Management and Outcome in a Case of Cerebral Phaeohyphomycosis Due to Rhinocladiella Mackenziei in an Immunocompetent Individual
Moneera Aldraihem1, Weam AlMasoud1, Rewaa AlQurashi2, Oweida Aldosari3, Abdulrahman Abu-Amarah4
1Adult Neurology, 2Internal Medicine, 3Infectious Diseases, King Fahad Medical City, 4King Fahad Medical City
Objective:

To explore the challenges in diagnosing and treating a rare case of cerebral phaeohyphomycosis due to Rhinocladiella mackenziei in an immunocompetent individual, with the aim to improve our understanding of this uncommon fungal infection.

Background:
Cerebral phaeohyphomycosis is a rare fungal infection of the central nervous system, predominantly caused by dematiaceous, filamentous molds. Despite its rarity, the disease is associated with severe prognoses and high mortality rates, irrespective of the immune status of the affected individuals. In this report, we present a case of successful management of this complex infection.
Design/Methods:
NA
Results:
An immunocompetent male in his 60s, with a history of occupational soil exposure, presented with left-sided heaviness and intermittent occipital headaches. He was diagnosed with cerebral phaeohyphomycosis due to Rhinocladiella mackenziei. Initial management involved empirical therapy for brain abscess including antibiotics (ceftriaxone, vancomycin, metronidazole) and dexamethasone, along with antiseizure medication (levetiracetam). This was administered for 4 weeks. The treatment protocol was then adjusted based on fungal culture results to targeted therapy with intravenous liposomal amphotericin B. Subsequent MRI showed recurrence of the right parietal lesion and the emergence of a smaller lesion. This led to re-aspiration and addition of posaconazole to the regimen. This combination therapy was sustained for 3 months. The patient was then discharged and prescribed isavuconazole for outpatient management. This case increases the total number of survivors among the 42 reported cases of this condition to 9, with 29 having resulted in death and the outcomes of 4 cases remaining unknown.
Conclusions:
This case highlights the potential for successful management of cerebral phaeohyphomycosis with a combination of surgical and medical therapies, including the use of triazole-class antifungal drugs like posaconazole and isavuconazole. Given its rarity, with only 42 cases reported globally, further research is needed to inform strategies for managing this severe infection.
10.1212/WNL.0000000000206373