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角膜巩膜移植术后鼻孢子虫病继发的大型前巩膜葡萄肿部分消退——病例报告

Partial regression of large anterior scleral staphyloma secondary to rhinosporidiosis after corneoscleral graft - a case report.

作者信息

Costa Elaine Fiod, Pinto Luciano Moreira, Campos Marcos Adriano Garcia, Gomes Thais Mota, Silva Gyl Eanes Barros

机构信息

Departamento de Medicina I, Universidade Federal do Maranhão, Praça Gonçalves Dias, 21 - Centro, São Luis,, MA, 65020-240, Brazil.

Departamento de Oftalmologia, Universidade Federal de São Paulo, Rua Botucatu, 821 - Vila Clementino, São Paulo, SP, 04023-062, Brazil.

出版信息

BMC Ophthalmol. 2018 Feb 27;18(1):61. doi: 10.1186/s12886-018-0725-2.

DOI:10.1186/s12886-018-0725-2
PMID:29486755
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC5830333/
Abstract

BACKGROUND

Rhinosporidiosis is a rare chronic infection of the mucous membranes caused by the Rhinosporidium seeberi. Approximately 15% of cases of rhinosporidiosis are ocular, occurring mainly in the tarsal conjunctiva. There are only 11 cases of scleral melt with staphyloma formation associated with bulbar conjuctival oculosporidiosis and none of them was associated with partial regression of the scleral ectasia after a corneoscleral tectonic graft.

CASE PRESENTATION

a 13-year-old girl with a progressively increasing black mass in the upper nasal part above the cornea of the left eye. The biomicroscopy revealed an oval, bluish mass measuring 10x10x5 mm with congestion of the overlying conjunctiva. Conjunctival biopsy showed sporoblasts of Rinosporidium seeberi. Treatment was conducted by conjunctival resection and tectonic corneoscleral graft (13x13mm) over the staphyloma. Within 1 year of follow-up the patient presented a partial staphyloma reduction, 9x9x2.5 mm, and the patch detached from the lesion. A novel surgical approach was done reducing the corneal patch and no recurrence was seen after 9 months.

CONCLUSIONS

This case is one of the largest anterior scleral staphylomas secondary to rhinosporidiosis described in the literature. Scleral anterior staphyloma partial regression is an unusual outcome after a tectonic corneoscleral graft. Infection resolution and graft covering of thinned area contributed to scleral reepithelization.

摘要

背景

鼻孢子虫病是由西伯鼻孢子菌引起的一种罕见的黏膜慢性感染。约15%的鼻孢子虫病病例累及眼部,主要发生于睑结膜。仅有11例巩膜溶解伴葡萄肿形成与球结膜眼鼻孢子虫病相关,且无一例在角膜巩膜结构性移植术后巩膜扩张部分消退。

病例报告

一名13岁女孩,左眼角膜上方鼻根部有一逐渐增大的黑色肿物。生物显微镜检查发现一个椭圆形、蓝色肿物,大小为10×10×5mm,其上覆盖的结膜充血。结膜活检显示西伯鼻孢子菌的孢子母细胞。通过结膜切除术及在葡萄肿上方行结构性角膜巩膜移植术(13×13mm)进行治疗。随访1年内,患者葡萄肿部分缩小,变为9×9×2.5mm,补片与病变分离。采用一种新的手术方法缩小角膜补片,9个月后未见复发。

结论

该病例是文献报道中最大的鼻孢子虫病继发的前巩膜葡萄肿之一。角膜巩膜结构性移植术后巩膜前葡萄肿部分消退是一种不寻常的结果。感染消退及移植片覆盖变薄区域有助于巩膜重新上皮化。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/faaa/5830333/033dc6062fec/12886_2018_725_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/faaa/5830333/c8a0f02463b7/12886_2018_725_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/faaa/5830333/a7bb6a7c0433/12886_2018_725_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/faaa/5830333/033dc6062fec/12886_2018_725_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/faaa/5830333/c8a0f02463b7/12886_2018_725_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/faaa/5830333/a7bb6a7c0433/12886_2018_725_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/faaa/5830333/033dc6062fec/12886_2018_725_Fig3_HTML.jpg

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本文引用的文献

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2
Ocular rhinosporidiosis: a case report from Delhi.眼部鼻孢子虫病:来自德里的一例病例报告。
J Infect Dev Ctries. 2012 Nov 26;6(11):825-7. doi: 10.3855/jidc.2397.
3
Tectonic corneal graft for conjunctival rhinosporidiosis with scleral melt.板层角膜移植物治疗伴有巩膜溶解的结膜鼻孢子虫病。
J Curr Ophthalmol. 2022 Nov 30;34(3):341-346. doi: 10.4103/joco.joco_33_22. eCollection 2022 Jul-Sep.
4
Evaluation of diagnosed cases of eye rhinosporidiosis in a public hospital of Maranhão, Northeast Brazil.巴西东北部马拉尼昂州一家公立医院确诊的眼部鼻孢子虫病病例评估。
BMC Ophthalmol. 2019 Nov 8;19(1):218. doi: 10.1186/s12886-019-1223-x.
Indian J Ophthalmol. 2011 May-Jun;59(3):251-3. doi: 10.4103/0301-4738.81046.
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Oculosporidiosis in a tertiary care hospital of western Orissa, India: a case series.印度奥里萨邦西部一家三级护理医院的眼孢子虫病:病例系列
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