Life-threatening airway obstruction caused by penicilliosis in a leukemic patient
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- Hsu, JH., Lee, MS., Dai, ZK. et al. Ann Hematol (2009) 88: 393. doi:10.1007/s00277-008-0606-5
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Penicilliosis is a progressive disseminated disease caused by Penicillium marneffei, an opportunistic fungal pathogen unique to immunocompromised hosts. Delayed diagnosis or inappropriate treatment could result in deep mycosis with high mortality. It is a common opportunistic infection in HIV-positive patients, especially in endemic areas such as southeast Asia, southern China, and Taiwan. However, it is uncommon in patients with hematologic malignancy. Here, we describe a leukemic patient who survived an episode of life-threatening airway obstruction caused by invasive pulmonary penicilliosis.
P. marneffei is a common opportunistic fungal pathogen in AIDS patients [1, 2]. In southeast Asia, it is the third most common opportunistic infection in AIDS patients, following tuberculosis and cryptococcosis [1, 2]. However, it is an unusual pathogen in patients with hematological malignancies [3, 4] and only 2% of infections in neutropenic patients with hematological malignancies are caused by P. marneffei . The pathogen is acquired via inhalation and results in initial pulmonary infection, followed by fungemia and dissemination of the infection. Common clinical manifestations include fever, skin lesions resembling molluscum contagiosum, cough, and infiltrates in chest radiographs[1, 2]. Without timely diagnosis and appropriate treatment, penicilliosis has a high mortality rate.
Our case is unique in that he survived the acute invasive fungal infection of a major bronchus that resulted in sloughing of a large piece of infected tissue containing cartilage that obstructed his airway resulting in atelectasis and arterial desaturation. This report indicates that pulmonary penicilliosis may cause severe airway obstruction, an unusual but potentially fatal complication, in leukemic patients. To our knowledge, this is the first reported case of airway obstruction caused by penicilliosis in leukemic patients. Even though the origin of the destructed cartilages in this case was not verified by bronchoscopy, it is highly possible that an invasive pulmonary mycosis may destruct bronchial cartilages and thus causes acute airway obstruction [5, 6]. The standard antifungal treatment of disseminated P. marneffei infection consists of parenteral amphotericin B followed by oral itraconazole therapy, and patients are maintained on itraconazole therapy to prevent relapse of the disease [7, 8]. However, in our patient, the response to amphotericin B was poor so that itraconazole was used for both primary and maintenance therapy. In summary, the present case demonstrates that penicilliosis, an unusual but potentially life-threatening infection, should be considered in leukemic patients with neutropenia. Atelectasis in the chest radiograph can be an important reminder of severe airway obstruction in patients with invasive mycosis.