Atypical femoral fracture related to a long-term bisphosphonate therapy has commonly been reported; however, a fracture at the site other than the femur has rarely been reported to date. Herein, we report a case of a patient on long-term bisphosphonate therapy who presented atypical tibial insufficiency fracture at the anterolateral aspect of diaphysis, without trauma. We, for the first time in Korea, present this case with a literature review.