Handa Tadamasa, Asanuma Kunihiro, Yuasa Hiroto, Nakamura Tomoki, Hagi Tomohito, Uchida Katsunori, Sudo Akihiro
Department of Orthopedic Surgery, Mie University School of Medicine, Tsu, Japan.
Department of Oncologic Pathology, Mie University School of Medicine, Tsu, Japan.
Case Rep Oncol. 2024 Oct 31;17(1):1266-1272. doi: 10.1159/000541480. eCollection 2024 Jan-Dec.
Osteochondromas are benign tumors that arise primarily in the metaphyseal region of long bones. The malignant transformation rate is estimated to be less than 1% and 1-3% in solitary and multiple osteochondromas, respectively. Transformation to osteosarcoma is very rare. Little information is available on treatment or outcome. A rare case of osteosarcoma arising from hereditary multiple osteochondromas of the right iliac bone is reported.
A 66-year-old woman presented with recurrent right abdominal pain. Computed tomography (CT) showed a mass protruding into the pelvic cavity, 9 cm × 7 cm × 7 cm, with bone destruction and internal calcification in the right iliac bone. A CT-guided biopsy was performed, and the diagnosis was osteosarcoma. After one course of chemotherapy with doxorubicin and ifosfamide, extensive resection of the tumor was performed. The pathology showed proliferation of highly pleomorphic dysplastic cells with bone formation inside the tumor just below the osteochondroma tissue, which led to the diagnosis of osteosarcoma arising from the osteochondroma. Three years after surgery, there was no evidence of recurrence or metastasis, and the patient was able to walk unassisted.
A case of osteosarcoma arising from an iliac lesion of hereditary multiple osteochondromas was described. Although no recurrence or metastasis has been observed 3 years after surgery, further follow-up is necessary due to the short time after surgery.
骨软骨瘤是主要发生于长骨干骺端的良性肿瘤。孤立性和多发性骨软骨瘤的恶性转化率估计分别低于1%和1%-3%。转变为骨肉瘤非常罕见。关于其治疗或预后的信息很少。本文报道了一例罕见的起源于右髂骨遗传性多发性骨软骨瘤的骨肉瘤病例。
一名66岁女性因反复右腹痛就诊。计算机断层扫描（CT）显示盆腔内有一肿块突出，大小为9 cm×7 cm×7 cm，右髂骨有骨质破坏及内部钙化。进行了CT引导下活检，诊断为骨肉瘤。在使用阿霉素和异环磷酰胺进行一个疗程的化疗后，对肿瘤进行了广泛切除。病理显示肿瘤内高度多形性发育异常细胞增殖，在骨软骨瘤组织下方有骨形成，由此诊断为由骨软骨瘤引起的骨肉瘤。术后三年，无复发或转移迹象，患者能够独立行走。
描述了一例起源于遗传性多发性骨软骨瘤髂骨病变的骨肉瘤病例。尽管术后三年未观察到复发或转移，但由于术后时间较短，仍需进一步随访。
