Primary cutaneous infection with Chrysosporium, a saprophytic fungus commonly found in soil, is believed to be very rare, with only two previously reported cases. We present a case of localized cutaneous Chrysosporium in an immunocompromised heart transplant patient. Considering that the histology of the skin in this case is superimposable on that seen in pulmonary Chrysosporium known as adiaspiromycosis, we regard the cutaneous variant in the absence of pulmonary disease as a distinct dermatologic entity. The low frequency of reports of primary cutaneous Chrysosporium infection suggests either underreporting of this diagnosis in the literature, or misidentification of this fungus as another more common mycotic species sharing morphologic similarities. By amplifying our understanding of Chrysosporium infection in the skin, this disorder will be easier to identify and treat.