INTRODUCTION: Tracheoesophageal fistula (TEF) is an uncommon problem that may present in adulthood as a latent manifestation of a congenital abnormality or as an acquired complication of a disease state, most commonly esophageal and lung malignancy. We report a case of massive tracheoesophageal fistula in a patient with rectal adenocarcinoma metastatic to the lungs, treated with radiation and a chemotherapy regimen including Bevacizumab.
CASE PRESENTATION: A 41 year-old white female presented to the hospital with two weeks of progressive, severe cough, sputum production as well as nausea and vomiting with any oral intake. She had a history of rectal adenocarcinoma metastatic to lung. Her initial cancer therapy two years prior included radiation followed by resection with adjuvant 5-fluorouracil, leucovorin, and oxiplatin. Since discovery of bilateral pulmonary metastases approximately sixteen months prior to presentation, she received 5-fluorouracil, leucovorin, irinotecan, bevacizumab, and capecitabine as well as stereotactic radiation to lung nodules, the last treatment occurring nine months prior. On admission, her chest x-ray revealed infiltrates in the right middle and right lower lobes. After no improvement despite several days of broad spectrum antibiotics a chest CT revealed a large tracheoesophageal fistula that appeared to communicate with a cavitary lung lesion. She was then transferred to our institution and underwent bronchoscopy. Bronchoscopy revealed complete obliteration of the mucosa circumferentially from mid trachea to proximal mainstem bronchi bilaterally. Further, obliteration of the carina and posterior membrane of the right upper lobe bronchus was noted with direct communication into the mediastinum and esophagus. There was no visible tumor and forceps biopsies were negative for malignancy. She subsequently underwent PEG tube insertion and an esophageal stent was placed. She was started empirically on amphotericin and voriconazole in addition to broad spectrum antibiotics. Her respiratory cultures grew only candida albicans but were otherwise negative. Four days following bronchoscopy her respiratory status declined and she required intubation. Ultimately, her family opted to withdraw care.
DISCUSSIONS: Tracheoesophageal fistula (TEF) remains a rare complication of malignancy with the majority of cases resulting from direct invasion by esophageal carcinoma or primary lung neoplasm. Less commonly, TEF results from instrumentation and local treatment including embolization or radiation. TEF carries a poor prognosis and prompt diagnosis and intervention is critical to minimize soiling of the airway and further complications. However, diagnosis can be difficult as patients typically present with pulmonary signs and symptoms mistakenly attributed to underlying malignancy or anti-neoplastic therapy. Bevacizumab is a frequently used component of the chemotherapy regimen for colorectal cancer and non-small cell lung cancer. Gastroenteral tract perforation and fistulization is a more commonly reported side effect of bevacizumab than respiratory complications. Rare reports of TEF following bevacizumab exist. A recent publication suggests that the anti-angiogenesis of Bevacizumab combined with mucosal injury of the esophagus contributes to TEF formation2;. Our patient is unique in the severity of tracheal injury when compared to other reported cases. Additionally, her radiation exposure was more remote than most, but not all, previously reported cases of TEF post bevacizumab.
CONCLUSION: In patients undergoing treatment for intra-thoracic malignancy who present with pulmonary complaints, the differential diagnosis is broad. Acquired TEF is a rare complication of malignancy but carries a poor prognosis and relies on early diagnosis for successful management. Our case suggests that a high suspicion for TEF should exist in patients with pulmonary complaints who received bevacizumab, even when radiation therapy is distant to presentation.
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