Blastomycosis in children is relatively infrequent. Further, osteomyelitis of the frontal bone, which prior to the antibiotic era occurred more frequently and was mostly due to pyogenic cocci, is rare today. We described a case of frontal osteomyelitis in a child due to blastomycosis which occurred in the absence of frontal sinuses. To our knowledge this rare combination of factors has not previously been reported.