Invasive aspergillosis primarily occurs in immunocompromised patients, while skeletal muscle involvement remains an exceptionally uncommon manifestation. Since 2000, only nine cases of invasive aspergillosis presenting with skeletal muscle abscesses have been documented. Here, we report a rare case of disseminated Aspergillus fumigatus infection characterized by extensive skeletal muscle abscesses. A 54-year-old martial artist with no previously recognized immunodeficiency presented with a one-year history of recurrent soft-tissue abscesses at multiple anatomical sites despite repeated surgical drainage and prolonged antibiotic therapy. On admission, he was cachectic and had multiple fluctuant subcutaneous and intramuscular abscesses, markedly elevated C-reactive protein (180.2 mg/L), and anemia. Evaluations revealed no previously recognized immunodeficiency or malignancy. However, comprehensive immunologic testing for underlying host defense defects was not performed. Chest and abdominopelvic computed tomography (CT) showed extensive chest wall and bilateral iliopsoas abscesses, as well as a cavitary lesion in the right middle lobe extending into the chest wall and associated with rib destruction. CT-guided aspiration of the right iliopsoas abscess demonstrated septate fungal hyphae, while fungal culture and multiplex polymerase chain reaction confirmed A. fumigatus . Culture of purulent specimens from the right chest wall abscess was also positive for A. fumigatus . Antifungal susceptibility testing by Etest showed minimum inhibitory concentrations (MICs) of 0.25 mg/L for voriconazole, 0.5 mg/L for amphotericin B, and 0.25 mg/L for isavuconazole, whereas higher MICs of 4.0 mg/L and 2.0 mg/L were observed for itraconazole and posaconazole, respectively. The patient received intravenous amphotericin B lipid complex combined with oral voriconazole, along with CT-guided drainage of the right iliopsoas abscess and repeated surgical debridement of the right chest wall and thigh abscesses. His clinical condition improved, and he was discharged after 52 days of hospitalization. Invasive aspergillosis with extensive skeletal muscle abscesses may occur in patients without recognized immunodeficiency and should be considered when recurrent multifocal abscesses fail to respond to appropriate antibacterial therapy. Timely initiation of antifungal therapy, together with aggressive surgical source control, may be critical for achieving favorable outcomes. Not applicable.
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