Infections affecting the central nervous system caused by Blastomyces dermatitidis are rare but curable. We describe a case of a 24-year-old man who presented to the emergency department with progressive bilateral lower extremity weakness over 1 month. On the day of admission, he had minimal muscle strength and was hyperreflexic in the lower extremities. Sensation, however, was intact. Skin examination revealed annular, raised, crusted lesions on his face and legs. A magnetic resonance imaging (MRI) scan showed marrow replacement of the T7 and T8 vertebral bodies and an epidural mass with cord compression. A chest radiograph showed an infiltrate, and a subsequent needle biopsy revealed yeast resembling B. dermatitidis. A skin biopsy was then obtained, and the culture grew out B. dermatitidis. He received 4 weeks of amphotericin B lipid complex (total of 6 grams), followed by oral itraconazole. After 1 week on antifungals, he was able to walk with a walker and the skin lesions virtually resolved. At 5 months he was ambulatory and riding a bicycle daily. Blastomycosis should be included in the differential diagnosis of epidural masses.