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1型神经纤维瘤病患者发生闭角型青光眼的罕见病例。

Unusual case of angle closure glaucoma in a patient with neurofibromatosis type 1.

作者信息

Mantelli Flavio, Abdolrahimzadeh Solmaz, Mannino Giuseppe, Lambiase Alessandro

机构信息

IRCCS Fondazione Bietti, Azienda Policlinico Umberto I, Rome, Italy.

Ophthalmology Unit, DAI Testa Collo, Azienda Policlinico Umberto I, Rome, Italy.

出版信息

Case Rep Ophthalmol. 2014 Nov 19;5(3):386-91. doi: 10.1159/000369334. eCollection 2014 Sep-Dec.

DOI:10.1159/000369334
PMID:25762928
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC4342860/
Abstract

We report the case of a 29-year-old female patient who presented with an acute onset of anisocoria, blurred vision, nausea and severe left-sided headache. There was no history of trauma, drug abuse, or instillation of topical mydriatic compounds. The ocular history was negative for similar events. On presentation, her visual acuity was 0.2 in the left and 1.0 in the right eye with a +2.5 dpt sph. correction. Slit-lamp examination demonstrated a shallow anterior chamber as well as the presence of iris nodules in both eyes. These nodules were identified as Lisch nodules as the patient referred to the previous diagnosis as being neurofibromatosis type 1. A third nerve palsy was considered, but a brain MRI showed normal results. Her ocular motility was normal, but the left pupil was mydriatic and poorly reacting to light, with an associated raised intraocular pressure (IOP) of 38 mm Hg. An examination of the fellow eye was normal, with the IOP measuring 18 mm Hg. Gonioscopy of the right eye showed a narrow angle. On further anamnestic investigation, the patient revealed that the pain and the blurred vision begun in the morning while she was helping her mother in the garden. Finally, after showing the patient a picture of Datura flowers, which she recognized immediately, we made the unusual diagnosis of angle closure glaucoma by Datura, a well-known toxic plant with mydriatic properties. The patient was successfully treated with systemic acetazolamide and topical pilocarpine.

摘要

我们报告了一例29岁女性患者的病例,该患者急性起病,出现瞳孔不等大、视力模糊、恶心及严重的左侧头痛。患者无外伤史、药物滥用史或局部使用散瞳剂的情况。眼部病史中无类似发作史。就诊时,其左眼视力为0.2,右眼视力为1.0,矫正度数为+2.5 dpt 球镜。裂隙灯检查显示前房浅,双眼均有虹膜结节。由于患者提及既往诊断为1型神经纤维瘤病,这些结节被确定为Lisch结节。考虑存在动眼神经麻痹,但脑部MRI结果正常。她的眼球运动正常,但左侧瞳孔散大,对光反应差,伴有眼压升高至38 mmHg。对侧眼检查正常,眼压为18 mmHg。右眼房角镜检查显示房角狭窄。在进一步询问病史时,患者透露疼痛和视力模糊是在早上她在花园帮母亲干活时开始的。最后,在给患者看了曼陀罗花的图片后,她立刻认出了这种花,我们做出了不寻常的诊断:由曼陀罗引起的闭角型青光眼,曼陀罗是一种具有散瞳特性的知名有毒植物。患者通过全身应用乙酰唑胺和局部应用毛果芸香碱得到了成功治疗。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e373/4342860/9b26dc898a41/cop-0005-0386-g03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e373/4342860/18621f816a4d/cop-0005-0386-g01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e373/4342860/9bda0872832e/cop-0005-0386-g02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e373/4342860/9b26dc898a41/cop-0005-0386-g03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e373/4342860/18621f816a4d/cop-0005-0386-g01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e373/4342860/9bda0872832e/cop-0005-0386-g02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e373/4342860/9b26dc898a41/cop-0005-0386-g03.jpg

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Recurrent third nerve palsy as the presenting feature of neurofibromatosis 2.复发性第三对脑神经麻痹作为神经纤维瘤病 2 的表现特征。
J Neuroophthalmol. 2012 Dec;32(4):329-31. doi: 10.1097/WNO.0b013e3182726b49.
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Congenital ectropion uvea and mechanisms of glaucoma in neurofibromatosis type 1: new insights.先天性葡萄膜外翻和 1 型神经纤维瘤病的青光眼发病机制:新的认识。
[1型神经纤维瘤病中的锐角阻滞]
Ophthalmologe. 2019 Apr;116(4):384-386. doi: 10.1007/s00347-018-0770-5.
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A review of the role of ultrasound biomicroscopy in glaucoma associated with rare diseases of the anterior segment.超声生物显微镜在前节罕见疾病相关性青光眼治疗中作用的综述
Clin Ophthalmol. 2016 Jul 29;10:1453-9. doi: 10.2147/OPTH.S112166. eCollection 2016.
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