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Ann Thorac Surg 1999;67:832-834
© 1999 The Society of Thoracic Surgeons


Case Reports

Surgical treatment of tracheomediastinal fistula from recurrent hodgkin’s lymphoma

David G. Tse, MDa, Adam Summers, MDb, James R. Sanger, MDb, George B. Haasler, MDa

a Division of Cardiothoracic, Medical College of Wisconsin, Milwaukee, Wisconsin, USA
b Division of Plastic Surgery, Medical College of Wisconsin, Milwaukee, Wisconsin, USA

Accepted for publication August 14, 1998.

Address reprint requests to Dr Haasler, Department of Cardiothoracic Surgery, Medical College of Wisconsin, 9200 W. Wisconsin Ave, Milwaukee, WI 53226
e-mail: ghaasler{at}mcw.edu


    Abstract
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 Abstract
 Introduction
 Comment
 References
 
The role of surgery in the management of Hodgkin’s disease is usually diagnostic because chemotherapy and radiation are often curative. We report here the surgical treatment of a tracheomediastinal fistula from recurrent Hodgkin’s lymphoma.


    Introduction
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 Abstract
 Introduction
 Comment
 References
 
The role of surgery in the management of Hodgkin’s disease is usually diagnostic because chemotherapy and radiation are often curative. Life-threatening problems may arise making an operation the only realistic approach. We report here the surgical treatment of a tracheomediastinal fistula from recurrent Hodgkin’s lymphoma.

A 29-year-old man with a history of stage IIIB nodular sclerosing Hodgkin’s lymphoma treated 17 years earlier with MOPP (mechlorethamine, vincristine, procarbazine, and prednisone) and ABV (doxorubicin, bleomycin, and vinblastine) chemotherapy presented with progressive mediastinal fibrosis, an evolving large confluent mediastinal mass, and an enlarging fistula involving the left tracheobronchial angle communicating anteriorly into the overlying mediastinum. Previous biopsies of the mediastinum by means of cervical mediastinoscopy, partial upper median sternotomy, left anterior mediastinotomy, and right thoracotomy had shown only fibrotic tissue and reactive lymph nodes but no tumor. Main right pulmonary artery compression compromising lung perfusion was treated with a self-expanding 12-mm x 40-mm endovascular coated Wallstent (Schneider, USA, Inc, Minneapolis, MN). Flexible fiberoptic bronchoscopy, with which a small anterior left mainstem bronchus ulcer had been previously noted, now revealed a large fistula. The resultant cavity walls included both main pulmonary arteries and measured 4 cm x 3 cm x 3 cm. The risk of massive hemorrhage into the airway mandated closure of this enlarging cavitary fistula (Fig 1).



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Fig 1. Computed tomographic scan showing tracheomediastinal fistula, large mediastinal mass, and compression of mediastinal structures.

 
At surgery, flexible fiberoptic bronchoscopy through a right-sided double-lumen endotracheal tube identified a large irregular space with necrotic debris and pulsatile mediastinal structures through a defect in the anterior distal trachea and proximal left mainstem bronchus. Through a median sternotomy, the anterior mediastinum was noted to be completely encased by a fibrotic dense mass that enveloped the aortic root, superior vena cava, and presenting surface of the pericardium. An uninvolved pericardial patch was harvested. The fistula cavity was approached between the aorta and superior vena cava by careful division of the fibrous tissue guided by needle aspiration to avoid great vessel injury. After reaching the cavity, the right pulmonary stent was visualized, lacking overlying pulmonary artery tissue. Two defects from the anterior distal trachea to the proximal left mainstem bronchus that were separated by a 3-mm bridge of cartilage constituted the base of the cavity. Debridement and several biopsies were performed. Repair of the tracheobronchial defect was accomplished with autogenous pericardial patch after insertion of a 1-cm x 4-cm coated Wallstent into the left main bronchus. Omental transposition into the area of repair obliterated the remaining cavity. Reinspection of the airways by bronchoscopy demonstrated good stent position with a nicely expanded proximal left main stem bronchus.

Biopsies from the fibrous cavity and tracheal cartilage bridge (but not from the harvested pericardium) were positive for recurrent nodular sclerosing Hodgkin’s lymphoma. Residual soft tissue infection was treated with 6 weeks of intravenous antibiotics. The patient also required insertion of a multisegment vascular Gianturco stent into the superior vena cava because of tumor encasement and incipient superior vena cava syndrome. Interval computed tomographic scans have since revealed celiac adenopathy. Postoperative chemotherapy and radiation treatments have been well tolerated. Diminution of the mediastinal mass, obliteration of the cavity, and incorporation of the pericardial patch with patent distal airways has been demonstrated on follow-up studies (Fig 2). He remains clinically stable at 4 months after operation.



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Fig 2. Computed tomographic scan 3 months after closure of tracheal bronchial fistula showing omental transposition filling anterior mediastinum and cavity, and a stent in the left main bronchus.

 

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Mediastinal Hodgkin’s lymphoma presenting solely as an intrathoracic neoplasm occurs in approximately 50% of Hodgkin’s lymphomas. The incidence of relapse isolated to the mediastinum is more remote (12% to 15%) and depends on both initial pathologic stage and treatment modality [1, 2]. Sequelae of mediastinal Hodgkin’s lymphoma have included encasement (superior vena cava syndrome, compression of main pulmonary arteries), bleeding, and perforation (tracheoesophageal fistula) [35]. Essential to treatment is obtaining a diagnosis, which is the usual role of surgery; thereafter, appropriate antineoplastic therapy can often cure difficult problems as illustrated in tracheoesophageal fistula secondary to Hodgkin’s lymphoma [5].

Fistulization between the trachea and anterovisceral mediastinum, an uncommon entity, and its surgical management have not been reported when arising from mediastinal Hodgkin’s lymphoma. This case highlights diagnosis and surgical treatment of a challenging, potentially disastrous problem. Repair of a tracheal defect with an autogenous pericardial patch in the presence of an infected cavity once infection was controlled appeared a viable option for separation of the great vessels from the tracheobronchial tree. Experimental studies of pericardial incorporation have pointed to reepithelialization and eventual stiffening [6]. Further stabilization was provided with endobronchial stenting. Elimination of the residual space after debridement and buttressing of major vascular structures was accomplished with viable vascular tissue, ie, omental transposition. Fortuitously, the concomitant mediastinal fibrosis and omental transplant may have decreased the risk of progressive mediastinitis and afforded better antibiotic penetration.

Hodgkin’s disease appearing like sclerosing mediastinitis has been previously reported [7]. A diagnosis of recurrent Hodgkin’s disease was finally obtained from mediastinal and tracheal biopsies to direct definitive treatment as multiple previous surgical procedures had failed to establish this. Initial biopsy specimens were nonspecific, having features consistent with multiple etiologic processes such as Castleman’s disease and postinfectious mediastinal fibrosis.

Lastly, the use of endovascular stents for management of major mediastinal great vessel compression is demonstrated here as an evolving alternative to surgical bypass. The coated right pulmonary artery stent undoubtedly prevented major hemorrhage from occurring before and during the surgical correction.


    References
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 Abstract
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 References
 

  1. Roach M., III, Brophy N., Cox R., et al. Prognostic factors for patients relapsing after radiotherapy for early Hodgkin’s disease. J Clin Oncol 1990;8:623-629.[Abstract]
  2. Healey E.A., Tarbel N., Kalish L., et al. Prognostic factors for patients with Hodgkin’s disease in first relapse. Cancer 1993;71:2613-2620.[Medline]
  3. Yellin A., Mandel M., Rechavi G., Newman Y., Ramot B., Lieberman Y. Superior vena cava syndrome associated with lymphoma. Am J Dis Child 1992;146:1060-1063.[Abstract/Free Full Text]
  4. Shields J.J., Cho K.J., Geisinger K.R. Pulmonary artery constriction by mediastinal lymphoma simulating pulmonary embolus. Am J Roentgenol 1980;135:147-150.[Abstract]
  5. Greven K.M., Evans L.S. The occurrence and management of esophageal fistulas resulting from Hodgkin’s disease. Cancer 1992;69:1031-1033.[Medline]
  6. Bryant L.R., Eiseman B. Replacement of tracheobronchial defects with autogenous pericardium. J Thorac Cardiovasc Surg 1964;48:733-740.
  7. Flannery M.T., Espino M., Altus P., Messina J., Wallach P.M. Hodgkin’s disease masquerading as sclerosing mediastinitis. South Med J 1994;87:921-923.[Medline]



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George B. Haasler
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Right arrow Articles by Haasler, G. B.


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