A 33-year old male Tunisian developed, without any apparent cause, an abscess of the brainstem which was shown by various mycological investigations to be due to a Blastomyces dermatitidis infection. This case is of interest not only because of the exceptional location of the abscess in the brainstem, but also because of its treatment: this was the first case where the fluconazole-flucytosine combination was used against blastomycosis with fully satisfactory results after a 28-month follow-up.