Introduction Blastomycosis is an endemic mycosis in the United States known to primarily cause pneumonia. However, dissemination to different organs including the musculoskeletal system has been described. Case report We report a case of mandibular blastomycosis in a healthy patient with no evidence of lung involvement. A 28 year-old female presented with recurrent right mandibular osteomyelitis despite courses of antibiotics and surgical debridement. She eventually underwent right hemimandibulectomy. Budding yeasts visualized on Gomori Methenamine-Silver (GMS) and Periodic acid-Schiff (PAS) were morphologically consistent with Blastomyces dermatitidis, and intra-operative cultures showed growth of mold identified as B. dermatitidis by DNA probe. She was placed on a prolonged course of itraconazole with clinical improvement. We also reviewed the literature and found 5 cases of similar presentation which we briefly summarized in this present case report. Conclusion Blastomycosis should be considered in patients with recurrent or persistent mandibular osteomyelitis even in immunocompetent individuals.