedure was performed, filling the pleural cavity with polymyxin, neomycin, and bacitracin. However, 4-months later she developed fevers with a right hydropneumothorax consistent with a recurrent bronchopleural fistula. She was started on intravenous antibiotics and underwent tube thoracostomy. Bronchoscopy demonstrated a small necrotic area in the central portion of the bronchial stump, although no frank hole was seen. Two 4 Fr Fogarty catheters were attached to the double-barreled BioGlue applicator. Using rigid bronchoscopy, the catheters were positioned over the weakened area, which was then completely covered with BioGlue. The patient was placed in the right lateral decubitus position for 1 minute to allow the adhesive to dry and to prevent occlusion of the left bronchial airways. Postoperatively, no air leak was found, and antibiotics were stopped after she defervesced. On postoperative day 3 the right hemithorax was filled with double antibiotic (DAB) solution, composed of doxycycline and polymyxin B, and the chest tube was removed. At 5-month follow-up she was much improved with complete filling of her right hemithorax on chest radiograph.

Patient 2 The second patient is a 42-year-old woman with a history of bicuspid aortic valve who had undergone multiple 0003-4975/04/$30.00 doi:10.1016/S0003-4975(03)00750-1

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