ABSTRACT Objectives The aim of this study is to report an unusual form of penile cancer presentation associated with myiasis infestation, treatment options and outcomes. Materials and Methods We studied 10 patients with suspected malignant neoplasm of the penis associated with genital myiasis infestation. Diagnostic assessment was conducted through clinical history, physical examination, penile biopsy, larvae identification and computerized tomography scan of the chest, abdomen and pelvis. Clinical and pathological staging was done according to 2002 TNM classification system. Radical inguinal lymphadenectomy was conducted according to the primary penile tumor pathology and clinical lymph nodes status. Results Patients age ranged from 41 to 77 years (mean=62.4). All patients presented squamous cell carcinoma of the penis in association with myiasis infestation caused by Psychoda albipennis. Tumor size ranged from 4cm to 12cm (mean=5.3). Circumcision was conducted in 1 (10%) patient, while penile partial penectomy was performed in 5 (50%). Total penectomy was conducted in 2 (20%) patients, while emasculation was the treatment option for 2 (20%). All patients underwent radical inguinal lymphadenectomy. Prophylactic lymphadenectomy was performed on 3 (30%) patients, therapeutic on 5 (50%), and palliative lymphadenectomy on 2 (20%) patients. Time elapsed from primary tumor treatment to radical inguinal lymphadenectomy was 2 to 6 weeks. The mean follow-up was 34.3 months. Conclusion The occurrence of myiasis in the genitalia is more common in patients with precarious hygienic practices and low socio-economic level. The treatment option varied according to the primary tumor presentation and clinical lymph node status.