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表现为托洛萨-亨特综合征的伴有新型致病性神经纤维瘤病 1 型突变的巨大血栓形成的海绵窦内颈动脉动脉瘤:病例报告及文献复习。

Giant thrombosed intracavernous carotid artery aneurysm presenting as Tolosa-Hunt syndrome in a patient harboring a new pathogenic neurofibromatosis type 1 mutation: a case report and review of the literature.

机构信息

Neuroradiology Unit, Department of Clinical and Experimental Medicine and Surgery.

Radiology Unit, Department of Medical, Surgical, Neurological, Metabolic and Aging Sciences.

出版信息

Neuropsychiatr Dis Treat. 2014 Jan 20;10:135-40. doi: 10.2147/NDT.S49784. eCollection 2014.

Abstract

Neurofibromatosis type 1 (NF1) is a relatively common single-gene disorder, and is caused by heterozygous mutations in the NF1 gene that result in a loss of activity or in a nonfunctional neurofibromin protein. Despite the common association of NF1 with neurocutaneous features, its pathology can extend to numerous tissues not derived from the neural crest. Among the rare cerebrovascular abnormalities in NF1, more than 85% of cases are of purely occlusive or stenotic nature, with intracranial aneurysm being uncommon. Predominantly, the aneurysms are located in the internal carotid arteries (ICAs), being very rare bilateral aneurysms. This report describes a very unusual case of fusiform aneurysms of both ICAs in a Caucasian NF1 patient, with a new pathogenic intragenic heterozygous deletion of the NF1 gene, presenting at age 22 years with Tolosa-Hunt syndrome, because of partial thrombosis of the left giant intracavernous aneurysm. Medical treatment with anticoagulant therapy allowed a good outcome for the patient. In conclusion, early identification of cerebral arteriopathy in NF1 and close follow-up of its progression by neuroimaging may lead to early medical or surgical intervention and prevention of significant neurologic complications.

摘要

神经纤维瘤病 1 型(NF1)是一种相对常见的单基因疾病,由 NF1 基因突变引起杂合子,导致神经纤维瘤蛋白失活或无功能。尽管 NF1 通常与神经皮肤特征相关,但它的病理学可以扩展到许多并非源自神经嵴的组织。在 NF1 中罕见的脑血管异常中,超过 85%的病例为单纯闭塞或狭窄性,颅内动脉瘤并不常见。主要的是,动脉瘤位于颈内动脉(ICAs),非常罕见的是双侧动脉瘤。本报告描述了一例非常不寻常的 NF1 患者双侧颈内动脉梭形动脉瘤病例,该患者存在 NF1 基因的新致病性种内杂合性缺失,22 岁时因左侧巨大海绵窦内动脉瘤部分血栓形成而出现托洛萨-亨特综合征。由于抗凝治疗,患者预后良好。总之,早期识别 NF1 中的脑动脉病变,并通过神经影像学密切随访其进展,可能导致早期进行药物或手术干预,预防严重的神经并发症。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2847/3901779/bcbea8dbdf78/ndt-10-135Fig1.jpg

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