A 54-year-old woman was referred to Saitama Cardiovascular and Respiratory Center in August 2000 for investigation of a cough, sputum, and an abnormal chest X-ray shadow. The cough and sputum had developed in December 1999 and had not improved. The patient had no relevant personal or family history. Physical examination did not reveal any abnormalities, including wheezes.\nChest X-ray showed bilateral, hilar band-like shadows (). Computed tomography (CT) of the chest showed mucous plugs in both B3 bronchi and consolidation (indicating atelectasis) in the peripheral part of the mucous plugs (). Pulmonary function tests did not indicate bronchial asthma. Laboratory tests showed a white blood cell count of 3,400/mm3, with 9.5 percent eosinophils (323/µl). Radioimmunosorbent test showed the IgE concentration to be within normal limits, and results of radioallergosorbent test for specific IgE antibodies against Aspergillus, Cladosporium, Candida, and Cryptococcus were all negative. No inflammatory reactions, assessed by the C-reactive protein level and erythrocyte sedimentation rate, were observed. Bronchoscopy disclosed thick white mucous plugs in both B3 bronchi () that could not be removed by forceps and suctioning. Removal was attempted again on another day but failed. Higher magnification of the mucous plug sample showed numerous eosinophils (). Grocott staining of the mucous plug sample showed mold with a fragmented appearance (). Transbronchial lung biopsy specimens showed infiltration of eosinophils in the wall of the right upper lobe bronchus. From the third to tenth day after cultivation, cultures of the mucous plugs, bronchial aspirates, and sputum samples yielded the same mycelial colonies, which were white and had a felt-like, fluffy appearance on Sabouraud dextrose agar plates (); however, no other pathogenic microorganisms were isolated. A methane-like odor was observed. With regard to form, there were narrow and thin walls approximately 1.5 mm in width, as well as wide and thick walls approximately 2.5 mm in width, with separate hyphae but no clamp connection. Considering the feature of colonies, form, odor, and the two types of hyphae in the colony, we hypothesized that it was a monokaryotic strain of S. commune. We consulted the Chiba University Research Center for Pathogenic Fungi and Microbial Toxicoses for identification of the fungus. Mating tests were performed [,,]. The clinical isolate was inoculated onto plates opposite each tester monokaryotic strain of S. commune, wherein it was dikaryotized and a fruit body was formed at the point of contact (). When co-cultured with a dikaryotic strain, it was dikaryotized and produced basidiocarps. From the foregoing, this fungus was identified as a monokaryotic mycelium of S. commune. At a later time, the presence of S. commune in the patient's garden was confirmed ().\nABPM, as well as ABPA (allergic bronchopulmonary aspergillosis), usually is diagnosed on the basis of Rosenberg’s seven primary criteria []: (1) episodic bronchial obstruction (asthma); (2) peripheral blood eosinophilia; (3) immediate scratch test reactivity to Aspergillus antigen; (4) precipitating antibodies to Aspergillus antigen; (5) elevated serum IgE concentrations; (6) history of pulmonary infiltrates (transient or fixed); and (7) central bronchiectasis. The diagnosis of ABPM is considered likely if the first six criteria were present, and the presence of all seven made the diagnosis certain. The present case satisfied only two (the second and the sixth criteria) of seven criteria (neither the third nor the fourth criteria were investigated because of methodological limitation), and diagnosis of ABPM cannot be established. We diagnosed the patient as MIB due to S. commune infection.\nComplete removal of the mucous plugs by bronchoscopy was attempted twice unsuccessfully, and the patient was treated with ITCZ for three months, beginning in September 2000. The cough improved and sputum diminished, and in December 2000, bronchoscopy disclosed that the mucous plugs had disappeared. Bronchoscopy was repeated in July 2001, January 2002, and 2003, and there has been no recurrence of the mucous plugs () or symptoms. The patient has been followed up on an outpatient basis for seven years and has shown no signs of recurrence.
A 38-year-old male presented with a cough for 3 days, hemoptysis, fevers, and chills for 1 day (). He denied cough, fevers, night sweats, or weight loss prior to 3 days before admission. He had emigrated to the U.S.A. from Mexico seven years previously and had no foreign travel subsequently. In his previous life, he had worked as an automobile mechanic, in construction, and gardening. His past medical history indicated treatment for a lung infection, with an inability to work, 9 years previously, but he did not recall the diagnosis. During that time, he was treated concurrently with 3 different types of medications for 7 months but was never told that he had tuberculosis. He had no history of risk factors for an anaerobic lung abscess, such as aspiration, alcohol use, smoking or seizures.\nOn admission, his temperature was 39.4°C, leukocyte count was 6500/mm3, and HIV test was negative. Therapy with piperacillin-tazobactam and vancomycin was initiated and the patient remained hospitalized for 2 weeks. Computed tomography of the lung on hospital day 2 demonstrated an 8.2 × 5.5 cm cystic structure in the left upper lobe with an internal air-fluid level, fluid debris levels and a thick, irregular wall, consistent with a pulmonary abscess. There were surrounding areas of “ground-glass” opacities and consolidation in the left upper and lower lobes. Sputum cultures grew methicillin-susceptible Staphylococcus aureus and Klebsiella oxytoca, prompting discontinuation of vancomycin on hospital day 8. On hospital day 9, transthoracic left upper lobe pulmonary abscess drainage was performed, with an aspiration of 140 cc of dark green–black fluid. Cytologic examination of the aspirated fluid did not show malignant cells, but had abundant neutrophils, numerous erythrocytes, and necrotic debris. Smear and culture for mycobacteria, and anaerobic culture, were negative. Fungal culture yielded two colonies of a filamentous non-sporulating fungus. The therapy was then switched to oral amoxicillin-clavulanate. At discharge, his cough had improved and he denied dyspnea.\nAfter discharge, he reported two weeks of mild intermittent dyspnea, but otherwise remained asymptomatic. The amoxicillin-clavulanate therapy was discontinued. However, serial chest radiographs demonstrated a persistent air-fluid level within a thin-walled cystic structure in the posterior left upper lobe. These radiographs were unchanged for four months. He reported two weeks of mild, intermittent dyspnea in the 4th-month post-discharge.\nA chest tomogram performed 5 months post-discharge, to better characterize the disease, revealed a thin-walled cystic cavity in the left upper lobe apical posterior segment, measuring 5.0 × 4.6 cm. The fluid within the lesion was hyperdense, and there was a calcified component posteriorly. Cephalad to the lesion, there was an architectural distortion and bronchiectasis with adjacent scarring and likely adjacent cicatricial emphysema. The oblique fissure was displaced anteriorly, posterior to this region, secondary to the associated volume loss and/or scar. There was less consolidation in this area compared to prior tomograms.\nA sputum bacterial culture was unremarkable, sputum smear for parasites was negative, as were antibody tests for Histoplasma, Coccidioides (IgG and IgM), and Echinococcus, and 2 blood cultures. A test for HIV was again negative. Sputum fungal culture grew Candida albicans and a filamentous fungus, morphologically similar to the previously isolated fungus. With a diagnosis of a persistent abscess, he proceeded to left upper lobectomy and extensive lysis of pleural adhesions. He was hospitalized 8 days, during which time he had intermittent fevers. Initially, there was a mild leukocytosis, which subsided. The intra-operative findings revealed an abscess that was located deep within the posterior aspect of the left upper lobe. The posterior fissure was completely fused and required extensive dissection. Intra-operative cultures were positive for a fungus, identified by sequencing as Gloeostereum sp. This was morphologically similar to the filamentous fungus that had been cultured on the previous occasions. Susceptibility testing of the fungus by macrobroth dilution indicated low MIC to amphotericin B (0.5 µg/ml), but high to voriconazole (MIC of >8 µg/ml) and fluconazole (MIC 16 µg/ml). Candida albicans was not cultured in the specimen, nor seen in the histopathology. No antifungal chemotherapy was given.\nHistopathology of the resected specimen was interpreted as chronic inflammation in a bronchogenic cyst with a lining of ciliated pseudostratified columnar epithelium, some muscle and fibrous tissue, subepithelial chronic inflammation and pulmonary congestion (). The cyst contents showed fungal hyphae (). The fungus was sent to the Westerdijk Fungal Biodiversity Institute for definitive speciation and deposited there under number CBS 145006. The patient was seen in clinic 2 weeks post-discharge, at which time he denied fevers, chills. He remained well in subsequent visits.