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Skull base osteomyelitis caused by Mycobacterium abscessus and Pseudozyma aphidis in an immunocompetent patient – a case report

Introduction: Skull base osteomyelitis (SBO) is a rare clinical entity that often remains unrecognized or misdiagnosed and is usually reported in elderly or immunocompromised patients. Mycobacterium abscessus complex is a group of rapidly growing, multidrug-resistant, nontuberculous mycobacteria (NMT) responsible mostly of healthcare-associated infections. Pseudozyma aphidis is an opportunistic yeast that rarely causes disease in humans. Case presentation: In 2014 and 2015, a 32-year- old, previously healthy female underwent three surgeries for inverted papilloma of the right ethmoid region. She was asymptomatic until August 2018, when she developed hyposmia and galactorrhea. In September 2018, right sphenoid sinus inflammation was diagnosed. Mycobacterium abscessus and Pseudozyma aphidis grew from the tissue culture. She was treated with oral voriconazole, levofloxacin, doxycycline, and trimethoprim-sulfamethoxazole for four months. Despite the antibiotic regimen, she developed pain behind her right eye and sixth cranial nerve palsy. Cranial MR demonstrated sphenoid sinusitis, osteomyelitis of the clivus, and hypophysitis. She was admitted to UHID, Zagreb, Croatia. A combination of imipenem, azithromycin, amikacin, and liposomal amphotericin B was initiated, and after four weeks, amphotericin B was switched to voriconazole. Additional microbiological evaluation confirmed M. abscessus subspecies massiliense with a non-functional erm gene, indicating the possibility of macrolide treatment. Treatment was successful, leading to clinical and radiological improvement. After discharge, outpatient antimicrobial therapy was continued. In total, the patient received antimicrobial treatment for 12 months. Two years after therapy completion, the patient is asymptomatic and completely recovered. Conclusion: This is a rare case of SBO caused by Mycobacterium abscessus and Pseudozyma aphidis in a young immunocompetent patient. Even though SBO is rare, it should be considered in a case of headache accompanied by signs of hypopituitarism. Uncommon pathogens should be examined. This report highlights the importance of an interdisciplinary approach to difficult-to-treat infections, leading to faster diagnosis, earlier pathogen-oriented therapy, and favorable outcomes.

Skull base ; osteomyelitis ; Mycobacterium abscessus ; Pseudozyma aphidis ; immunocompetent patient

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