Difficult-to-treat central nervous system HIVunrelated cryptococcoma cured with voriconazole

R. Manfredi, S. Sabbatani, F. Chiodo (Bologna, I)

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Introduction: When considering cryptococcosis in a setting other than HIV disease,very few episodes are documented,and CNS cryptococcoma is an absolutely rare event. Case report: The authors describe an exceptional cerebral cryptococcoma occurred in a chronic nephropathic HIV-negative patient (p) suffering from homocystinuria, favorably cured with combined neurosurgery and voriconazole, after a limited response to high-dose fluconazole. In our case, after surgical intervention confirming a diagnosis of cryptococcoma, a first treatment line carried out with iv fluconazole did not lead to a sustained improvement, as the new antimycotic derivative voriconazole did at 400 mg/day, later in the disease course. After patient’s death due to a underlying progressive renalvascular disease,necropsy examination excluded any sign of infiltration due to Cryptococcus neoformans; voriconazole administration can be therefore considered eradicating. Discussion: When clinicians face an expansive cerebral mass, before starting therapy it is mandatory to establish an early presumptive diagnosis, in order to target antimicrobials and recommend eventual surgical drainage. Among a broad spectrum of infectious etiologies, it is suggested to take into consideration also of a fungal abscess, whose evolution would certainly be disadvantaged by symptomatic steroids (like happened in our p before that a specific diagnosis was confirmed), and/or antibacterial agents. In our case, both surgical and medical therapy of a brain cryptococcoma in anon-HIV-infected p have been performed: the surgical management helped to remove the majority of the brain abscess and to formulate the diagnosis, as well as to release an initial hydrocephalus. Also in HIV-infected p, cryptococcoma is a relatively rare feature, and it hardly appears isolated,and not associated with a meningeal cryptococcosis. As far as we know, there are no prior reports of primary cryptococcomas in chronic nephropatic, HIV-negative p, as well as in p with homocystinuria. Although being a rare occurrence, cerebral cryptococcoma may represent an emerging issue in the next future, because of its relationship with a broadening range of risk factors, including malignancies, neutropenia, end-organ failure, bone marrow and solid organ transplantation, and multiple primary-secondary immunodeficiency. With the mounting increase in the frequency of p with end-organ kidney disease,it is possible that also cryptococcosis may emerge as a more frequent complication.

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    • ECCMID 15th (2005)