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Spontaneous Pneumothorax in Patients with Leukemia: First Sign of Invasive Mycosis?
- Corrado Girmenia, MD;
- Vittorio Donato, MD;
- Anna Paola Iori, MD; and
- Pietro Martino, MD
- From Universita degli Studi La Sapienza, Rome, Italy. Requests for Reprints: Pietro Martino, MD, Istituto di Ematologia, Via Benevento 6, 00161 Rome, Italy. Grant Support: In part by grant 92.0221.939 from Progetto Finalizzato Applicazioni Cliniche alla Ricerca Oncologica, Centro Nazionale delle Ricerche, Italy.
Abstract
Two patients with leukemia, who underwent remission induction chemotherapy and allogeneic bone marrow transplantation, respectively, reported chest pain and dyspnea after recovery from neutropenia. In both patients, chest radiographs showed only a pneumothorax, whereas computed tomographic scanning also showed a peripheral pseudomycetoma. Antifungal treatment was instituted. The occurrence of spontaneous pneumothorax in patients with hematologic malignancies, particularly after recovery from neutropenia, may indicate the presence of a subpleural infection. Computed tomographic scanning can reveal peripheral pulmonary lesions that are not detected by chest roentgenography.
In recent years, pulmonary fungal infections have been increasingly reported in neutropenic patients with hematologic malignancies [1, 2]. Usually, roentgenographic findings do not provide any clues to identifying the specific pathogen except in a few cases in which late development of a cavitary lesion can suggest infection with Aspergillus species [3-5]. Pneumothorax from the rupture of a peripheral fungal lesion into the pleural space may be a severe complication in patients who survive neutropenia [6]. We recently observed two patients with leukemia who each developed a spontaneous pneumothorax that represented the first sign of a pulmonary fungal infection. No pulmonary lesion was detected by chest roentgenography, and only computed tomography (CT) scanning showed the cause of spontaneous pneumothorax.
Case Reports
Case 1
Acute nonlymphoid leukemia was diagnosed in a 17-year-old boy in June 1990. Remission-induction cytotoxic chemotherapy was started. Nine days after hospital admission, the patient was febrile and profoundly neutropenic. Broad-spectrum antibacterial therapy was promptly started. A chest roentgenogram was negative for pulmonary infiltrates. After 7 days of persisting fever, empiric therapy with amphotericin B (1 mg/kg) was begun, and fever disappeared within the following 5 days. A repeated chest roentgenogram was done and showed no evidence of pulmonary infection.
On hospital day 38, after recovery from neutropenia (neutrophil count, 5500/mm3), the patient reported right-sided chest pain. A chest radiograph showed a small right pneumothorax but no evidence of pulmonary lesions. Computed tomographic scanning also showed a peripheral pseudomycetoma in the homolateral middle-lung field open in the pleural space (Figure 1). Pneumothorax spontaneously resolved. Therapy with amphotericin B was given for 8 weeks (total dose, 1925 mg), leading to partial resolution of pulmonary lesion.
Case 2
A 39-year-old man was diagnosed with chronic myelogenous leukemia in September 1990. In March 1992, he underwent allogeneic bone marrow transplantation. Seven days after bone marrow infusion, the patient had cutaneous signs of grade I graft-versus-host disease, and treatment with prednisone (280 mg/d) was begun. Because of progression of graft-versus-host disease and hepatic involvement, he was eventually treated with antilymphocyte serum and anti-CD-25 immunotoxin. On day 25 after bone marrow infusion, engraftment was observed and the peripheral neutrophil count increased progressively. On day 53, the patient developed dyspnea, and a small right pneumothorax was documented by chest roentgenogram. Another radiograph done after resolution of the pneumothorax did not show pulmonary lesions, whereas CT scanning after 6 days showed a subpleural cavitary lesion in the upper-right lung field. Antifungal therapy was started, but hepatic failure progressively worsened, and the patient died on day 76 after bone marrow infusion. An autopsy was not done.
Discussion
Invasive mycosis usually manifests in neutropenic patients as a febrile episode without specific clinical signs [1, 2]. In patients who survive, signs of fungal invasion of tissue may be detected after recovery from neutropenia. Hepatosplenic lesions and pulmonary pseudomycetoma may represent late clinical manifestations of infection with Candida and Aspergillus species, respectively [2-5].
The common characteristic of the two cases we report is the development of a spontaneous pneumothorax before the documentation of a pulmonary fungal infection. In a previous study [6], we observed that some patients with a pulmonary pseudomycetoma developed a spontaneous pneumothorax in association with a peripheral fungal lesion, which was not detected by chest roentgenography but was detected by CT scanning. On the basis of this experience, we suspected the presence of a subpleural fungal infection in the two patients we describe, and this was confirmed by CT scanning.
In both patients, the peripheral cavitary lesion detected by CT scanning was the unique pulmonary fungal localization and remained undetected by repeated chest roentgenography done after resolution of the pneumothorax. The diagnosis of fungal infection in these severely immunocompromised patients was important, enabling the institution of antifungal treatment to prevent a potentially fatal fungal dissemination.
The occurrence of spontaneous pneumothorax in patients with hematologic malignancies, particularly after recovery from neutropenia, may indicate the presence of a subpleural fungal infection. Computed tomographic scanning seems to be necessary to show peripheral pulmonary lesions that are not detectable by chest roentgenography.
- Copyright 2004 by the American College of Physicians
