Ann Thorac Surg 1998;66:1409-1410
© 1998 The Society of Thoracic Surgeons
Case Reports
Hodgkin’s disease presenting with a bronchoesophageal fistula
Edmund S. Kassis, BSa,
Chandra P. Belani, MDb,
Peter F. Ferson, MDa,
Robert J. Keenan, MDa,
James D. Luketich, MDa
a Section of Thoracic Surgery, Department of Surgery, University of Pittsburgh School of Medicine, Pittsburgh, Pennsylvania USA
b Division of Medical Oncology, Department of Medicine, University of Pittsburgh School of Medicine, Pittsburgh, Pennsylvania USA
Accepted for publication April 23, 1998.
Address reprint requests to Dr Luketich, 3471 Fifth Ave, Suite 300, Pittsburgh, PA 15213
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Abstract
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Bronchoesophageal fistula is a rare complication of Hodgkin’s disease. We report a case of Hodgkin’s disease presenting with a bronchoesophageal fistula that was successfully treated with bipolar esophageal exclusion and substernal gastric bypass. Direct invasion from mediastinal lymph nodes was the probable cause. Although bronchoesophageal fistula can result as a complication during the course of treatment for Hodgkin’s disease, it rarely occurs as the presenting feature. Early recognition and surgical treatment are key aspects of management.
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Introduction
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Bronchoesophageal fistulas (BEFs) in patients with Hodgkin’s disease (HD) most commonly result from radiation therapy with or without chemotherapy [1]. Bronchoesophageal fistulas rarely occur as the presenting feature of HD. The degree of pulmonary contamination, type of treatment initiated, and the time interval from diagnosis to management of BEF have the most profound impact on survival [2]. The initial treatment should be directed toward the treatment of the BEF, followed by definitive management of the malignant condition.
A 70-year-old man with a 60 pack-year smoking history presented with dysphagia to solids, productive cough, and a 13.5-kg weight loss over a 3-month period. Initially, he was treated with oral antibiotics for a presumed respiratory tract infection, without significant improvement. There was no history of hemoptysis, vomiting, nausea, fever, or chills. Past medical history was remarkable for prostate cancer, which was treated with radiation therapy 1 year before the current presentation and was deemed to be in clinical remission. Physical examination revealed a thin man without any evidence of peripheral adenopathy. Breath sounds were harsh and diminished at the right base. The remainder of the examination was unremarkable.
Chest radiography demonstrated consolidation of the right lower lobe and patchy infiltrates of the bilateral lower lobes. Computed tomography of the chest and a barium esophagogram were performed (Fig 1). Bronchoscopy revealed a large ulcerating defect on the medial wall of bronchus intermedius extending down to the opening of the lower lobe orifice. Esophagoscopy demonstrated a necrotic defect of the esophageal wall approximately 25 cm from the incisors, communicating with the bronchus intermedius. The patient underwent a bipolar esophageal exclusion and substernal gastric bypass to the cervical esophagus. The lower esophagus was transhiatally dissected to the inferior pulmonary vein and removed. The proximal esophageal remnant was oversewn. Biopsy of several lymph nodes in the area was performed. The postoperative course was uneventful. A barium swallow 1 week postoperatively revealed no anastomotic leak. The patient was discharged home on postoperative day 9.
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Fig 1. (A) Computed tomogram of the chest demonstrating a communication between the midesophagus and bronchus intermedius. Note the mural thickening of the adjacent esophageal wall and the presence of mediastinal adenopathy. Abdominal computed tomography revealed the presence of gastrohepatic adenopathy. (B) Barium swallow demonstrating extravasation of barium from the esophagus into the right tracheobronchial tree.
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Pathologic analysis of the biopsy specimens was consistent with the diagnosis of nodular sclerosing HD (Fig 2). The tumor displayed the following immune marker pattern: CD30 (Ber H2) was positive; CD15 (Leu M1), Lewis X, and EMA were negative. The final diagnosis of HD was established based on histologic appearance, characteristic immune marker pattern, and immunoperoxidase staining.
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Fig 2. Photomicrograph of a lymph node biopsy specimen. Note the effacement of nodal architecture and presence of lymphocytes, histiocytes, plasma cells, and large, multinucleated Reed-Sternberg cells. (Hematoxylin and eosin; x40 before 50% reduction.)
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Staging demonstrated negative bone marrow, spleen, and liver involvement. Stage was determined to be IIIB-E. A chemotherapeutic intervention with doxorubicin, bleomycin, vinblastine, and dacarbazine was initiated. The patient had a radiographic partial response and remained well for 6 months. However, disease progression occurred and the patient died 8 months after the initial presentation. There was no evidence of recurrent BEF.
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Comment
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Malignant BEF is a devastating and often rapidly fatal complication occurring in a variety of malignancies. In the largest series of BEFs reported, 207 cases of malignant fistulas were described [2]. The most common tumors resulting in fistula were esophageal (77%) and lung (16%) carcinoma. Only 2 cases of BEF (0.1%) were a result of HD.
Bronchoesophageal fistula is an uncommon complication of lymphoma. It is more likely to be associated with nodular sclerosing HD than other types of lymphoma [1]. Before our case, there have been 38 reported cases of BEF secondary to HD [3]. Our case is unusual in that the BEF was the initial presentation of the HD. This has been reported only twice previously [3, 4]. In contrast to our patient, the 2 prior cases revealed no evidence of peripheral, hilar, or mediastinal adenopathy, raising the possibility of hematogenous spread from another site. Marquand and associates [4] reported that biopsies performed at the site of the fistula did not prove the diagnosis of HD. Instead, a diagnosis of bronchial carcinoma was made. Hodgkin’s disease was not proved until 11 months after presentation, when splenic involvement was noted. Small and colleagues [3] described a patient who was diagnosed with BEF secondary to HD and was treated successfully with operation followed by combination chemotherapy. The patient had a complete response, and therapy was stopped. Seven months after treatment he had recurrence with bone marrow involvement. In all 3 cases, the BEF developed without prior radiation or chemotherapy. This is unusual because the majority of cases of BEF in patients with lymphoma are secondary to radiation or chemotherapy [1].
Patients with intrathoracic HD commonly have associated mediastinal or hilar adenopathy. In reports by Johnson and coworkers [5] and Filly and associates [6] only 4 of 344 patients (1.1%) with intrathoracic HD had no evidence of adenopathy. Bronchoesophageal fistula is rarely caused by a lymphoma that originates in the esophagus or bronchus [7, 8]. In our patient, the source of the fistula was probably direct invasion from mediastinal lymph nodes.
Although BEF in the patient with HD is a serious complication, it carries a better prognosis than does a fistula secondary to bronchogenic or esophageal malignancies [1]. Importantly, the only long-term survivors in patients with BEF secondary to lymphoma are those who undergo successful closure or bypass of the fistula [1]. Once they are successfully treated, their long-term outcome is comparable with that of patients who do not have a fistula [1].
After surgical management, our patient improved symptomatically, but unfortunately succumbed to his primary disease 8 months later. Early recognition of BEF is thus of utmost importance, and aggressive management of BEF appears to improve overall outcome.
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References
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- Perry R.R., Rosenberg R.K., Pass H.I. Tracheoesophageal fistula in the patient with lymphoma: case report and review of the literature. Surgery 1989;105:770-777.[Medline]
- Burt M., Martini N., Bains M.S., Ginsberg R.J., McCormack P.M., Rusch V.W. Malignant esophagorespiratory fistula: management options and survival. Ann Thorac Surg 1991;52:1222-1229.[Abstract/Free Full Text]
- Small D., Caplan S., Sheiner N., Trudel M. Tracheoesophageal fistula as the initial presentation of Hodgkin’s disease. South Med J 1995;88:664-666.[Medline]
- Marquand J., Baumelou E., Fernandez N., et al. Fistule broncho-oesophagienne, premier signe d’une maladie de Hodgkin: A propos d’un cas. Chirurgie 1985;111:857-861.[Medline]
- Johnson D.W., Hoppe R.T., Cox R.S., Rosenberg S.A., Kaplan H.S. Hodgkin’s disease limited to intrathoracic sites. Cancer 1983;52:8-13.[Medline]
- Filly R., Blank N., Castellino R.A. Radiographic distribution of intrathoracic disease in previously untreated patients with Hodgkin’s disease and non-Hodgkin’s lymphoma. Radiology 1976;120:277-281.[Abstract/Free Full Text]
- Harper P.G., Fisher C., McLennan K., Souhami R.L. Presentation of Hodgkin’s disease as an endobronchial lesion. Cancer 1984;53:147-150.[Medline]
- Kelly J.P., Roper L.L. Treatment of malignant tracheoesophageal fistula due to histiocytic lymphoma. South Med J 1984;77:1203-1205.[Medline]
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Abstract
Introduction
