BACKGROUND Mucormycosis, a cause of opportunistic infections in immunocompromised patients, is rarely identified in the penis. The literature often describes drastic surgical interventions or rapid patient demise, with scant mention of surgical management specifics. The objective of this report is to detail our experience with this unique infection and highlight the utility of intraoperative frozen margins during surgical management. CASE REPORT Herein, we describe successful treatment of a 55-year-old man with biopsy-proven B-cell acute lymphoblastic leukemia (B-ALL) undergoing Hyper-CVAD (Cyclophosphamide, Vincristine, Adriamycin, and Dexamethasone) therapy who initially presented with an asymptomatic violaceous lesion of the penis. Differential diagnoses ranged from infectious, vasculogenic, and pharmacologic in nature. Ultimately, a punch biopsy tissue culture confirmed angioinvasive fungal infection with Rhizopus and Fusarium species. Initial debridement combined with intravenous antifungal therapy was unsuccessful. However, partial penectomy with use of intraoperative frozen margins, a 5-week course of antifungal therapy, and continued B-ALL treatment allowed effective and lasting resolution of the infection, with partial penile preservation. CONCLUSIONS This case supports a multidisciplinary approach as the primary treatment for penile angioinvasive fungal infections. This includes treatment of the underlying immunocompromising condition, appropriate intravenous antifungal therapy, and urgent operative debridement. This report highlights the importance of utilizing intraoperative frozen sections to ensure negative margins and to optimize overall tissue sparing in this anatomically sensitive area.