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12 年后因蝶骨翼发育不良出现症状性眼球内陷的 1 型神经纤维瘤病患者:病例报告及文献复习。

Symptomatic enophthalmos due to sphenoid wing dysplasia appearing over 12 years in a patient with neurofibromatosis type 1: a case report and literature review.

机构信息

Department of Neurology, Tokyo Metropolitan Children's Medical Center, Tokyo, Japan.

Department of Radiology, Tokyo Metropolitan Children's Medical Center, Tokyo, Japan.

出版信息

J AAPOS. 2022 Aug;26(4):210-214.e1. doi: 10.1016/j.jaapos.2022.02.013. Epub 2022 May 21.

Abstract

Sphenoid wing dysplasia (SWD) is a common orbital complication of neurofibromatosis type 1 (NF1). However, enophthalmos associated with SWD is extremely rare, and details of its natural history are unclear. We present the case of a 14-year-old boy with an early childhood diagnosis of NF1 presenting with left blepharophimosis and enophthalmos for several months. Imaging demonstrated enlargement of the left lateral SWD, progression of the posteromedial deviation of the orbital contents, and sphenoid/ethmoid sinus deformation due to left temporal lobe compression over 12 years. Two characteristic changes were revealed on imaging: enlargement of the middle cranial fossa and deformation of the sphenoid/ethmoid sinuses. The orbital contents were compressed by the intracranial pressure of the temporal lobe and were displaced posteromedially into the space created by the deformed sphenoid/ethmoid sinuses. Because orbital symptoms can gradually become apparent over years with the progression of SWD and skeletal growth, long-term follow-up of orbital symptoms is recommended in patients with NF1.

摘要

蝶骨翼发育不良(SWD)是神经纤维瘤病 1 型(NF1)的常见眼眶并发症。然而,与 SWD 相关的眼球内陷极为罕见,其自然病史的细节尚不清楚。我们报告了一例 14 岁男孩,他在幼儿时期被诊断出患有 NF1,表现为左眼眼睑退缩和眼球内陷数月。影像学显示左侧外侧蝶骨翼发育不良增大,眶内容物向中后移位,蝶骨/筛窦变形,这是由于 12 年来左颞叶受压所致。影像学显示出两个特征性变化:颅中窝增大和蝶骨/筛窦变形。眶内容物被颞叶颅内压压缩,向中后移位到变形的蝶骨/筛窦所形成的空间。由于 SWD 和骨骼生长的进展,眼眶症状可能会在数年内逐渐显现,因此建议 NF1 患者进行长期的眼眶症状随访。

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