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1 대한안과학회지 2018 년제 59 권제 10 호 J Korean Ophthalmol Soc 2018;59(10): ISSN (Print) ISSN (Online) Case Report 급성폐쇄각녹내장을동반한비전형적인보그트 - 고야나기 - 하라다병 1 예 A Case of Atypical Vogt-Koyanagi-Harada Disease Preceded by Acute Angle Closure Glaucoma 최민규 전연숙 김지택 Min Gyu Choi, MD, Yeoun Sook Chun, MD, PhD, Jee Taek Kim, MD, PhD 중앙대학교의과대학중앙대학교병원안과학교실 Department of Ophthalmology, Chung-Ang University Hospital, Chung-Ang University College of Medicine, Seoul, Korea Purpose: To report a case of atypical Vogt Koyanagi Harada disease that occurred after an acute angle closure glaucoma attack. Case summary: A 48-year-old female presented with bilateral visual disturbance accompanied by headache and ocular pain. The patient had no specific past medical or family history except taking oral contraceptives for 10 years. Despite the normalization of intraocular pressure in a local clinic, a shallow-depth anterior chamber and forward displacement of the iris lens diaphragm remained unresolved. The depth of the anterior chamber had increased in both eyes after laser therapy but without recovery of her visual acuity. B-scans showed ciliochoroidal effusion. Anterior chamber inflammation was observed in both eyes. Optical coherence tomography showed lobulated and serous retinal detachment involving the macula of both eyes. However, fluorescence angiography findings showed no multiple hyperfluorescence, which is unusual for typical cases of Vogt Koyanagi Harada disease. The patient was diagnosed with atypical Vogt Koyanagi Harada disease and was treated with eyedrops and intravenous steroid pulse therapy, after which she was converted to oral medications with immunosuppressants. After 1 month, no serous retinal detachment was detected. After 3 months, best corrected visual acuity (logmar) was 0.0 in both eyes, and there has been no recurrence on follow-up. Conclusions: Atypical Vogt Koyanagi Harada disease at presentation can mimic acute attacks of angle closure glaucoma. Therefore, if there is no improvement after treatment for angle closure glaucoma including laser iridotomy, other diseases including Vogt Koyanagi Harada disease must be considered and the patient should be closely monitored. J Korean Ophthalmol Soc 2018;59(10): Keywords: Angle closure glaucoma, Atypical, Laser iridotomy, Vogt Koyanagi Harada disease Received: Revised: Accepted: Address reprint requests to Jee Taek Kim, MD, PhD Department of Ophthalmology, Chung-Ang University Hospital, #102 Heukseok-ro, Dongjak-gu, Seoul 06973, Korea Tel: , Fax: jeetaek-kim@hanmail.net * Conflicts of Interest: The authors have no conflicts to disclose. 보그트-고야나기-하라다 (Vogt Koyanaki Harada) 병은초기에는주로후극부를침범하여전안부로진행하는양측성의육아종성전체포도막염이다. 1 발병기전이뚜렷하게밝혀지지는않았으나멜라닌세포를침범하는자가면역반응에의한것으로알려져있고, 아시아인을비롯한유색인종에서보다흔히보고되고있으며피부백반, 백모증, 청력장애, 뇌막자극증상등이동반된다. 2 보그트-고야나기-하라다병에서관찰되는녹내장은치료 c2018 The Korean Ophthalmological Society This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited. 978

- 최민규외 : 급성폐쇄각녹내장을동반한하라다병 - 를위해사용한스테로이드에의한만성개방각녹내장과질병자체에의한포도막염으로발생한주변홍채유착또는홍채후유착에의한폐쇄각녹내장의형태로주로발생한다. 드물게는보그트-고야나기-하라다병의재발시급성폐쇄각녹내장의형태로발생한예가보고된바있다.

2 - 최민규외 : 급성폐쇄각녹내장을동반한하라다병 - 를위해사용한스테로이드에의한만성개방각녹내장과질병자체에의한포도막염으로발생한주변홍채유착또는홍채후유착에의한폐쇄각녹내장의형태로주로발생한다. 드물게는보그트-고야나기-하라다병의재발시급성폐쇄각녹내장의형태로발생한예가보고된바있다. 3 보그트- 고야나기-하라다병의증례는드물지않게볼수있는질환이지만, 급성기에전방각폐쇄녹내장의양상으로발생하는증례는매우드물고국내에서는아직보고된바가없기에본증례를보고하고자한다. 증례보고 48세여자가 4일전부터시작된두통및안통을동반한양안시력저하를주소로내원하였다. 과거력상 10여년간경구용피임약 desogestrel 0.15 mg/ethinyl estradiol 0.02 mg (Mercilon, Organon, Oss, Netherlands) 복용이외에다른질환은없이건강하였다. 보그트-고야나기-하라다병과동반되어나타날수있는다른증상 ( 이명, 청력감소, 경부강직및피부과적증상 ) 은없었다. 1일전타병원에서안압이우안 34 mmhg, 좌안 34 mmhg로측정되었고양안전방의깊이가얕아져있어급성폐쇄각녹내장진단하에 acetazolamide 250 mg (Acetazol, Hanlim, Seoul, Korea) 을하루 2회경구투여, 2% dorzolamide hydrocholide/0.5% timolol maleate 고정복합체 (Cosopt, MSD, Blue Bell, PA, USA) 양안하루 2회점안, 2% pilocarpine hydrochloride (Isopto Carpine, Alcon, Fort Worth, TX, USA) 양안하루 4회점안하였다. 1일경과후동일한타병원에서안압이우안 23 mmhg, 좌안 23 mmhg로감소하였으나, 최대교정시력 (logmar) 은우안 0.5, 좌안 0.5 및양안얕은전방의깊이가지속되어같은날본원으로진료의뢰되었다. 내원당시최대교정시력 (logmar) 은우안 0.4, 좌안 0.5 였고안압은우안 18 mmhg, 좌안 19 mmhg였다. 세극등현미경검사에서 2% pilocarpine hydrochloride의사용으로인한양안의심한축동을확인할수있었고, 양안모두홍채-수정체가로막이앞으로이동되어있었으며중심부전방깊이는각막두께의 1/4배였다. 골드만전방각경검사에서전방향모두 Spaeth 분류기준으로 A10s의소견을보였으며전안부빛간섭단층촬영에서얕아진전방깊이와함께골드만전방각경으로확인하지못한열린전방각저를확인할수있었다 (Fig. 1A, B). 양안의심한축동상태로인하여안저검사에제한이있었다. 2% dorzolamide hydrocholide/ 0.5% timolol maleate 고정복합체양안하루 2회점안을유지하고, 악성녹내장의가능성을배제할수없어 2% pilocarpine hydrochloride는중단하였다. 양안레이저홍채절개술, 아르곤레이저주변홍채성형술을시행하였고 2% homatropine hydrobromide (Homapine, Hanlim, Seoul, Korea) 양안하루 2회점안하였다. 레이저시행및조절마비제의점안후전안부빛간섭단층촬영에서시행전보다중심부전방깊이가각막두께의 2배로넓어졌으나호전정도가일반적인급성폐쇄각녹내장의치료에서관찰되는것보다작았다 (Fig. 1C, D). 함께시행한안구초음파검사에서맥락막삼출소견을, 망막빛간섭단층촬영에서구획화된망막하액을확인할수있었다 (Fig. 2). 그러나레이저시행 1주경과후추적관찰하기로하였던환자는개인사정으로 2주경과후에내원하였음에도불구하고전방의깊이는여전히얕은상태였고최대교정시력 (logmar) 또한우안 0.4, 좌안 0.5로변화가없었으며망막하액의증가소견또한확인되어망막분과로의뢰되었다. 당시양안전안부염증은 1+ 였고안저검사와빛간섭단층촬영에서시신경유두부종과황반부를침범하는악화된장액 A B C D Figure 1. Optical coherence tomography images of anterior segment in both eyes. Optical coherence tomography images of anterior segment show narrow anterior chamber angle with forward displacement of iris-lens diaphragm in the right eye (A) and the left eye (B). After laser iridotomy and argon laser peripheral iridoplasty, anterior chamber angle deepened in the right eye (C) and the left eye (D). But it was not enough to cause improvement. 979

3 - 대한안과학회지 2018 년제 59 권제 10 호 - A B C D Figure 2. B-scan and optical coherence tomography images in both eyes. B-scan images demonstrate ciliochoroidal effusion (white asterisk) in the right eye (A) and the left eye (B). Optical coherence tomography shows multiple serous retinal detachment in both eyes (C, D) on day 6 after onset. OD = oculus dexter; OS = oculus sinister. 망막박리가관찰되었으나, 형광안저혈관조영술에서는이와일치하는다발성의국소형광누출소견은보이지않았다 (Fig. 3). 초진시시행한망막빛간섭단층촬영의구획화된망막하액, 안구초음파검사에서의맥락막삼출소견, 2주경과후확인된양안의전안부염증소견은일반적인보그트-고야나기-하라다병의임상양상과유사했지만형광안저혈관조영술에서의전형적인다발성국소형광누출소견을확인할수없었고레이저홍채절개술을비롯한폐쇄각녹내장의치료시행후에도회복되지않는전방깊이등을토대로초기에급성폐쇄각녹내장으로발현된비전형적인보그트-고야나기-하라다병으로진단하고부신피질호르몬인 methylprednisolone (Salon, Hanlim, Seoul, Korea) 을하루 1 g씩 3일간정맥내투여하였다. 고용량부신피질호르몬요법후양안최대교정시력 (logmar) 은 0.2로호전되었고전방의깊이또한회복되었으며장액망막박리도호전되어 prednisolone (Solondo, Yuhan, Seoul, Korea) 55 mg (1 mg/kg), cyclosporine (CIPOL-N, Suheung Co., Ltd., Seoul, Korea) 100 mg 하루 1회경구투여로전환하였다. 점안제로는 2% homatropine hydrobromide 양안하루 3회, 1% prednisolone acetate (Predforte, Allergan, Irvine, CA, USA) 양안하루 6회점안하였다. 1달뒤양안전안부염증및장액망막박리모두소실되었다. 3달뒤최대교정시력 (logmar) 양안 0.0, 안압은우 안 18 mmhg, 좌안 16 mmhg였고중심부전방깊이는각막두께의 4배로유지되었으며이후경과관찰기간동안안압상승, 전안부염증또는장액망막박리의재발은없었다. 고찰 보그트-고야나기-하라다병은멜라닌세포를함유하는조직의탈색을일으키는전신적인자가면역질환으로양측성의육아종성전체포도막염이대표적인눈증상이다. 치료와연관된부신피질호르몬의장기사용에따른백내장과녹내장이대표적인합병증으로진단된환자중 20% 에서녹내장이발생한다고알려져있다. 4 치료중간에장액망막박리의악화와함께전방깊이의감소와폐쇄각녹내장이나타나는경우는종종보고된바있으나 5 급성폐쇄각녹내장이선행한비전형적인보그트-고야나기-하라다병의예는비교적드문편으로해외문헌에서 Rathinam et al 6 이 3예, Yang et al 7 이 8예, Yao et al 8 이 4예를보고한바있으나, 아직국내에서는보고된바가없다. 보그트-고야나기-하라다병은초기에는후극부를중심으로생기고면역억제제및부신피질호르몬치료중에는전안부염증의형태로재발하는것이일반적인경우이다. 하지만병의기전상멜라닌세포의염증으로, 멜라닌세포가있는곳이라면전신의어느곳이든지염증이생길수있다. 980

- 최민규외 : 급성폐쇄각녹내장을동반한하라다병 - A B C D E F G H Figure 3. Fundus photographs, optical coherence tomographic images and fluorescein angiographic images in both eyes.

4 - 최민규외 : 급성폐쇄각녹내장을동반한하라다병 - A B C D E F G H Figure 3. Fundus photographs, optical coherence tomographic images and fluorescein angiographic images in both eyes. Fundus photographs show disc swelling and serous retinal detachment in both eyes (A, B). Optical coherence tomography images demonstrate serous retinal detachment in both eyes (C, D). Fluorescein angiographic images at early phase do not show hyperfluorescence matching with findings in optical coherence tomography in both eyes (E, F) and peripheral leakages were observed at late phase in both eyes (G, H) on 3 weeks after onset. 따라서주변부섬모체에생긴염증이섬유주에염증을야기하여안압을상승시키는한편, 섬모체의부종을만들어섬모체를앞으로전위시킴으로써섬모체소대가이완되고섬모체, 홍채, 수정체가앞으로이동하여홍채-수정체가로막이앞쪽으로이동하였을것으로생각할수있다 급성폐쇄각녹내장이선행하는경우초기에녹내장으로진단된다면정확한진단이늦어질수있고필요하지않은시술을받게될수있다. Yang et al 7 은초기에전방각폐쇄양상을보인보그트- 고야나기-하라다병 8명, 16안중 6안에서불필요한섬유주절제술을받았고보그트-고야나기-하라다병으로진단되기까지평균 37일이소요되었다고보고하 였다. 본증례또한양안에불필요한레이저홍채절개술, 아르곤레이저주변홍채성형술을시행하였고, 보그트-고야나기 -하라다병의진단은약 2주가량지연되었다. 불필요한시술과진단의지연을막기위하여기존에보고되었던증례들의특징을파악할필요가있는데첫째, 전형적인급성폐쇄각녹내장환자들에비하여안압상승의정도가높지않다는것이다. Rathinam et al 6 이보고한 3예, 6안의안압은 mmhg, Yang et al 7 이보고한 8예, 16안의안압은 mmhg, Yao et al 8 이보고한 4예, 8안의안압은 mmhg 였으며본증례또한안압이양안 34 mmhg 였다. 둘째, 급성폐쇄각녹내장으로보이는보그트-고야나 981

5 - 대한안과학회지 2018 년제 59 권제 10 호 - 기-하라다병은남성에비하여여성에서다소흔하게발견된다. Yao et al 8 의보고에서는 4예중 2예만이여성이었으나, Rathinam et al 6 의보고에서는 3예중 2예가, Yang et al 7 의보고에서는 8예중 6예가여성이었다. 셋째, 급성폐쇄각녹내장의경우주로 60대에생기는것으로알려져있으나 12 보그트-고야나기-하라다병은주로 30-40대의젊은층에서주로발생한다. 13 급성폐쇄각녹내장으로나타난보그트-고야나기-하라다병의증례보고들에서도주로 10-50대의연령분포를보이며, 60대이후는매우드물다. 넷째, 망막빛간섭단층촬영이나초음파생체현미경검사에서장액망막박리또는섬모체부종등을확인할수있다. 요약하면, 비교적젊은여성에서 40 mmhg 미만의안압상승이동반된급성폐쇄각녹내장의경우본증례와같은급성폐쇄각녹내장으로나타난보그트-고야나기-하라다병을의심해볼수있다. 본증례는발병초기에급성폐쇄각녹내장양상을보여레이저치료후단순하게녹내장만생각하다진단이지연된비전형적인보그트-고야나기-하라다병으로, 국내에는보고된바가없기에문헌고찰과함께보고하는바이다. REFERENCES 1) Yang P, Ren Y, Li B, et al. Clinical characteristics of Vogt- Koyanagi-Harada syndrome in Chinese patients. Ophthalmology 2007;114: ) Sugiura S. Vogt-Koyanagi-Harada disease. Jpn J Ophthalmol 1978;22: ) Forster DJ, Rao NA, Hill RA, et al. Incidence and management of glaucoma in Vogt-Koyanagi-Harada syndrome. Ophthalmology 1993;100: ) Ofri R, Shub N, Galin Z, et al. Effect of reproductive status on intraocular pressure in cats. Am J Vet Res 2002;63: ) Kim JH, Lim JS, Lee JW, et al. Bilateral acute myopia and angle-closure due to ciliochoroidal effusion in Vogt-Koyanagi- Harada syndrome. J Korean Ophthalmol Soc 2012;53: ) Rathinam SR, Namperumalsamy P, Nozik RA, Cunningham ET Jr. Angle closure glaucoma as a presenting sign of Vogt-Koyanagi- Harada syndrome. Br J Ophthalmol 1997;81: ) Yang P, Liu X, Zhou H, et al. Vogt-Koyanagi-Harada disease presenting as acute angle closure glaucoma at onset. Clin Exp Ophthalmol 2011;39: ) Yao J, Chen Y, Shao T, et al. Bilateral acute angle closure glaucoma as a presentation of vogt-koyanagi-harada syndrome in four chinese patients: a small case series. Ocul Immunol Inflamm 2013;21: ) Gohdo T, Tsukahara S. Ultrasound biomicroscopy of shallow anterior chamber in Vogt-Koyanagi-Harada syndrome. Am J Ophthalmol 1996;122: ) Kawano Y, Tawara A, Nishioka Y, et al. Ultrasound biomicroscopic analysis of transient shallow anterior chamber in Vogt- Koyanagi-Harada syndrome. Am J Ophthalmol 1996;121: ) Kishi A, Nao-i N, Sawada A. Ultrasound biomicroscopic findings of acute angle-closure glaucoma in Vogt-Koyanagi-Harada syndrome. Am J Ophthalmol 1996;122: ) Edwards RS. Behaviour of the fellow eye in acute angle-closure glaucoma. Br J Ophthalmol 1982;66: ) Beniz J, Forster DJ, Lean JS, et al. Variations in clinical features of the Vogt-Koyanagi-Harada syndrome. Retina 1991;11:

- 최민규외 : 급성폐쇄각녹내장을동반한하라다병 - = 국문초록 = 급성폐쇄각녹내장을동반한비전형적인보그트 - 고야나기 - 하라다병 1 예 목적 : 급성폐쇄각녹내장이선행한비전형적인보그트 - 고야나기 - 하라다병 1 예를보고하고자한다. 증례요약 : 48 세여자가 4 일전부터시작된두통및안통을동반한양안시력저하를주소로내원하였다.

6 - 최민규외 : 급성폐쇄각녹내장을동반한하라다병 - = 국문초록 = 급성폐쇄각녹내장을동반한비전형적인보그트 - 고야나기 - 하라다병 1 예 목적 : 급성폐쇄각녹내장이선행한비전형적인보그트 - 고야나기 - 하라다병 1 예를보고하고자한다. 증례요약 : 48 세여자가 4 일전부터시작된두통및안통을동반한양안시력저하를주소로내원하였다. 환자는 10 년간경구용피임약복용의과거력이외에특이사항은없었다. 타병원에서처방받은안압하강제사용후안압은정상화되었으나양안전방의깊이는여전히얕았고, 홍채 - 수정체가로막이전방으로전위되어있어본원으로진료의뢰되었다. 본원에서레이저홍채절개술시행후전방의깊이는불완전하게회복되었으나, 여전히시력은회복되지않았으며안구초음파검사에서맥락막삼출소견을확인하였다. 양안의전안부염증과빛간섭단층촬영에서황반부에막구조물로구획화된장액망막박리가관찰되었으나, 전형적인보그트 - 고야나기 - 하라다병과는달리형광안저혈관조영술에서는이와일치하는다발성의형광누출소견은보이지않았다. 비전형적인보그트 - 고야나기 - 하라다병으로진단하고점안치료와더불어고용량부신피질호르몬요법후부신피질호르몬과면역억제제를경구투여하였다. 1 개월뒤장액망막박리는소실되었고 3 개월뒤시력은호전되었으며현재까지재발없이경과관찰중이다. 결론 : 본증례를통하여비전형적인보그트 - 고야나기 - 하라다병의초기소견으로급성폐쇄각녹내장이선행해서나타날수있음을경험하였다. 따라서레이저홍채절개술을비롯한폐쇄각녹내장의치료시행후에도호전되지않을경우보그트 - 고야나기 - 하라다병을포함한다른질환의동반가능성을반드시염두에두고, 추적관찰을해야할것이다. < 대한안과학회지 2018;59(10): > 최민규 / Min Gyu Choi 중앙대학교의과대학중앙대학교병원안과학교실 Department of Ophthalmology, Chung-Ang University Hospital, Chung-Ang University College of Medicine 983

<283732372D3733312920B4D9C3CAC1A120BCD2C7C1C6AEC4DCC5C3C6AEB7BBC1EEC0C720B3EBBEC8C0C720BDC3B7C2BAB8C1A4BFA120B4EBC7D120C0AFBFEBBCBA20C6F2B0A1283035292E687770>

<283732372D3733312920B4D9C3CAC1A120BCD2C7C1C6AEC4DCC5C3C6AEB7BBC1EEC0C720B3EBBEC8C0C720BDC3B7C2BAB8C1A4BFA120B4EBC7D120C0AFBFEBBCBA20C6F2B0A1283035292E687770> 대한안과학회지 제 49 권 제 5 호 2008 J Korean Ophthalmol Soc 49(5):727-731, 2008 DOI : 10.3341/jkos.2008.49.5.727 다초점 소프트콘택트렌즈의 노안의 시력보정에 대한 유용성 평가 김현경 1 김효명 2 정성근 1 가톨릭대학교 의과대학 성모병원 안과학교실 1, 고려대학교 의과대학 안암병원 안과학교실