Kim Hyung Kyung, Park Soonchan, Ryu Chang-Woo
Department of Pathology, Seoul National University Bundang Hospital Seongnam, Gyeonggi-do, Republic of Korea.
Department of Radiology, Kyung Hee University Hospital at Gangdong, College of Medicine, Kyung Hee University Seoul, Republic of Korea.
Am J Transl Res. 2022 Sep 15;14(9):6407-6411. eCollection 2022.
A 24-year-old patient with left eye proptosis and intermittent pain for 5 months was admitted to our hospital. Physical examination revealed neither extra ocular muscle limitations nor visual field defects. Magnetic resonance imaging (MRI) revealed a multicystic mass in the left extraconal space compressing the superior oblique muscle and adjacent frontal lobe. Layered hemorrhage was observed within the lesion in the 1-month follow-up MRI. Dynamic contrast enhanced imaging showed mild increased perfusion of the surrounding peripheral portion. Magnetic resonance spectroscopy showed an increased lactate/lipid peak of 1.3 ppm. Combined open and endonasal surgery was performed, and the final diagnosis was psammomatoid ossifying fibroma. The tumor was positive for vimentin, and negative for smooth muscle actin, S100 and epithelial membrane antigen. Despite its rarity, psammomatoid ossifying fibroma should be considered when multicystic lesions with peripheral enhancement near the orbit exhibit progressive inner hemorrhage.
一名24岁患者因左眼突出及间歇性疼痛5个月入院。体格检查未发现眼外肌受限或视野缺损。磁共振成像（MRI）显示左锥外间隙有一多房性肿块，压迫上斜肌和相邻额叶。在1个月后的随访MRI中，病变内观察到分层出血。动态对比增强成像显示周围外周部分灌注轻度增加。磁共振波谱显示乳酸/脂质峰在1.3 ppm处升高。进行了开放手术联合鼻内镜手术，最终诊断为砂粒体样骨化纤维瘤。肿瘤波形蛋白阳性，平滑肌肌动蛋白、S100和上皮膜抗原阴性。尽管砂粒体样骨化纤维瘤罕见，但当眼眶附近有外周强化的多房性病变出现进行性内出血时，应考虑该病。
