Background Primary leiomyosarcoma(LMS) very rarely arises in the broad ligament. Metastasis is usually to lungs, liver, and so on, but seldomly to adrenal gland. Clinical presentation and imaging findings of metastatic broad ligament LMS are often nonspecific.The diagnosis is mainly based on histopathology and immunohistochemistry findings, and surgical resection is the preferred treatment method. Case presentation We present an extremely rare case of 49-year-old woman with recurrent broad ligament LMS metastasizing to right adrenal gland. Previously, she was diagnosed with broad ligament LMS and underwent laparoscopic hysterectomy plus bilateral salpingo-oophorectomy. The disease-free interval was 3 years until metastases were found. Computerized tomography (CT) performed, revealed right adrenal lesion. Adrenal lesion was completely resected. Pathological examination showed both the primary and the secondary LMS to have features of well-differentiated tumors. Conclusions Up to know, we highlight the uncommon case of broad ligament LMS with the adrenal metastatis. It is indicated that well-differentiated of broad ligament LMS also have the malignant potential of distant metastasis even if the patient have performed surgical treatment. We hope this uncommon case helps clinicians gain further understanding of metastasized broad ligament LMS.
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