A 14-yr-old girl complained of nasal blockage accompanied by yellowish mucopurulent rhinorrhea. No history of asthma, other allergic diseases, or medical or surgical treatment was identified. No potential risk factors such as diabetes mellitus, immunodeficiency, or previous facial trauma were found. Laboratory investigations revealed a white blood cell count of 15,600/mm3 with 3.7% eosinophils and a high level of serum IgE, 149 IU/mL (0-100 IU/mL). The results of the multiple antigen simultaneous test (MAST) for specific serum IgE antibodies against Aspergillus, Penicillium, Cladosporium, and Alternaria were negative. The results of the Uni-CAP system (Pharmacia, Sweden) for specific serum IgE antibodies against Aspergillus fumigatus, Cladosporium herbarum, and Alternaria tenuis were in the normal range. A non-enhanced computed tomography (CT) scan of the ostiomeatal unit revealed soft-tissue opacification in the right frontal, ethmoid, and maxillary sinuses (). Bony remodeling and destruction was found in the right lamina papyracea. Endonasal endoscopic surgery was undertaken. Mucosal swelling and polyposis were found in the right middle meatus, maxillary sinus, ethmoid sinus, and frontal sinus, which encroached on the right lamina papyracea. The frontal recess was filled with mucoid secretions, which were collected for histological and microbiological investigations. Histological examination of the material obtained from the frontal recess showed allergic mucin, characterizing Charcot-Leyden crystals, numerous eosinophils, and a few septate fungal hyphae ().\nThe material was plated onto PDA plates and incubated at room temperature, 30℃, and 37℃. Growth of a white, densely woolly mold with a yellow-to-brown reverse was observed after 3 days at all temperatures (). The fungus was sensitive to cycloheximide (400 µg/mL) and produced a tart and disagreeable smell, particularly at 37℃. Microscopic examination of the mycelia showed no conidia or sporangiospores, but revealed both narrow and wide hyphal filaments, which were hyaline, septate, branched hyphae without clamp connections and spicules (). Macroscopic examination did not reveal structures such as fruiting bodies even after 2 months of culture.\nThe ITS and D1/D2 regions of the 26S ribosomal DNA was sequenced. The ITS region (including the 5.8S rRNA gene) and the 26S rRNA gene D1/D2 domains were amplified with the following primer pairs: pITS-F (5'-GTCGTAACAAGGTTAACCTGCGG-3') and pITS-R (5'-TCCTCCGCTTATTGATATGC-3'), and NL1 (5'-GCATATCAATAAGCGGAGGAAAAG-3') and NL4 (5'-GGTCCGTGTTTCAAGACGG-3') []. The fungus was identified (Basic Local Alignment Search Tool, ) as S. commune (accession number HM595605.1) with 100% homology. The patient was given oral corticosteroids and antibiotics for 1 week after surgery. Topical nasal steroids were administered to suppress the immune response and minimize recurrence. The patient has been followed up for 3 months and shows no signs of recurrence, except mild mucosal swelling.
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.