Primary uterine osteosarcoma arising in a leiomyoma with rapid local recurrence: A case report

• Published in: Gynecologic oncology reports 2022-12, Vol.44, p.101102, Article 101102
• Main Authors: Ruhotina, Merima, Kukla, Joanna, Wilcox, Annemieke, Murphy, Colleen, Menderes, Gulden
• Format: Article
• Language: English
• Subjects: Case Report; Extraskeletal osteosarcoma; Soft tissue neoplasms; Uterine osteosarcoma
• ISSN: 2352-5789; 2352-5789
• DOI: 10.1016/j.gore.2022.101102

•Primary uterine osteosarcoma is an extremely rare and aggressive neoplasm.•There is no clear consensus about the optimal systemic treatment.•Chemotherapy treatment options can be deduced from previous studies of bone and soft tissue sarcomas. Extraskeletal osteosarcoma is an extremely rare malignant neoplasm. Literature regarding primary osteosarcoma of the uterus is confined to only a small number of case reports. Case: A 57-year-old female with a history of uterine fibroids presented to the emergency department with abdominal pain. Imaging was notable for an enlarged uterus with a 15 cm calcified fibroid extending along the posterior uterus. The patient underwent a laparotomy for total hysterectomy and bilateral salpingo-oophorectomy. Pathological evaluation of the specimen yielded mesenchymal proliferation with osteoid formation and tumor cells with densely eosinophilic cytoplasmresembling osteoblasts with a final diagnosis of primary uterine osteosarcoma. Multidisciplinary tumor board recommended against adjuvant treatment, given the lack of evidence for improved outcomes for early-stage uterine sarcomas. The patient was followed up with surveillance visits every-three months, entailing physical examination and computed tomography(CT) scans. Unfortunately, she had locoregional oligometastatic recurrence of her disease at 1-year follow up. Primary uterine osteosarcoma is an extremely rare and aggressive neoplasm with limited understanding regarding optimal treatment options.