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一例严重巩膜炎如何导致严重的全身后遗症

How a Devastating Case of Sclerokeratitis Ended up with Serious Systemic Sequelae.

作者信息

Bataillie Sophie, Van Ginderdeuren Rita, Van Calster Joachim, Foets Beatrijs, Delbeke Heleen

机构信息

Ophthalmology, University Hospital Leuven, Leuven, Belgium.

出版信息

Case Rep Ophthalmol. 2020 Jul 14;11(2):348-355. doi: 10.1159/000508326. eCollection 2020 May-Aug.

DOI:10.1159/000508326
PMID:32884549
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC7443666/
Abstract

A 35-year old soft contact lens wearer with a proven bilateral keratitis developed a nodular scleritis. Based on the stepladder approach described by Iovieno et al. [Ophthalmology. 2014 Dec;121(12):2340-7], nonsteroidal anti-inflammatory drugs, methylprednisolone, and later azathioprine were added to the antiamoebic treatment. Unfortunately, there was further deterioration and an endophthalmitis developed. Unbearable pain and concerns of spread to the brain urged an enucleation. Histopathological examination confirmed cysts in the cornea, sclera, retina, choroid, and vitreous body. As a side effect of the immunosuppressive treatment, the patient developed myopathy, pulmonary aspergillosis, and an avascular necrosis of the hip. Scleritis is a devastating complication of keratitis with a poor prognosis and a high enucleation rate. sclerokeratitis is, due to cyst-free biopsies, mostly assigned to an immune-mediated mechanism, justifying the use of immunosuppressive treatment. Scleritis in our case contributed to the extracorneal spread of . Our case is the first documented extracorneal spread of without previous surgery. Extracorneal spread of should be considered, even in the case of false-negative biopsies. We strongly recommend serial sections of the retrieved scleral specimen in case of negative histopathological examination to exclude an infection. Even when an immune-mediated scleritis is suspected, systemic immunosuppressive treatment should always be used with the greatest caution. Awareness of the side effects and monitoring by an experienced physician is mandatory.

摘要

一名35岁的软性隐形眼镜佩戴者,已确诊双侧角膜炎,随后发展为结节性巩膜炎。根据Iovieno等人描述的阶梯式治疗方法[《眼科学》。2014年12月;121(12):2340 - 7],在抗阿米巴治疗中添加了非甾体抗炎药、甲基泼尼松龙,随后又添加了硫唑嘌呤。不幸的是,病情进一步恶化,发展为眼内炎。难以忍受的疼痛以及担心感染扩散至脑部促使进行眼球摘除术。组织病理学检查证实角膜、巩膜、视网膜、脉络膜和玻璃体中有囊肿。作为免疫抑制治疗的副作用,患者出现了肌病、肺曲霉菌病和髋部缺血性坏死。巩膜炎是角膜炎的一种毁灭性并发症,预后不良且眼球摘除率高。由于活检未发现囊肿,巩膜角膜炎大多归因于免疫介导机制,这证明了免疫抑制治疗的合理性。我们病例中的巩膜炎导致了[病原体名称未给出]的角膜外扩散。我们的病例是首例有记录的未经先前手术的[病原体名称未给出]角膜外扩散。即使活检结果为假阴性,也应考虑[病原体名称未给出]的角膜外扩散。如果组织病理学检查结果为阴性,我们强烈建议对获取的巩膜标本进行连续切片,以排除感染。即使怀疑是免疫介导的巩膜炎,全身免疫抑制治疗也应始终极其谨慎地使用。必须了解副作用并由经验丰富的医生进行监测。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5b31/7443666/8eee69926dbc/cop-0011-0348-g03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5b31/7443666/9b0d777bc746/cop-0011-0348-g01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5b31/7443666/b3736f70fca8/cop-0011-0348-g02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5b31/7443666/8eee69926dbc/cop-0011-0348-g03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5b31/7443666/9b0d777bc746/cop-0011-0348-g01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5b31/7443666/b3736f70fca8/cop-0011-0348-g02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5b31/7443666/8eee69926dbc/cop-0011-0348-g03.jpg

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本文引用的文献

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Int J Ophthalmol. 2019 Dec 18;12(12):1966-1971. doi: 10.18240/ijo.2019.12.20. eCollection 2019.
2
Osteonecrosis of the Femoral Head.股骨头坏死
Orthop Clin North Am. 2019 Apr;50(2):139-149. doi: 10.1016/j.ocl.2018.10.001. Epub 2019 Feb 12.
3
Orbital implant exposure after Acanthamoeba panophthalmitis.棘阿米巴全眼球炎后的眼眶植入物暴露
Am J Ophthalmol Case Rep. 2018 Jan 31;10:48-50. doi: 10.1016/j.ajoc.2018.01.043. eCollection 2018 Jun.
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Acanthamoeba in the eye, can the parasite hide even more? Latest developments on the disease.棘阿米巴眼中的寄生虫,还能藏得更深吗?这种疾病的最新进展。
Cont Lens Anterior Eye. 2018 Jun;41(3):245-251. doi: 10.1016/j.clae.2017.12.017. Epub 2017 Dec 19.
5
ACANTHAMOEBA ENDOPHTHALMITIS AFTER RECURRENT KERATITIS AND NODULAR SCLERITIS.复发性角膜炎和结节性巩膜炎后棘阿米巴性眼内炎
Retin Cases Brief Rep. 2017;11(2):180-182. doi: 10.1097/ICB.0000000000000323.
6
Use of multiple immunosuppressive agents in recalcitrant ACANTHAMOEBA scleritis.多种免疫抑制剂在顽固性棘阿米巴性巩膜炎中的应用。
BMJ Case Rep. 2015 Apr 15;2015:bcr2014208536. doi: 10.1136/bcr-2014-208536.
7
Acanthamoeba sclerokeratitis: epidemiology, clinical features, and treatment outcomes.棘阿米巴性角膜炎:流行病学、临床特征和治疗结果。
Ophthalmology. 2014 Dec;121(12):2340-7. doi: 10.1016/j.ophtha.2014.06.033. Epub 2014 Aug 2.
8
Histopathological examination of Acanthamoeba sclerokeratitis.棘阿米巴性硬化性角膜炎的组织病理学检查。
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Scleral and intraocular amoebic dissemination in Acanthamoeba keratitis.棘阿米巴角膜炎中的巩膜和眼内阿米巴播散。
Cornea. 2013 Dec;32(12):1625-7. doi: 10.1097/ICO.0b013e31829ded51.
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