无低温血浆输注治疗木质结膜炎。

IF 0.6 Q4 OPHTHALMOLOGY
Case Reports in Ophthalmology Pub Date : 2025-07-07 eCollection Date: 2025-01-01 DOI:10.1159/000546953
Anna Bonde Scheel-Bech, Marie Louise Roed Rasmussen, Steffen Heegaard, Ida Maria Schmidt, Jakob Stensballe
{"title":"无低温血浆输注治疗木质结膜炎。","authors":"Anna Bonde Scheel-Bech, Marie Louise Roed Rasmussen, Steffen Heegaard, Ida Maria Schmidt, Jakob Stensballe","doi":"10.1159/000546953","DOIUrl":null,"url":null,"abstract":"<p><strong>Introduction: </strong>This case report presents the clinical, histological, and genetic characteristics of a patient with ligneous conjunctivitis and to provide a long-term treatment schedule, using cryo-depleted plasma infusions in combination with topical cyclosporine as a successful therapeutic option.</p><p><strong>Case presentation: </strong>A 12-year-old Turkish girl experienced years of ocular itching, irritation, and increased light sensitivity. Initial clinical examination revealed the presence of pseudomembranes and giant papillae on the tarsal conjunctivae of both eyes. The biopsy of a membrane from the left eye was taken for histological examination. Manifestations of severe comorbidity since infancy (congenital hydrocephalus, asthma, recurrent respiratory infections, and impaired wound healing) and parental consanguinity prompted a genetic evaluation. The pseudomembranes were composed of fibrin, granulation tissue, and inflammatory cells consistent with the diagnosis of ligneous conjunctivitis. The genetic analysis revealed homozygosity for a pathogenic missense variant in plasminogen (PLG), causing PLG deficiency. Due to severe systemic complications, the patient was successfully treated with cryo-depleted plasma infusions and cyclosporine eye drops, resulting in the complete remission of the ligneous conjunctivitis.</p><p><strong>Conclusion: </strong>This case report highlights the importance of considering PLG deficiency in patients with recurring chronic conjunctivitis. Long-term treatment with cryo-depleted plasma infusions in combination with short-term use of topical cyclosporine may provide an efficient therapeutic option for ligneous conjunctivitis in patients with PLG deficiency.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"542-550"},"PeriodicalIF":0.6000,"publicationDate":"2025-07-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12331225/pdf/","citationCount":"0","resultStr":"{\"title\":\"Cryo-Depleted Plasma Infusions for Ligneous Conjunctivitis.\",\"authors\":\"Anna Bonde Scheel-Bech, Marie Louise Roed Rasmussen, Steffen Heegaard, Ida Maria Schmidt, Jakob Stensballe\",\"doi\":\"10.1159/000546953\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><strong>Introduction: </strong>This case report presents the clinical, histological, and genetic characteristics of a patient with ligneous conjunctivitis and to provide a long-term treatment schedule, using cryo-depleted plasma infusions in combination with topical cyclosporine as a successful therapeutic option.</p><p><strong>Case presentation: </strong>A 12-year-old Turkish girl experienced years of ocular itching, irritation, and increased light sensitivity. Initial clinical examination revealed the presence of pseudomembranes and giant papillae on the tarsal conjunctivae of both eyes. The biopsy of a membrane from the left eye was taken for histological examination. Manifestations of severe comorbidity since infancy (congenital hydrocephalus, asthma, recurrent respiratory infections, and impaired wound healing) and parental consanguinity prompted a genetic evaluation. The pseudomembranes were composed of fibrin, granulation tissue, and inflammatory cells consistent with the diagnosis of ligneous conjunctivitis. The genetic analysis revealed homozygosity for a pathogenic missense variant in plasminogen (PLG), causing PLG deficiency. Due to severe systemic complications, the patient was successfully treated with cryo-depleted plasma infusions and cyclosporine eye drops, resulting in the complete remission of the ligneous conjunctivitis.</p><p><strong>Conclusion: </strong>This case report highlights the importance of considering PLG deficiency in patients with recurring chronic conjunctivitis. Long-term treatment with cryo-depleted plasma infusions in combination with short-term use of topical cyclosporine may provide an efficient therapeutic option for ligneous conjunctivitis in patients with PLG deficiency.</p>\",\"PeriodicalId\":9635,\"journal\":{\"name\":\"Case Reports in Ophthalmology\",\"volume\":\"16 1\",\"pages\":\"542-550\"},\"PeriodicalIF\":0.6000,\"publicationDate\":\"2025-07-07\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12331225/pdf/\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Case Reports in Ophthalmology\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.1159/000546953\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"2025/1/1 0:00:00\",\"PubModel\":\"eCollection\",\"JCR\":\"Q4\",\"JCRName\":\"OPHTHALMOLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Case Reports in Ophthalmology","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.1159/000546953","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2025/1/1 0:00:00","PubModel":"eCollection","JCR":"Q4","JCRName":"OPHTHALMOLOGY","Score":null,"Total":0}
引用次数: 0

摘要

本病例报告介绍了木质结膜炎患者的临床、组织学和遗传特征,并提供了一个长期的治疗方案,使用低温耗尽血浆输注联合局部环孢素作为一种成功的治疗选择。病例介绍:一名12岁的土耳其女孩经历了多年的眼部瘙痒,刺激和增加的光敏感性。初步临床检查发现双眼底结膜有假膜及巨大乳头状。取左眼膜活检作组织学检查。婴儿时期的严重合并症表现(先天性脑积水、哮喘、反复呼吸道感染和伤口愈合受损)和父母的血缘关系促使进行遗传评估。假膜由纤维蛋白、肉芽组织和炎性细胞组成,符合木质结膜炎的诊断。遗传分析显示纤溶酶原(PLG)的致病性错义变异纯合性,导致PLG缺陷。由于出现严重的全身并发症,患者经低温血浆输注和环孢素滴眼液治疗成功,木质结膜炎完全缓解。结论:本病例报告强调了在复发性慢性结膜炎患者中考虑PLG缺乏的重要性。长期低温血浆输注联合短期局部使用环孢素可能为PLG缺乏患者木质结膜炎提供有效的治疗选择。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

Cryo-Depleted Plasma Infusions for Ligneous Conjunctivitis.

Cryo-Depleted Plasma Infusions for Ligneous Conjunctivitis.

Cryo-Depleted Plasma Infusions for Ligneous Conjunctivitis.

Introduction: This case report presents the clinical, histological, and genetic characteristics of a patient with ligneous conjunctivitis and to provide a long-term treatment schedule, using cryo-depleted plasma infusions in combination with topical cyclosporine as a successful therapeutic option.

Case presentation: A 12-year-old Turkish girl experienced years of ocular itching, irritation, and increased light sensitivity. Initial clinical examination revealed the presence of pseudomembranes and giant papillae on the tarsal conjunctivae of both eyes. The biopsy of a membrane from the left eye was taken for histological examination. Manifestations of severe comorbidity since infancy (congenital hydrocephalus, asthma, recurrent respiratory infections, and impaired wound healing) and parental consanguinity prompted a genetic evaluation. The pseudomembranes were composed of fibrin, granulation tissue, and inflammatory cells consistent with the diagnosis of ligneous conjunctivitis. The genetic analysis revealed homozygosity for a pathogenic missense variant in plasminogen (PLG), causing PLG deficiency. Due to severe systemic complications, the patient was successfully treated with cryo-depleted plasma infusions and cyclosporine eye drops, resulting in the complete remission of the ligneous conjunctivitis.

Conclusion: This case report highlights the importance of considering PLG deficiency in patients with recurring chronic conjunctivitis. Long-term treatment with cryo-depleted plasma infusions in combination with short-term use of topical cyclosporine may provide an efficient therapeutic option for ligneous conjunctivitis in patients with PLG deficiency.

求助全文
通过发布文献求助,成功后即可免费获取论文全文。 去求助
来源期刊
CiteScore
0.90
自引率
0.00%
发文量
129
审稿时长
12 weeks
期刊介绍: This peer-reviewed online-only journal publishes original case reports covering the entire spectrum of ophthalmology, including prevention, diagnosis, treatment, toxicities of therapy, supportive care, quality-of-life, and survivorship issues. The submission of negative results is strongly encouraged. The journal will also accept case reports dealing with the use of novel technologies, both in the arena of diagnosis and treatment. Supplementary material is welcomed. The intent of the journal is to provide clinicians and researchers with a tool to disseminate their personal experiences to a wider public as well as to review interesting cases encountered by colleagues all over the world. Universally used terms can be searched across the entire growing collection of case reports, further facilitating the retrieval of specific information. Following the open access principle, the entire contents can be retrieved at no charge, guaranteeing easy access to this valuable source of anecdotal information at all times.
×
引用
GB/T 7714-2015
复制
MLA
复制
APA
复制
导出至
BibTeX EndNote RefMan NoteFirst NoteExpress
×
提示
您的信息不完整,为了账户安全,请先补充。
现在去补充
×
提示
您因"违规操作"
具体请查看互助需知
我知道了
×
提示
确定
请完成安全验证×
copy
已复制链接
快去分享给好友吧!
我知道了
右上角分享
点击右上角分享
0
联系我们:info@booksci.cn Book学术提供免费学术资源搜索服务,方便国内外学者检索中英文文献。致力于提供最便捷和优质的服务体验。 Copyright © 2023 布克学术 All rights reserved.
京ICP备2023020795号-1
ghs 京公网安备 11010802042870号
Book学术文献互助
Book学术文献互助群
群 号:604180095
Book学术官方微信