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一例先天性特发性眼外肌肥大

A Case of Congenital Idiopathic Enlargement of Extraocular Muscles.

作者信息

Negishi Takashi, Nakao Shintaro

机构信息

Department of Ophthalmology, Juntendo University, Tokyo, JPN.

Department of Ophthalmology, Faculty of Medicine, Juntendo University, Tokyo, JPN.

出版信息

Cureus. 2024 May 2;16(5):e59496. doi: 10.7759/cureus.59496. eCollection 2024 May.

Abstract

The purpose of this case report is to report a case of congenital idiopathic enlargement of extraocular muscles. A four-month-old girl showed limitation of adduction and supraduction in the right eye. A computerized axial tomography (CAT) scan revealed hypertrophy of the lateral rectus muscle and inferior rectus muscle of the right eye. Thyroid hormone and antibody levels were normal. No inflammatory findings on magnetic resonance imaging (MRI). A traction test under general anesthesia revealed a strong limitation of supraduction and a mild limitation of adduction. Therefore, the inferior rectus muscle was recessed 4.5 mm at the age of six months. A partial biopsy of the inferior rectus showed no inflammatory cell infiltration. After the first surgery, the patient's limitation of supraduction improved, but the limitation of adduction persisted. So, a 5 mm recession of the right lateral rectus muscle was added at one year and one month. However, the hypertropia of the sound eye became stronger after treatment of amblyopia. Because of the strong limitation of supraduction, tenotomy of the inferior rectus was performed at the age of six years. Postoperatively, no impairment of infraduction occurred, and the limitation of supraduction was mildly improved. Since the findings on MRI were not changed through our observation period, we concluded that the patient had idiopathic external ophthalmoplegia.

摘要

本病例报告的目的是报告一例先天性特发性眼外肌肥大。一名4个月大的女孩右眼内收和上转受限。计算机断层扫描(CAT)显示右眼外直肌和下直肌肥大。甲状腺激素和抗体水平正常。磁共振成像(MRI)未发现炎症表现。全身麻醉下的牵引试验显示上转严重受限,内收轻度受限。因此,在6个月大时对下直肌进行了4.5毫米的后徙术。下直肌部分活检未发现炎性细胞浸润。首次手术后,患者的上转受限有所改善,但内收受限仍然存在。所以,在1岁1个月时又对右眼外直肌进行了5毫米的后徙术。然而,弱视治疗后健眼的上斜视变得更严重。由于上转严重受限,在6岁时对下直肌进行了腱切断术。术后,下转未出现损伤,上转受限稍有改善。由于在我们的观察期内MRI检查结果未改变,我们得出结论,该患者患有特发性外展神经麻痹。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/cecb/11143438/5db89c50f2ef/cureus-0016-00000059496-i01.jpg

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