1. INTRODUCTIONThromboembolism is a common and potentially life-threatening complication that can arise from various medical conditions, including malignancy and a spectrum of benign diseases. Adenomyosis, characterized by the presence of endometrial glands and stroma within the myometrium, is a benign uterine disorder that typically presents in middle-aged postpartum women with symptoms of menorrhagia, dysmenorrhea, and chronic pelvic pain. The true prevalence of adenomyosis remains unknown, although estimates derived from patients referred for pelvic imaging suggest a range of 20% to 34%.1 The association between adenomyosis and thrombosis was first described by Kupryjanczyk in 1991.2 Subsequently, several isolated case reports and case series have documented the occurrence of ischemic stroke3-20 and pulmonary embolism (PE)21, 22 in patients with adenomyosis, suggesting a possible association with hypercoagulability induced by this disorder. However, there are few reports of cases complicated by isolated deep vein thrombosis (DVT). 23, 24 In this report, we present a case of isolated distal deep venous thromboembolism (IDDVT) in a patient with a 1.5-year history of adenomyosis. We also conducted a comprehensive literature review to explore the clinical features, treatment approaches, and prognosis of thrombotic complications associated with this condition.