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Abstract：
BACKGROUND: Myositis ossificans circumscripta is a self-limiting, benign, ossifying lesion that can affect any type of soft tissue. It is most commonly found in muscles as a solitary lesion. A history of recent trauma has been reported in approximately 50% of cases. Clinically, MOC presents as a painful swelling, which rapidly increases in size. The pain and inflammatory symptoms spontaneously disappear after approximately 2-6 weeks, and the mass stabilizes or decreases. Radiologically, myositis ossificans circumscripta can be divided into two phases. The first is the acute phase, which is followed by the mature phase 2-6 weeks later. During the acute phase, the radiological aspect does not show any specific abnormality. In the mature phase, plain radiographs and computed tomography show blurred calcifications around a hypodense center. We describe here the first case of myositis ossificans circumscripta, with appropriate follow-up, occurring during sunitinib exposure.
METHODS: We report a case of myositis ossificans circumscripta in a 34-year-old man (ethnicity unknown) receiving sunitinib for metastatic alveolar soft part sarcoma of the left thigh after surgery and radiotherapy. Four months after the first dose of sunitinib, the patient experienced painful swelling in the surgical scar area. Magnetic resonance imaging showed diffuse and marked edema of the anterior compartment of the thigh, without nodular lesions circumscribing a central core, and without bone signal abnormality. The increased visibility of the intermuscular fascia and convergence of normal muscle fibers (black hole effect), without the displacement seen in tumors, were suggestive of myositis. Therefore, antiangiogenic treatment was discontinued, and the symptoms rapidly resolved within a few days. Three weeks after the discontinuation of sunitinib, the inflammatory findings completely disappeared. Two months after the diagnosis of myositis ossificans circumscripta, plain radiographs and computed tomography showed an extensive calcified mass measuring > 12 cm. The continuation of favorable clinical outcomes was confirmed.
CONCLUSIONS: To the best of our knowledge, this is the first case of myositis ossificans circumscripta with appropriate follow-up occurring during sunitinib exposure. Owing to multimodal treatment of sarcoma, we cannot rule out the radiotherapy and surgery causality.
METHODS: We report a case of myositis ossificans circumscripta in a 34-year-old man (ethnicity unknown) receiving sunitinib for metastatic alveolar soft part sarcoma of the left thigh after surgery and radiotherapy. Four months after the first dose of sunitinib, the patient experienced painful swelling in the surgical scar area. Magnetic resonance imaging showed diffuse and marked edema of the anterior compartment of the thigh, without nodular lesions circumscribing a central core, and without bone signal abnormality. The increased visibility of the intermuscular fascia and convergence of normal muscle fibers (black hole effect), without the displacement seen in tumors, were suggestive of myositis. Therefore, antiangiogenic treatment was discontinued, and the symptoms rapidly resolved within a few days. Three weeks after the discontinuation of sunitinib, the inflammatory findings completely disappeared. Two months after the diagnosis of myositis ossificans circumscripta, plain radiographs and computed tomography showed an extensive calcified mass measuring > 12 cm. The continuation of favorable clinical outcomes was confirmed.
CONCLUSIONS: To the best of our knowledge, this is the first case of myositis ossificans circumscripta with appropriate follow-up occurring during sunitinib exposure. Owing to multimodal treatment of sarcoma, we cannot rule out the radiotherapy and surgery causality.
摘要：
背景：骨化性肌炎是一种自我限制的，良性,骨化性病变可影响任何类型的软组织。它最常见于肌肉中作为孤立性病变。据报道，约有50％的病例有近期创伤史。临床上,MOC表现为疼痛性肿胀，尺寸迅速增加。疼痛和炎症症状在大约2-6周后自发消失，质量稳定或减少。放射学上,骨化性肌炎可分为两个阶段。首先是急性期，随后是2-6周后的成熟期。在急性期,放射学方面没有显示任何特定的异常。在成熟阶段,X线平片和计算机断层扫描显示低密度中心周围有模糊的钙化。我们在这里描述了第一例骨化性肌炎，通过适当的后续行动，发生在舒尼替尼暴露期间。
方法：我们报告一例34岁男性（种族不详）的骨化性肌炎病例，在手术和放疗后接受舒尼替尼治疗左大腿转移性肺泡软组织肉瘤。第一次服用舒尼替尼四个月后，患者在手术疤痕区域经历了疼痛的肿胀。磁共振成像显示大腿前室弥漫性和明显的水肿，没有围绕中央核心的结节性病变，并且没有骨信号异常。肌肉间筋膜的可见性增加和正常肌纤维的会聚（黑洞效应），没有在肿瘤中看到的移位，提示肌炎。因此,停止抗血管生成治疗,症状在几天内迅速缓解。舒尼替尼停药三周后，炎症的发现完全消失了.诊断为骨化性肌炎后两个月，X线片和计算机断层扫描显示大范围钙化肿块，测量>12cm。证实了良好临床结局的延续。
结论：据我们所知，这是首例局限性骨化性肌炎病例,在舒尼替尼暴露期间进行了适当的随访.由于肉瘤的多模式治疗，不能排除放疗和手术的因果关系.
方法：我们报告一例34岁男性（种族不详）的骨化性肌炎病例，在手术和放疗后接受舒尼替尼治疗左大腿转移性肺泡软组织肉瘤。第一次服用舒尼替尼四个月后，患者在手术疤痕区域经历了疼痛的肿胀。磁共振成像显示大腿前室弥漫性和明显的水肿，没有围绕中央核心的结节性病变，并且没有骨信号异常。肌肉间筋膜的可见性增加和正常肌纤维的会聚（黑洞效应），没有在肿瘤中看到的移位，提示肌炎。因此,停止抗血管生成治疗,症状在几天内迅速缓解。舒尼替尼停药三周后，炎症的发现完全消失了.诊断为骨化性肌炎后两个月，X线片和计算机断层扫描显示大范围钙化肿块，测量>12cm。证实了良好临床结局的延续。
结论：据我们所知，这是首例局限性骨化性肌炎病例,在舒尼替尼暴露期间进行了适当的随访.由于肉瘤的多模式治疗，不能排除放疗和手术的因果关系.
