Background: Gonadal vein thrombosis (GVT) has been reported in association with malignancy and pelvic inflammatory conditions. Patients who develop GVT often require systemic anticoagulation to reduce the risk of pulmonary embolism and other local and distant thromboembolic effects. As the gonadal vein courses from the pelvis toward its outlet in the upper abdomen, its intimate relationship to the ureter in the setting of vascular pathology may pose a risk for urinary obstruction in the adult. We are reporting a rare case of GVT leading to ureteral obstruction and acute kidney injury (AKI) in a young otherwise healthy male and provide a review of similar literature. Case Presentation: We describe a case of an otherwise healthy 29-year-old African American adult male presenting with acute diverticulitis and associated left GVT with no evidence of hypercoagulability, leading to ureteral obstruction, hydronephrosis, and AKI. Treatment with ureteral stent placement, endovascular intervention, and systemic anticoagulation led to resolution of his condition. Conclusion: This report details a rare case of confirmed GVT in an adult male with resultant urinary obstruction. Decompression of the collecting system and treatment of the significant venous obstruction with surgical intervention, combined with medical systemic anticoagulation, were effective in reversing the underlying cause.