Department of Ophthalmology, Kyung Hee University Hospital, Kyung Hee University School of Medicine, 892, Dongnam-ro, Gangdong-gu, Seoul, Republic of Korea, 05278.
Department of Medicine, Graduate School, Kyung Hee University, 23, Kyungheedae-ro, Dongdaemun-gu, Seoul, Republic of Korea, 02447.
BMC Ophthalmol. 2020 Aug 17;20(1):334. doi: 10.1186/s12886-020-01604-9.
Neuromuscular hamartoma is a very rare tumor; with only five cases reported in the orbit. It often occurs in infants and young children and involves large peripheral nerves, but there has been only few reports of occurrence in the orbit and adults.
This paper describes a 70-year-old man with an incidental orbital mass detected by an imaging test and who later developed associated symptoms. The mass was diagnosed as neuromuscular hamartoma. Superior rectus muscle recession and inferior rectus muscle resection were performed in the right eye for hypertropia secondary to postoperative inferior rectus muscle palsy. Hypertropia in the right eye and diplopia improved after surgery, and regular follow-up is underway.
This is the first case of an incidentally detected orbital mass diagnosed by histologic examination as a neuromuscular hamartoma in an older patient whose proptosis progressed after a long period of inactivity.
神经肌肉性错构瘤是一种非常罕见的肿瘤,仅在眼眶中报告过五例。它常发生于婴儿和幼儿,累及大的外周神经,但在眼眶和成年人中仅少数有报道。
本文描述了一例 70 岁男性,因影像学检查偶然发现眶内肿块,随后出现相关症状。该肿块被诊断为神经肌肉性错构瘤。因术后下直肌麻痹导致的斜视,右眼行上直肌后徙和下直肌切除术。右眼的斜视和复视在手术后得到改善,目前正在进行定期随访。
这是首例经组织学检查诊断为神经肌肉性错构瘤的偶然发现的眶内肿块的病例,该患者的眼眶病变进展发生于长时间无进展之后。