Tracheoesophageal fistula (TEF) in adult patients has been reported to mainly occur in patients with carcino- ma of the lung and esophagus. TEF is an uncommon complication of leukemia and lymphoma. Aspergillus has newly emerged at a leading cause of death due to in- fectious fungal organisms in the immunocompromised host (1). Aspergillus bronchitis is a relatively indolent process that is typically diagnosed only on autopsy (2).
We report here on a case of TEF that evolved in a pa- tient during chemotherapy for acute lymphoblastic leukemia (ALL).
Case Report
A 46-year-old woman presented with a 2-month histo- ry of a palpable, non-tender mass in her right lower neck. On admission, the laboratory studies were re- markable for the blast form cells; her white blood cell
count was 26,200/mm3, the Hb was 4.1 g/dL and the platelet count was 28,000/mm3. The bone marrow biop- sy revealed ALL. We started induction chemotherapy with cyclophosphamide, daunolubicin, vincristine, prednisone and L-asperginase. During chemotherapy, she had a trouble swallowing, a productive coughing and excessive secretions. Chest X-ray films showed re- lapsing bronchopneumonia, collapse of the left lower lobe and a 4 cm×2 cm sized, ovoid soft tissue mass plugging the left main bronchus (Fig. 1). Chest CT demonstrated an irregular soft tissue mass that was plugging left main bronchus with TEF (Figs. 2A and 2B).
Bronchoscopy revealed a large TEF together with a white mass-like lesion that filled the lower trachea and left main bronchus to the distal 1/3 level (Fig. 3). The op- erative findings were TEF around the left carina, a 7 cm
×2 cm sized oval-shaped defect of the tracheal cartilage and membranous part and a 7 cm×2 cm sized esophageal defect. The white soft tissue mass plugging the left main bronchus was a flap-like detachment of the tracheal and esophageal wall. We performed TEF divi- sion, primary repair of the esophagus and repair of the tracheal defect via patch closure with bovine pericardi- um. Tissue of tracheal biopsy and the white mass-like lesion in the lower trachea and left main bronchus re-
J Korean Radiol Soc 2006;54:269-272
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Tracheoesophageal Fistula Resulting from Invasive
Aspergillosis in Acute Lymphoblastic Leukemia: A Case Report1
Si Won Kang, M.D.
1Department of Diagnostic Radiology, Daejeon St. Mary’s Hospital, College of Medicine, Catholic University
Received October 7, 2005 ; Accepted December 10, 2005
Address reprint requests to : Si Won Kang, M.D., Department of Diagnostic Radiology, Daejeon St. Mary’s Hospital, Catholic University, 520-2, Daehung-dong, Jung-gu, Daejeon 301-723, Korea.
Tel. 82-42-220-9837 Fax. 82-42-252-6807 E-mail: [email protected]
Tracheoesophageal fistula (TEF) in adult patients is an uncommon complication in leukemia. We present here on a case of TEF in a 46-year-old woman with ALL. The pa- tient was asymptomatic and TEF is resulted from aspergillus bronchitis during the chemotherapy for acute lymphoblastic leukemia (ALL).
Index words :Trachea Fistula Aspergillosis Leukemia
vealed aspergillosis with infarction (Fig. 4). The patient displayed persistent leakage of secretions at the opera- tion site and an increasing right pleural effusion.
Esophagogram showed leakage of water soluble con- trast media at the primary repaired site of the esophageal defect. So, we performed a second operation of cervical esophagogastrostomy and feeding jejunosto- my. Thereafter, her general condition was improved without leakage of secretions at the operation site, and the right pleural effusion was gradually decreased. She was gradually administered a regular diet.
Discussion
The term TEF is used to refer any fistula formation be- tween the esophagus and the trachea or lung, regardless of the origin of the lesion, including bronchoesophageal fistula. The development of TEF in the setting of malig- nancy is generally a poor prognostic event (3), and aspi- ration pneumonia is the immediate cause of death in the greater proportion of patients. Medical, surgical and en- doscopic methods have been described to treat TEF.
The only effective treatment is to exclude the fistula from the alimentary tract. Self-expanding metal stents are used for palliative reasons to seal the TEF and allow the patient to resume an oral food intake (4). The pa- tients with a large fistula who are in a poor medical con- dition or who suffer from repeated bouts of aspiration pneumonia require more urgent treatment. Our case al- so had a large fistula and relapsing aspiration pneumo-
nia. Since long survival following treatment for leukemia is common, the early recognition and repair of TEF can be lifesaving.
Aspergillus is a ubiquitous air-borne fungal agent that is frequently found to colonize the paranasal sinuses of patients with chronic sinusitis, and it may form as- pergillomata (5). Aspergillus infections are on the in- crease as the number of immunocompromised patients continuous to grow. The most frequent underlying con- ditions in the patients with invasive pulmonary as- pergillosis are leukemia and lymphoma, and prolonged granulocytopenia is a major risk factor. The increased use of immunosuppressive agents for organ transplanta-
Si Won Kang: Tracheoesophageal Fistula Resulting from Invasive Aspergillosis in Acute Lymphoblastic Leukemia
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A B
Fig. 2. A. Axial contrast enhanced CT (5-mm collimination) at the mediastinal window setting shows the tracheoesophageal fistula.
B. CT scan at the lung window setting shows an irregular, soft tissue mass (aspergilloma, white arrow) in the left main bronchus, tracheoesophageal fistula (black arrow) and focal consolidations in the right upper lobe.
Fig. 1. Chest PA shows about a 4 cm×2 cm sized, ovoid soft tissue mass plugging the left main bronchus (black arrows) and an air-filled esophagus (white arrows).
tion and autoimmune disorders is partly responsible for this increasing incidence of patients with Aspergillus in- fections, and the growing number of patients infected with HIV likewise contributes to this population. The chemotherapeutic regimens used for the treatment of hematologic malignancies and solid tumors and for in- duction therapy for bone marrow transplantation also render a significant number of patients susceptible to opportunistic infections (5-7). The pathophysiology of invasive aspergillus infection includes soft tissue exten- sion, vascular invasion and infection (6). This process can result in the transmural necrosis of a viscus (tra- chea, bronchus or the gastrointestinal tract). Vascular in- vasion may also result in hemorrhage severe enough to cause death (8). Massive hemoptysis, bronchopleural fis- tula and TEF are possible with pulmonary involvement.
The early recognition of aspergillosis is critical to achieve an optimal therapeutic result for immunocom- promised patients. Delayed diagnosis and treatment may lead to the progression of infection and usually to a fatal outcome. Treatment of aspergillus infections in the immunocompromised population includes correcting the immunodeficiency, high dose intravenous ampho- tericin B, resection of the necrotic tissue and reconstruc- tion of any involved structures when it is feasible (esophagus, trachea, vessels, etc) (9). Invasive aspergillo- sis is being seen with increasing frequency as the popu- lation of immunocompromised patients continuous to grow. This is a report on TEF that resulted from inva-
sive aspergillus infection in an immunocompromised host. Early recognition and intervention may be life sav- ing for patients with this infection, which frequently portends a fatal outcome.
References
1. Peterson PK, McGlave P, Ramsay NK, Rhame F, Cohen E, Ferry GS 3rd, et al. A prospective study of infectious disease following bone marrow transplantation: emergence of aspergillus and cy- tomegalovirus as the major causes of mortality. Infect Control 1983;
4:81-89
2. Young RC, Bennett JE, Vogel CL, Carborne PP, DeVita VT.
Aspergillosis: the spectrum of the disease in 98 patients. Medicine 1970;49:147-173
3. Daranceau A, Jamieson, GG. Malignant tracheoesophageal fistula.
Ann Thorac Surg 1984;37:346-354
4. Bernal AB, Rochling FA, DiBaise JK. Lymphoma and tracheoe- sophageal fistula: indication for a removable esophageal stent. Dis Esophagus 2005;18:57-59
5. Hebert PA, Bayer AS. Fungal pneumonia: invasive pulmonary as- pergillosis. Chest 1981;80:220-225
6. Albelda SM, Gefter WB, Epstein DM, Miller WT. Bronchopleural fistula complicating invasive pulmonary aspergillosis. Am Rev Respir Dis 1982;126:163-165
7. Berlinger NT, Freeman TJ. Acute airway obstruction due to necro- tizing tracheobronchial aspergillosis in immunocompromised pa- tients: a new clinical entity. Ann Otol Rhinol Laryngol 1989;98:718- 720
8. Schubert MM, Peterson DE, Myers JD, Hackman R, Thomas ED.
Head and neck aspergillosis in patients undergoing bone marrow transplantation. Cancer 1986;57:1092-1096
9. Stack BC, Ridley MB, Greene JN, Hubbell DS. Tracheoesophageal fistula and sinusitis from invasive aspergillosis. Otolaryngol Head Neck Surg 1997;116:116-119
J Korean Radiol Soc 2006;54:269-272
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Fig. 4. Microphoto-image shows the aspergillus colony with septate hyphae. (Gomori-methanamine silver stain, ×200) Fig. 3. Bronchoscopy revealed a large tracheoesophageal fistu-
la with a white mass like lesion that filled the lower trachea and left main bronchus.
Si Won Kang: Tracheoesophageal Fistula Resulting from Invasive Aspergillosis in Acute Lymphoblastic Leukemia
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대한영상의학회지 2006;54:269-272
림프모구성 백혈병 환자에서 침습성 아스페르길루스증 합병으로 발생된 기관지 식도 누출공: 증례 보고1
1대전성모병원 영상의학과 강 시 원
백혈병 환자에서 기관지 식도 누출공은 매우 드믄 합병증이다. 저자들은 최근 46세 여자, 림프모구성 백혈병환자에 서 화학요법 중에, 침습성 아스페르길루스증 합병으로 발생된 기관지 식도 누출공 1예를 경험하였기에 문헌고찰과 함 께 보고한다.
