Shiao W Wong, Sasa Pockar, Laura R Steeples, Felipe Dhawahir-Scala, Salma Babiker, Nicholas P Jones
{"title":"toll样受体4突变的成人双侧顺序性单纯疱疹2型全眼炎","authors":"Shiao W Wong, Sasa Pockar, Laura R Steeples, Felipe Dhawahir-Scala, Salma Babiker, Nicholas P Jones","doi":"10.1080/09273948.2025.2524586","DOIUrl":null,"url":null,"abstract":"<p><strong>Purpose: </strong>To present the first reported case of bilateral sequential Herpes Simplex Virus Type 2 (HSV-2) panophthalmitis causing severe vision loss in a young adult with a toll-like receptor 4 (TLR-4) mutation, with a review of previously reported cases of HSV-2 panophthalmitis.</p><p><strong>Methods: </strong>Retrospective case report.</p><p><strong>Results: </strong>A 29-year-old man presented with a severely painful, swollen left eye and vision loss. Visual acuity was no light perception in both eyes. Six years earlier, he had developed right panophthalmitis, the cause confirmed as Herpes Simplex Virus Type 2 (HSV-2) by polymerase chain reaction (PCR) on aqueous humour (AH) sampling, with a poor outcome. At current presentation, PCR of AH and vitreous samples again confirmed HSV-2 infection. A poor initial response to high-dose intravenous antiviral led to the addition of high-dose systemic corticosteroid. Subsequent phacovitrectomy enabled partial visual recovery. Genetic testing revealed TLR-4 mutation rs4986790 A/G.</p><p><strong>Conclusion: </strong>HSV-2 panophthalmitis is rare and visually destructive, and we present the first reported case of bilateral disease. We hypothesise that TLR-4 mutation, not previously reported in association with HSV ocular inflammation, may have contributed to excessive inflammation and severe ocular damage. We suggest comprehensive immunological evaluation in all patients with severe viral intraocular inflammation. Prompt diagnosis with PCR and immediate high-dose antiviral therapy is critical and immune abnormality may require amended management.</p>","PeriodicalId":19406,"journal":{"name":"Ocular Immunology and Inflammation","volume":" ","pages":"1-4"},"PeriodicalIF":2.6000,"publicationDate":"2025-07-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Bilateral Sequential Herpes Simplex Type 2 Panophthalmitis in an Adult with a Toll-Like Receptor 4 Mutation.\",\"authors\":\"Shiao W Wong, Sasa Pockar, Laura R Steeples, Felipe Dhawahir-Scala, Salma Babiker, Nicholas P Jones\",\"doi\":\"10.1080/09273948.2025.2524586\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><strong>Purpose: </strong>To present the first reported case of bilateral sequential Herpes Simplex Virus Type 2 (HSV-2) panophthalmitis causing severe vision loss in a young adult with a toll-like receptor 4 (TLR-4) mutation, with a review of previously reported cases of HSV-2 panophthalmitis.</p><p><strong>Methods: </strong>Retrospective case report.</p><p><strong>Results: </strong>A 29-year-old man presented with a severely painful, swollen left eye and vision loss. Visual acuity was no light perception in both eyes. Six years earlier, he had developed right panophthalmitis, the cause confirmed as Herpes Simplex Virus Type 2 (HSV-2) by polymerase chain reaction (PCR) on aqueous humour (AH) sampling, with a poor outcome. At current presentation, PCR of AH and vitreous samples again confirmed HSV-2 infection. A poor initial response to high-dose intravenous antiviral led to the addition of high-dose systemic corticosteroid. Subsequent phacovitrectomy enabled partial visual recovery. Genetic testing revealed TLR-4 mutation rs4986790 A/G.</p><p><strong>Conclusion: </strong>HSV-2 panophthalmitis is rare and visually destructive, and we present the first reported case of bilateral disease. We hypothesise that TLR-4 mutation, not previously reported in association with HSV ocular inflammation, may have contributed to excessive inflammation and severe ocular damage. We suggest comprehensive immunological evaluation in all patients with severe viral intraocular inflammation. Prompt diagnosis with PCR and immediate high-dose antiviral therapy is critical and immune abnormality may require amended management.</p>\",\"PeriodicalId\":19406,\"journal\":{\"name\":\"Ocular Immunology and Inflammation\",\"volume\":\" \",\"pages\":\"1-4\"},\"PeriodicalIF\":2.6000,\"publicationDate\":\"2025-07-14\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Ocular Immunology and Inflammation\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.1080/09273948.2025.2524586\",\"RegionNum\":4,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q2\",\"JCRName\":\"OPHTHALMOLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Ocular Immunology and Inflammation","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1080/09273948.2025.2524586","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q2","JCRName":"OPHTHALMOLOGY","Score":null,"Total":0}
Bilateral Sequential Herpes Simplex Type 2 Panophthalmitis in an Adult with a Toll-Like Receptor 4 Mutation.
Purpose: To present the first reported case of bilateral sequential Herpes Simplex Virus Type 2 (HSV-2) panophthalmitis causing severe vision loss in a young adult with a toll-like receptor 4 (TLR-4) mutation, with a review of previously reported cases of HSV-2 panophthalmitis.
Methods: Retrospective case report.
Results: A 29-year-old man presented with a severely painful, swollen left eye and vision loss. Visual acuity was no light perception in both eyes. Six years earlier, he had developed right panophthalmitis, the cause confirmed as Herpes Simplex Virus Type 2 (HSV-2) by polymerase chain reaction (PCR) on aqueous humour (AH) sampling, with a poor outcome. At current presentation, PCR of AH and vitreous samples again confirmed HSV-2 infection. A poor initial response to high-dose intravenous antiviral led to the addition of high-dose systemic corticosteroid. Subsequent phacovitrectomy enabled partial visual recovery. Genetic testing revealed TLR-4 mutation rs4986790 A/G.
Conclusion: HSV-2 panophthalmitis is rare and visually destructive, and we present the first reported case of bilateral disease. We hypothesise that TLR-4 mutation, not previously reported in association with HSV ocular inflammation, may have contributed to excessive inflammation and severe ocular damage. We suggest comprehensive immunological evaluation in all patients with severe viral intraocular inflammation. Prompt diagnosis with PCR and immediate high-dose antiviral therapy is critical and immune abnormality may require amended management.
期刊介绍:
Ocular Immunology & Inflammation ranks 18 out of 59 in the Ophthalmology Category.Ocular Immunology and Inflammation is a peer-reviewed, scientific publication that welcomes the submission of original, previously unpublished manuscripts directed to ophthalmologists and vision scientists. Published bimonthly, the journal provides an international medium for basic and clinical research reports on the ocular inflammatory response and its control by the immune system. The journal publishes original research papers, case reports, reviews, letters to the editor, meeting abstracts, and invited editorials.