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泡性角结膜炎——一例采用全身性生物免疫抑制疗法治疗的非典型重症病例。

Phlyctenular keratoconjunctivitis - an atypically severe case treated with systemic biologic immunosuppressive therapy.

作者信息

Valério Sequeira Valadares Joana, Bastos-Carvalho Ana, Pedroso Franco José Manuel, Mourão Ana Filipa, Monteiro-Grillo Manuel

机构信息

Centro Hospitalar Lisboa Norte, Hospital Santa Maria, Lisbon, Portugal.

Centro Hospitalar Lisboa Norte, Hospital Santa Maria, Lisbon, Portugal; Centro Hospitalar Lisboa Ocidental, Egas Moniz Hospital, Lisbon, Portugal.

出版信息

GMS Ophthalmol Cases. 2014 Jan 10;4:Doc02. doi: 10.3205/oc000015. eCollection 2014.

DOI:10.3205/oc000015
PMID:27625937
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC5015615/
Abstract

PURPOSE

To report an atypically severe and refractory phlyctenular keratoconjunctivitis case treated successfully with systemic biologic immunosuppressive therapy.

METHODS

A 10-year-old female was followed in the ophthalmology clinic for three years for a severe form of bilateral PKC. The patient was treated for blepharitis and intestinal parasitosis, and underwent topical corticosteroid therapy, followed by subconjunctival injections and systemic corticosteroids with no clinical improvement. An association of topical cyclosporine A and oral methotrexate had no clinical response either. Phlyctenae of the cornea remained evident with neovascularization, progressive peripheral corneal thinning and occasional anterior chamber reaction.

RESULTS

The patient was treated with a combination of infliximab and methotrexate and corticosteroid therapy was tapered, with a fast and sustained resolution of the symptoms and corneal signs. Eleven months past initiation of the treatment, the patient remains asymptomatic and without any recurrence of the disease.

CONCLUSION

Phlyctenular keratoconjunctivitis may present with a broad spectrum of symptoms and signs, and its severity varies significantly. In cases of severe PKC, which are refractory to conventional therapy, systemic biologic immunosuppressive therapy may be a valuable alternative.

摘要

目的

报告一例采用全身生物免疫抑制疗法成功治疗的非典型严重难治性泡性角结膜炎病例。

方法

一名10岁女性因严重双侧泡性角结膜炎在眼科门诊随访三年。该患者接受了睑缘炎和肠道寄生虫病治疗,接受了局部皮质类固醇治疗,随后进行结膜下注射和全身皮质类固醇治疗,但临床症状无改善。局部应用环孢素A联合口服甲氨蝶呤也无临床反应。角膜上的泡疹仍然明显,伴有新生血管形成、周边角膜进行性变薄以及偶尔的前房反应。

结果

患者接受英夫利昔单抗联合甲氨蝶呤治疗,同时逐渐减少皮质类固醇治疗,症状和角膜体征迅速且持续缓解。治疗开始11个月后,患者无症状,疾病无任何复发。

结论

泡性角结膜炎可能表现出广泛的症状和体征,其严重程度差异显著。对于传统治疗难治的严重泡性角结膜炎病例,全身生物免疫抑制疗法可能是一种有价值的替代方法。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2ab0/5015615/94ee8df20e82/OC-04-02-g-002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2ab0/5015615/bad77dfb26ae/OC-04-02-g-001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2ab0/5015615/94ee8df20e82/OC-04-02-g-002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2ab0/5015615/bad77dfb26ae/OC-04-02-g-001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2ab0/5015615/94ee8df20e82/OC-04-02-g-002.jpg

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本文引用的文献

1
Tacrolimus ointment 0.03% for treatment of refractory childhood phlyctenular keratoconjunctivitis.他克莫司软膏 0.03% 治疗难治性儿童滤泡性角结膜炎。
Cornea. 2012 Aug;31(8):950-2. doi: 10.1097/ICO.0b013e318243f69d.
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Resolution of childhood recurrent corneal phlyctenulosis following eradication of an intestinal parasite.肠道寄生虫根除后儿童复发性角膜小泡病的消退
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英夫利昔单抗治疗儿童慢性非感染性葡萄膜炎:长期随访的回顾性病例系列研究
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Topical cyclosporine A in severe steroid-dependent childhood phlyctenular keratoconjunctivitis.局部应用环孢素A治疗重度依赖类固醇的儿童泡性角结膜炎。
Am J Ophthalmol. 2006 Jan;141(1):62-66. doi: 10.1016/j.ajo.2005.08.035.
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Phlyctenular keratitis associated with meibomitis in young patients.年轻患者中与睑板腺炎相关的泡性角膜炎
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Phlyctenular eye disease: a reappraisal.泡性眼病:重新评估
Jpn J Ophthalmol. 2000 Mar-Apr;44(2):146-50. doi: 10.1016/s0021-5155(99)00185-9.