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대한안과학회지 2016 년제 57 권제 1 호 J Korean Ophthalmol Soc 2016;57(1):150-154 ISSN 0378-6471 (Print) ISSN 2092-9374 (Online) http://dx.doi.org/10.3341/jkos.2016.57.1.150 Case Report 안면부대상포진이후발생한다발성장액성맥락망막병증 1 예 Multiple Serous Chorioretinopathy after Facial Herpes Zoster 현상윤 김동윤 채주병 Sang Yoon Hyun, MD, Dong Yoon Kim, MD, PhD, Ju Byung Chae, MD, PhD 충북대학교의과대학안과학교실 Department of Ophthalmology, Chungbuk National University College of Medicine, Cheongju, Korea Purpose: To report a case of multiple serous chorioretinopathy after facial herpes zoster. Case summary: A 48-year-old male visited our clinic due to visual disturbance in the left eye which occurred 3 days after left facial pain and vesicles. Chemosis and multiple serous retinal detachments were found. The patient was diagnosed with multiple serous chorioretinopathy due to herpes zoster virus and was started on intravenous acyclovir at a dose of 10 mg/kg every 8 hours for 9 days and herpes eye ointment 5 times daily. After the initial treatment, oral prednisolone 60 mg was given daily for 6 days. Skin lesions were cleared, and abnormal fundus and visual acuity improved after treatment. Conclusions: Ophthalmopathy including multiple serous chorioretinopathy should be considered in managing herpes zoster ophthalmicus patients. J Korean Ophthalmol Soc 2016;57(1):150-154 Key Words: Chorioretinopathy, Herpes zoster ophthalmicus, Serous retinal detachment Varicella-zoster virus (VZV) 는인간헤르페스바이러스 3형이라고도부르며 1차감염시수두를일으키며재활성화될시대상포진을일으킨다. 1 뇌신경침범은전체대상포진의약 13-20% 정도를차지하며, 5번뇌신경이가장흔하게침범된다. 5번뇌신경은 3 개의분지로나뉘어져있는데안분지, 상악분지, 하악분지로나뉘며안분지가다른 2개의분지보다약 20배더잘감염되고, 안부대상포진환자의 50% 에서안과적인합병증이 Received: 2015. 4. 24. Revised: 2015. 6. 18. Accepted: 2015. 9. 25. Address reprint requests to Ju Byung Chae, MD, PhD Department of Ophthalmology, Chungbuk National University Hospital, #776 1sunhwan-ro, Seowon-gu, Cheongju 28644, Korea Tel: 82-43-269-6333, Fax: 82-43-269-5263 E-mail: cjbmed@naver.com * This work was supported by the research grant of Chungbuk National University in 2014. 발생한다. 2,3 한국인안부대상포진환자에서가장흔한임상양상은결막염인데, 점상각막염, 각막내피세포염, 각막침윤, 거짓가지모양각막염, 각막점액플라크, 외안근마비, 녹내장, 공막염순으로나타나는것으로알려져있으며후안부병변은드문것으로보고되었다. 4-6 후안부에발생하는병변으로는유리체혼탁, 망막출혈, 시신경병증, 급성망막괴사등이보고되어있다. 7-11 저자들은삼차신경의안분지, 상악분지영역에발생한안부대상포진환자에서속발적으로발생한, 다발성국소적, 장액성망막박리를동반한맥락망막병증환자 1예를경험하였고드문케이스로서이를보고하고자한다. 증례보고 48세남자환자가 3일전부터시작된급격한좌안시력저하를주소로본원안과에내원하였다. 환자는내원하기 6 일전좌측안면부위의통증을동반한눈주위수포성병변 c2016 The Korean Ophthalmological Society This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/3.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited. 150

- 현상윤외 : 안면부대상포진후발생한장액맥락망막병증 - A B Figure 1. Face photos showing facial herpes zoster. (A) Skin eruption corresponding to the dermatome of left ophthalmic branch of trigeminal nerve is observed. Nasal lesion shows Hutchinson's sign. (B) Oral mucosal erosion and pseudomembrane are observed at left upper soft plate. A B C D E F G Figure 2. Fundus photography, fluorescein angiography and optical coherence tomography showing multiple chorioretinopathy. (A) Multiple localized retinal detachments with subretinal fluid were found in macular and superior macular area. (B) Fluorescein angiography shows multiple hyperfluorescent leaking points with localized serous detachment (arrowheads) between optic disc and macular area. (C) After treatment (two months later), fluorescein angiography shows no hyperfluorescence. (D) Multiple subretinal fluid and increased choroidal thickness were found. (E) After treatment, superior subretinal fluid moves downward to fovea. (F) After 1 month, subretinal fluid decreased. (G) After 2 months, subretinal fluid disappeared and there was no retinochoroidal inflammation. 과입천장의백태가생겼다고하였다 (Fig. 1). 과거력상 10 년전부터당뇨병이있었으며, 인슐린주사를통해혈당이 양호하게조절되었고이외특별한이상소견은없었다. 피부병변은좌측삼차신경의안분지, 상악분지가지배하는영역 151

- 대한안과학회지 2016 년제 57 권제 1 호 - 을따라발생하였다. 초진시우안교정시력 0.9 좌안교정시력 0.07이었으며안압은우안 17 mmhg, 좌안 11 mmhg 였다. 세극등검사상좌안결막부종과각막에점상각막미란이관찰되었으나전방에염증을나타내는세포소견은없었다. 안저소견상황반부및황반상측에다량의망막하액이관찰되었고 (Fig. 2A) 형광안저촬영에서다발성, 국소적장액성망막박리부위에플루레신고임에의한과형광소견과, 시신경과황반부사이에맥락막이상부위로예상되는점상의다발성과형광소견이관찰되었다 (Fig. 2B). 빛간섭단층촬영검사상혼탁한망막하액을동반한다발성장액성망막박리소견을보였고 Enhanced depth imaging 모드검사에서도맥락막두께가 451 μm로두꺼워져있는모습이었다 (Fig. 2D). 대상포진바이러스에의한다발성장액성맥락막망막병증진단하에 acyclovir (Zovirax, Glaxosmithkline, UK) 10 mg/kg을 1일 3회씩, 9일간정맥투여했으며동시에경구용 Prednisolone 20 mg을하루 3번 6일간투여한후 3 주에걸쳐감량하였다. 이외에점안제로 acyclovir 안연고 (Herpesid eye oint, Samil, Seoul, Korea) 를하루 5회점안하게하였다. 치료후일주일에, 상측에존재하던망막하액이황반부로이동하였고그이후망막하액은점점감소되었으며, 피부증상은호전되었다 (Fig. 2E). 1달뒤빛간섭단층촬영에서황반부에소량의망막하액만남아있었으며 (Fig. 2F) 2달뒤시행한빛간섭단층촬영에서망막하액은모두소실되었고형광안저촬영상누출소견없이정상소견이관찰되었다 (Fig. 2C, G). 2달째좌안교정시력 0.4로회복되었다. 고찰 안부대상포진은제5뇌신경인삼차신경에헤르페스바이러스가침범하면서발생하고, 이신경의 3개분지중제1 분지인안분지가다른 2개의분지보다약 20배나더잘감염된다. 2 제1 분지가운데하나인코섬모체신경은각막, 공막홍채, 맥락막등안구와코끝피부, 안검피부에분포한다. Hutchinson 증상은코끝피부에병변이나타나는것을말하며 Zaal et al 12 은 Hutchinson 증상이나타날때코섬모체신경의침범으로안구염증가능성이높아진다고보고하였다. 해부학적으로코섬모체신경은장섬모체신경과단섬모체신경으로나뉘며이신경이공막을지나맥락막계에분포하게된다 (Fig. 3). 12-14 본증례에서환자는코끝피부에병변이있는 Hutchinson 증상양성이었다. 이러한맥락막및후극부의염증소견은코섬모체신경쪽의장섬모체신경과단섬모체신경을통한대상포진의활성화가원인이될것으로생각된다. 섬모체신경이공막을지나맥락막계에 Figure 3. Anatomy of Ophthalmic branch in trigeminal nerve. Long posterior ciliary nerve and short posterior ciliary nerve cover posterior scleral area in the orbit. 분포한다는것을생각할때전안부를통한염증의파급과관련없이후극부의공막및맥락막에분포하는섬모체신경이본증례의맥락망막염증을유발했을것으로생각하였다. 본증례에서한가지특이한점은, 제5뇌신경 ( 삼차신경 ) 의상악분지가관여하는연구개부위의심한염증이다. 해부학적으로상악분지가안구뒤쪽을관여하지는않지만, 특이하게도맥락망막염증과연구개부위의심한염증이동반되었다. 일반적인장액맥락망막병증의경우국소적이면서, 명확한누출점이관찰되는경우가많으나, 본증례는바이러스감염이후속발된염증으로인한광범위한맥락망막병증으로인해, 명확한누출점의형태보다는다발성의국소적, 미만성누출의형태로발현되었다. 초기형광안저촬영상 (Fig. 2B) 다발성누출점들이망막하액부위에여러개의병변으로관찰되었으며, 이는전형적인중심장액맥락망막병증에서보이는명확한국소적누출점보다는약하지만, 다발성의미만성누출점의형태로서관찰된다고생각하였다. 누출의양상이다른이유는바이러스감염에의한이차적맥락망막병증이넓은부위의맥락막에분포해서국소적이기보다는다발성, 미만성의누출점을만들었을것이라고생각하였다. 만일인도시아닌그린안저촬영을실시하였다면맥락막의병변을더잘관찰할수있었을것으로생각되나인도시아닌그린안저촬영은시행하지못하였다. 우리는본증례에서보이는다발성맥락막염을동반한장액성망막박리의양상이보그트-고야나기-하라다증후군의임상양상과유사한양상을보인다고생각하였다. 보크트-고야나기-하라다증후군은여러개독립된장액성망막 152

- 현상윤외 : 안면부대상포진후발생한장액맥락망막병증 - 박리형태로나타났다가시간이경과하면서합쳐지고빛간섭단층촬영검사에서망막박리부위의망막하액소견과망막내부에염증성섬유소들이있는낭포성구조물이관찰되는데이는본증례의안저소견및빛간섭단층촬영소견과유사하다. 15 보크트-고야나기-하라다병의발병원인은정확히알려져있지않다. 가능한원인가설중하나가망막색소세포에대한자가면역질환이다. 망막색소세포에대하여 T 림프구발현이염증을일으킬수있는데이에대한원인가설은유전가설, 감염가설, 면역가설이있다. 그중감염가설에따르면바이러스감염에의한항원-항체반응으로활성화된 T 림프구가면역반응을일으키는것으로알려져있다. 16 보크트-고야나기-하라다병의치료는 T-림프구의면역반응을억제하기위하여고용량스테로이드치료를시행하고있으며, 본증례에서도후안부병변이바이러스의직접적인독성뿐만아니라항원항체면역반응을매개로해서일어날수도있다는점을고려하여 acyclovir와함께경구용스테로이드를사용하였고증상호전을경험하였다. 바이러스에의해발생하는맥락막염증및장액성망막박리를이해하는데있어서본증례는그임상양상을이해하는데도움이된다고생각되며, 보그트-고야나기-하라다증후군의병인론에대한가설들중감염가설을설명하는데에도도움을줄수있다고생각한다. 본증례는바이러스로인한맥락막과관련된후극부염증을이해하는데도움이될것으로생각된다. 결론적으로, 본증례는대상포진바이러스가코섬모체신경을통해맥락막계에영향을미쳐다발성의맥락막염증을일으켰을것으로생각할수있다. 이러한경우전안부의이상소견없이도, 맥락막염증과이차적인장액망막박리를유발할수있을것으로생각된다. 현재까지안부대상포진환자의후안부병변중시신경염, 유리체혼탁, 급성망막괴사등의질병이보고되었으나, 본증례에서저자들은전방및유리체의염증소견없이, 망막의괴사를동반하지않는맥락망막병증환자를경험하였다. 안부대상포진환자에서시력저하가발생할때, 맥락막염을동반한장액망막박리가발생할수있음을염두에두어야할것으로생각한다. REFERENCES 1) Lee HK. Virus keratitis. In: Korea External Eye Disease Society, ed. Cornea, 3rd ed. Seoul: Ilchokak, 2013; chap. 8. 2) Liesegang TJ. Herpes zoster ophthalmicus natural history, risk factors, clinical presentation, and morbidity. Ophthalmology 2008; 115(2 Suppl):S3-12. 3) Lee HJ, Kim SY, Jung MS. The clinical characteristics of facial herpes zoster in Korean patients. J Korean Ophthalmol Soc 2010;51:8-13. 4) Chung YR, Chang YH, Kim DH, Yang HS. Ocular manifestations of herpes zoster ophthalmicus. J Korean Ophthalmol Soc 2010;51: 164-68. 5) Lee WB, Liesegang TJ. Herpes zoster keratitis. In: Krachmer JH, Mannis MJ, Holland EJ, eds. Cornea, 3rd ed. Philadelphia: Elsevier Mosby, 2011; v. 1. chap. 40. 6) Park SH, Kim WJ, Yang SW, Kim MS. Herpes zoster ophthalmicus complicated by hyphema, glaucoma and external ophthalmoplegia. J Korean Ophthalmol Soc 2007;48:1573-8. 7) Kim M, Choi MY, Chae JB. Complicated ophthalmopathy in herpes zoster ophthalmicus including vitreous opacity, retinal hemorrhage and optic neuropathy. J Korean Ophthalmol Soc 2013;54: 513-7. 8) Culbertson WW, Blumenkranz MS, Pepose JS, et al. Varicella zoster virus is a cause of the acute retinal necrosis syndrome. Ophthalmology 1986;93:559-69. 9) Zamora RL, del Priore LV, Storch GA, et al. Multiple recurrent branch retinal artery occlusions associated with varicella zoster virus. Retina 1996;16:399-404. 10) Gilden DH, Kleinschmidt-DeMasters BK, Wellish M, et al. Varicella zoster virus, a cause of waxing and waning vasculitis: the New England Journal of Medicine case 5-1995 revisited. Neurology 1996;47:1441-6. 11) Kim JY, Ahn M, Lee DW. Two cases of optic neuritis in herpes zoster ophthalmicus. J Korean Ophthalmol Soc 2008;49:1028-32. 12) Zaal MJ, Völker-Dieben HJ, DAmaro J. Prognostic value of Hutchinson s sign in acute herpes zoster ophthalmicus. Graefes Arch Clin Exp Ophthalmol 2003;241:187-91. 13) Monica Evans. Anatomy of the Uvea. In: Yanoff M, Ducker JS, eds. Ophthalmology, 4th ed. Philadelphia: Elsevier Mosby, 2014; chap. 7.1. 14) Dutton JJ. Atlas of Clinical and Surgical Orbital Anatomy, 2nd ed. London: Elsevier, 2011; 51-82. 15) Moorthy RS, Inomata H, Rao NA. Vogt-Koyanagi-Harada syndrome. Surv Ophthalmol 1995;39:265-92. 16) Greco A, Fusconi M, Gallo A, et al. Vogt-Koyanagi-Harada syndrome. Autoimmun Rev 2013;12:1033-8. 153

- 대한안과학회지 2016 년제 57 권제 1 호 - = 국문초록 = 안면부대상포진이후발생한다발성장액성맥락망막병증 1 예 목적 : 안면부대상포진이후발생한다발성장액성맥락망막병증환자 1 예를보고하고자한다. 증례요약 : 48 세남자가 3 일전시작된좌안주위통증과안검부종, 안검수포성병변발생후동반된시력저하를주소로내원하였다. 세극등검사상좌안전방및유리체염증소견없이, 맥락막염증을동반한다발성장액성망막박리소견이관찰되었다. 대상포진바이러스에의한다발성장액성맥락망막병증진단하에 9 일간의정맥내 acyclovir 주사및 6 일간의경구스테로이드를투여하였다. 대상포진에의한전안부감염을예방하기위해, acyclovir 안연고를하루 5 회사용하였다. 약물치료후피부병변과장액성망막박리및맥락망막병증은호전되었으며시력도호전되었다. 결론 : 안부대상포진환자에서갑작스런시력저하가동반될때전안부염증없이도후극부에다발성장액성맥락망막병증이동반될수있다. < 대한안과학회지 2016;57(1):150-154> 154