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A 15-year-old Caucasian male with Wiskott-Aldrich syndrome underwent an allogeneic bone marrow transplant. His post transplant course was complicated by graft-versus-host disease. The patient developed disseminated (lung, skin, brain and vertebral body) aspergillosis, with culture confirmation. The patient received 236 days of therapy with various agents including lipid formulations of amphotericin B, 17 days of voriconazole and 71 days of caspofungin prior to starting micafungin in combination with AmBisome. He eventually received a partial response.
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| Diagnosis:
Proven pulmonary aspergillosis - with probable dissemination to skin, brain and vertebral body based on radiographic evidence with lung tissue culture positive for A. fumigatus.
Response to antifungal
therapy:
Progression on Abelcet and intravenous itraconazole, with dissemination to the brain. Then stable on voriconazole for a few days and then very high dose Ambisome. Likely worsening of a pre-existing osteomyelitis lesion, despite improvement in cerebral aspergillosis, and so a switch to caspofungin. Stable on Caspofungin for 70 days then deterioration in cerebral aspergillosis. Then stable on micafungin and Ambisome, eventually showing a partial response to anti-fungal treatment.
Outcome:
This is a remarkable patient and series of events. Disseminated aspergillosis in the context of HSCT is usually fatal, especially if the brain is involved. This young man survived, but was slowly deteriorating after 8 months of various therapies including voriconazole and caspofungin. He improved on micafungin and AmBisome, but resolution of infection in his chest required an increase in dose of micafungin to 6.8mg/Kg per dose, given 3 times weekly with Ambisome After nearly 2 years of therapy with micafungin, he had not manifested any significant toxicity, and had a partial response.
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