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1 대한안과학회지 2014 년제 55 권제 10 호 J Korean Ophthalmol Soc 2014;55(10): pissn: eissn: ase Report 장액망막박리가동반된레버특발성성상신경망막염에서안내베바시주맙주사효과 1 예 Intravitreal evacizumab Injection for Serous Retinal Detachment ssociated with Leber s Idiopathic Stellate Neuroretinitis 이동현 1 고형준 1 김성수 2 이성철 2 김민 1 Dong Hyun Lee, MD 1, Hyoung Jun Koh, MD, PhD 1, Sung Soo Kim, MD, PhD 2, Sung hul Lee, MD, PhD 2, Min Kim, MD 1 연세대학교의과대학강남세브란스병원안과학교실 1, 연세대학교의과대학세브란스병원안과학교실 2 Department of Ophthalmology, Gangnam Severance Hospital, Yonsei University ollege of Medicine 1, Seoul, Korea Department of Ophthalmology, Severance Hospital, Yonsei University ollege of Medicine 2, Seoul, Korea Purpose: To report the effectiveness of intravitreal bevacizumab treatment for serous retinal detachment associated with Leber s idiopathic stellate neuroretinitis. ase summary: 56-year-old male visited our clinic complaining of visual disturbance for three days in his right eye. His best corrected visual acuity was 0.5. Relative afferent pupillary defect and pain when moving eyes were noted in his right eye as well as inflammatory cells in the vitreous cavity. On funduscopic examination, disc swelling with hemorrhage and stellate-shaped hard exudates were noted at the perifovea. Ishihara color vision test showed anomalous trichromacy in his right eye. Hyperfluorescence around the disc was observed on fundus fluorescein angiography. Optical coherence tomography showed disc swelling with serous retinal detachment at the fovea. Inferior altitudinal scotoma was noted on visual field examination. The patient underwent intravitreal bevacizumab injection and topical steroid medication. fter six days, the patient s symptoms and disc swelling improved, and decreased subretinal fluid was observed. fter six weeks, his best corrected visual acuity was 1.0. Nine weeks later, visual field examination showed nonspecific scotoma. onclusions: Intravitreal bevacizumab injection is useful for treating serous retinal detachment associated with Leber s idiopathic stellate neuroretinitis. J Korean Ophthalmol Soc 2014;55(10): Key Words: evacizumab, Leber s idiopathic stellate neuroretinitis, Optic disc edema, Serous retinal detachment Received: Revised: ccepted: ddress reprint requests to Min Kim, MD Department of Ophthalmology, Gangnam Severance Hospital, #211 Eonju-ro, Gangnam-gu, Seoul , Korea Tel: , Fax: minkim76@yuhs.ac * This study was presented as a poster at the 111th nnual Meeting of the Korean Ophthalmological Society 신경망막염은시신경유두부종및그에따라오는별모양의황반삼출물을특징으로하는염증성질환이다. 1 알려진원인으로는세균, 바이러스, 진균, 기생충감염, 자가면역질환등이있다. 감염성질환중에서가장흔한형태는 cat scratch disease로이질환은 artonella henselae에의해유발되는것으로알려졌다. 2 신경망막염의치료로는, 감염원인에대한치료및염증조절을위한정맥혹은경구스테로이드치료, 면역억제치료를고려할수있다. 1 그중에서도 Leber 특발성성상신경망막염 (Leber s idiopathic stellate c2014 The Korean Ophthalmological Society This is an Open ccess article distributed under the terms of the reative ommons ttribution Non-ommercial License ( which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited. 1562

- 이동현외 : 베바시주맙을이용한신경망막염치료 - neuroretinitis) 은 1916년 Theodor Leber에의해처음기술된질환으로, 대개단측성으로발생하고, 안저검사상성상황반부삼출물이있으며, 저절로호전되는경과를거치고, 알려진원인이없는것을특징으로하는질환이다.

2 - 이동현외 : 베바시주맙을이용한신경망막염치료 - neuroretinitis) 은 1916년 Theodor Leber에의해처음기술된질환으로, 대개단측성으로발생하고, 안저검사상성상황반부삼출물이있으며, 저절로호전되는경과를거치고, 알려진원인이없는것을특징으로하는질환이다. 3 베바시주맙 (evacizumab) 은항혈관내피세포성장인자 (antivascular endothelial growth factor, anti-vegf) 로다양한안과질환에서적용되고있는약제이며, 특히연령관련황반변성, 4 증식당뇨망막병증 5 등의치료에서그효과를인정받고있다. kesbi et al 6 은 IRVN (Idiopathic retinitis, vasculitis, aneurysms, and neuroretinitis) 환자에서유리체강내베바시주맙주사, 유리체절제술및안내레이저술을시행한후망막신생혈관이퇴화하고시력이호전된증례를보고하였으며, Sawhney et al 7 역시 IRVN 환자에서유리체강내베바시주맙주사, 범망막광응고술, 유리체절제술을병용하여치료한사례를발표한바있다. 본저자들은장액성망막박리를동반한신경망막염환자에서유리체강내베바시주맙주사치료시행후호전된증례 1예를경험하였기에이를보고하고자한다. 증례보고 특별한병력이없는 56세남자환자가 3일전부터검정색물체가우안을가리는증상을주소로내원하였다. 우안 시야장애외에전신적으로호소하는다른증상은없었다. 최대교정시력은우안 0.5, 좌안 1.0, 안압은양안정상이었다. 전안부검사상특이소견없었으며, 양안대광반사는정상이었으나우안의상대적구심성동공운동장애를보이고있었다. 우안의경미한안구이동통을호소하였다. 안저검사상우안시신경유두부종및경계불명확, 시신경유두출혈, 황반부의저색소성변화, 성상경성삼출물, 망막색소상피층의반점이관찰되었다 (Fig. 1). 색각검사상우안전색약소견이보였다. 형광안저촬영상초기동맥기부터우안시신경유두에서누출로인한과형광소견보였으며, 이후정맥기, 후기에도과형광이지속되었다 (Fig. 2). 빛간섭단층촬영상시신경유두부종, 황반부의장액성망막박리가관찰되었다 (Fig. 3). 시야검사상우안하측의수평시야결손이관찰되었다 (Fig. 4). 전방에염증세포는관찰되지않았으나유리체강에염증세포가 0.5+ 만큼관찰되었다. 세균, 결핵균, 매독, 단순헤르페스바이러스, 대상포진바이러스, 거대세포바이러스, 간염, 톡소포자충및개회충, 적혈구침강속도, 반응성단백질, 항핵항체, 항중성구세포질항체등의혈청학적검사및단순흉부촬영을시행하였고, 유리체강내베바시주맙 (vastin, Genentech, San Francisco, ) 1.25 mg 주사치료및스테로이드안약 (Prednisolone acetate) 하루 4회점안치료를시작하였다. 혈청학적검사및단순흉부촬영상특이이상소견보이지않았다. D Figure 1. onsecutive fundus color photographs of the patient. t the initial visit, disc swelling with blurring of the disc margin, disc hemorrhage, and stellateshaped hard exudates at the perifovea are noted (). Six days after the first injection of bevacizumab, improvement of disc swelling but slight increased exudates at the fovea was noted (). Six weeks after injection of bevacizumab (). Note the resolution of disc swelling and improvement of stellate-shaped hard exudates at the perifovea (). Fifteen weeks after the first visit, disc swelling with hemorrhage is completely regressed, and hard exudates remaining at the perifovea are markedly decreased compared to the fundus color photograph taken at 6 days after the first injection of bevacizumab (D). 1563

- 대한안과학회지 2014년 제 55 권 제 10 호 - Figure 2. Fundus fluorescein angiography at the initial visit.

Fundus fluorescein angiography at the initial visit (,, ).

onsecutive optical coherence tomography (OT) images of the patient.

Six days after injection of bevacizumab, improvement of serous retinal detachment with decreased amount of subretinal fluid is

Six weeks after injection of bevacizumab, note the complete resolution of subretinal fluid and disruption of inner segment/outer

Fifteen weeks after the first visit, disrupted IS/OS junction line at the nasal perifovea shows more improvement compared to the

가리는 증상이 호전되었다. 안저검사상 시신경유두부종이 3). 주사치료 9주 후 시야검사상 우안의 비특이적인 암점 감소하였으나 성상 경성삼출물이 증가하였다(Fig. 1). 빛 을 제외한 다른 시야장애 소견 관찰되지 않았다(Fig.

3 - 대한안과학회지 2014년 제 55 권 제 10 호 - Figure 2. Fundus fluorescein angiography at the initial visit. Early hyperfluorescence at the optic disc continues until the late phase. Fundus fluorescein angiography at the initial visit (,, ). Early hyperfluorescence at the optic disc () continues until the late phase (, ). D Figure 3. onsecutive optical coherence tomography (OT) images of the patient. Serous retinal detachment at the fovea and around the disc with subretinal fluid is noted (). Six days after injection of bevacizumab, improvement of serous retinal detachment with decreased amount of subretinal fluid is noted (). Six weeks after injection of bevacizumab, note the complete resolution of subretinal fluid and disruption of inner segment/outer segment (IS/OS) junction line at the nasal perifovea (). Fifteen weeks after the first visit, disrupted IS/OS junction line at the nasal perifovea shows more improvement compared to the OT image taken at 6 weeks after injection of bevacizumab (D). 주사치료 6일째 환자가 호소하던, 검정색 물체가 우안을 분절의 접합부의 국소적인 결손 소견이 호전되었다(Fig. 가리는 증상이 호전되었다. 안저검사상 시신경유두부종이 3). 주사치료 9주 후 시야검사상 우안의 비특이적인 암점 감소하였으나 성상 경성삼출물이 증가하였다(Fig. 1). 빛 을 제외한 다른 시야장애 소견 관찰되지 않았다(Fig. 4). 간섭단층촬영 결과상 망막하액의 감소를 확인하였다(Fig. 주사치료 15주 후 극소량의 성상 경성삼출물만 관찰되었으 3). 주사치료 3주 후 우안 최대교정시력은 0.8로 호전되었 며(Fig. 1D), 빛간섭단층촬영상 내분절과 외분절의 접합부 으며, 안저검사상 성상 경성삼출물도 줄어들었다. 빛간섭단 의 국소적인 결손 소견이 더욱 회복되었다(Fig. 3D). 층촬영상 망막하액이 완전히 흡수된 소견이 관찰되었으나 망막 내분절과 외분절의 접합부(Inner segment/outer seg- 고 찰 ment junction line, IS/OS junction line)의 국소적인 결손을 보였다. 주사치료 6주 후 우안 최대교정시력은 1.0으로 더 본 증례는 전형적인 Leber 특발성 성상 신경망막염의 양 욱 좋아졌으며, 안저검사상 성상 경성삼출물의 감소, 시신 상을 보였던 사례로, 인플루엔자 양 증상(influenza-like ill- 경유두부종의 관해가 관찰되었다(Fig. 1). 빛간섭단층촬영 ness)은 없었으나 특징적인 단안 시신경유두부종 및 성상 상 망막하액이 관찰되지 않았으며, 중심와의 내분절과 외 경성삼출물을 보였으며, 혈청학적 검사를 통해 감염 및 자 1564

4 - 이동현외 : 베바시주맙을이용한신경망막염치료 - Figure 4. utomated visual field at the initial visit () and after 9 weeks (). lthough the reliability of the examination at 9 weeks is lower inferior altitudinal scotoma had disappeared at 9 weeks, compared to the initial visual field. POS = positive; NEG = negative; S = apostilbs; RX = prescription; DS = diopter sphere; D = diopter cylinder; X = axis; GHT = glaucoma hemifield test; VFI = visual field index; MD = mean deviation; PSD = pattern standard deviation. 가면역질환에의한신경망막염을배제할수있었던경우이다. Jung et al 8 에따르면시신경유두부종을보였던한국인환자의 58안중 3안에서신경망막염이관찰된바있으며, 국내에서 Han et al 9 에의해 Leber 특발성성상신경망막염이보고된바있다. 신경망막염환자에서염증의조절을위해경구혹은정맥스테로이드치료를시행할수있으며, Empeslidis et al 10 은유리체출혈을동반한 IRVN 증후군환자에서경구스테로이드치료및유리체절제술후에도호전을보이지않는황반부종에대해유리체강내덱사메타손 (Dexamethasone) 삽입물주사를시행한후황반부종이호전된사례를보고한바있다. 한편유리체강내베바시주맙주사는시신경유두혈관염, 11 방사선시신경병증, 12 앞허혈시신경병증, 13 당뇨시신경유두병증 14 등여러시신경관련질환에서효과가있는것으로알려졌으며, akir et al 15 은심한황반부종이동반된특발성신경망막염환자에서유리체강내베바시주맙주사및트리암시놀론주사를시행 하여치료한사례를보고한바있다. 베바시주맙이신경망막염환자에서시신경유두부종의호전및조기회복을유발하는정확한기전은알려진바없지만, Rappoport et al 16 에의하면망막신경손상 (optic nerve crush) 마우스모델을대상으로유리체강내베바시주맙주사를시행했을때시신경유두부종의감소, 모세혈관누출의감소, 시신경미세혈관구조관류의증가및신경절세포복합체의세포자멸사감소가관찰됨을밝혔다. 이같은연구결과로미루어볼때혈관내피세포성장인자가혈관투과성 (vascular permeability) 을증가시켜시신경유두부종및장액망막박리를일으키며, 그에따라유리체강내로항혈관내피세포성장인자를주입하여혈관내피세포성장인자에의한혈관투과성 (vascular permeability) 증가를억제하고, 시신경유두부종및장액망막박리를호전시킬수있을것으로생각한다. 본증례는이에근거하여 Leber 특발성성상신경망막염환자에서유리체강내베바시주맙주사치료를일차치료로선택하였고, 시 1565

5 - 대한안과학회지 2014 년제 55 권제 10 호 - 력, 증상및검사소견상의호전을불러올수있었다. 결론적으로, 장액성망막박리를동반한 Leber 특발성성상신경망막염환자에서유리체강내베바시주맙주입술이치료로서사용될수있다. REFERENES 1) Purvin V, Sundaram S, Kawasaki. Neuroretinitis: review of the literature and new observations. J Neuroophthalmol 2011;31: ) Suhler E, Lauer K, Rosenbaum JT. Prevalence of serologic evidence of cat scratch disease in patients with neuroretinitis. Ophthalmology 2000;107: ) T L. Die pseudonephritischen Netzhauterkrnakungen, die Retinitis stellata: Die Purtschersche Netzhauteffektion nach schwerer Schadelverletzung. In: Graefe, Saemisch T, eds. Graefe- Saemisch Handbuch der Gesamten ugerheikunde, 2nd ed. Leipzig: Englemann, 1916; v ) very RL, Pieramici DJ, Rabena MD, et al. Intravitreal bevacizumab (vastin) for neovascular age-related macular degeneration. Ophthalmology 2006;113: e5. 5) very RL, Pearlman J, Pieramici DJ, et al. Intravitreal bevacizumab (vastin) in the treatment of proliferative diabetic retinopathy. Ophthalmology 2006;113:1695.e ) kesbi J, rousseaud FX, dam R, et al. Intravitreal bevacizumab (vastin) in idiopathic retinitis, vasculitis, aneurysms and neuroretinitis. cta Ophthalmol 2010;88:e ) Sawhney GK, Payne JF, Ray R, et al. ombination anti-vegf and corticosteroid therapy for idiopathic retinal vasculitis, aneurysms, and neuroretinitis syndrome. Ophthalmic Surg Lasers Imaging Retina 2013;44: ) Jung JJ, aek SH, Kim US. nalysis of the causes of optic disc swelling. Korean J Ophthalmol 2011;25: ) Han DH, Sohn HJ, Lee DY, Nam DH. Leber's idiopathic stellate neuroretinitis with peripapillary serous retinal detachment. J Korean Ophthalmol Soc 2011;52: ) Empeslidis T, anerjee S, Vardarinos, Konstas G. Dexamethasone intravitreal implant for idiopathic retinal vasculitis, aneurysms, and neuroretinitis. Eur J Ophthalmol 2013;23: ) Erdurman F, Durukan H, Mumcuoğlu T, Hürmeriç V. Intravitreal bevacizumab treatment of macular edema due to optic disc vasculitis. Ocul Immunol Inflamm 2009;17: ) Finger PT. nti-vegf bevacizumab (vastin) for radiation optic neuropathy. m J Ophthalmol 2007;143: ) ennett JL, Thomas S, Olson JL, Mandava N. Treatment of nonarteritic anterior ischemic optic neuropathy with intravitreal bevacizumab. J Neuroophthalmol 2007;27: ) l-dhibi H, Khan O. Response of diabetic papillopathy to intravitreal bevacizumab. Middle East fr J Ophthalmol 2011;18: ) akir M, ekiç O, ozkurt E, et al. ombined intravitreal bevacizumab and triamcinolone acetonide injection for idiopathic neuroretinitis. Ocul Immunol Inflamm 2009;17: ) Rappoport D, Morzaev D, Weiss S, et al. Effect of intravitreal injection of bevacizumab on optic nerve head leakage and retinal ganglion cell survival in a mouse model of optic nerve crush. Invest Ophthalmol Vis Sci 2013;54: = 국문초록 = 장액망막박리가동반된레버특발성성상신경망막염에서안내베바시주맙주사효과 1 예 목적 : 장액성망막박리를동반한 Leber 특발성성상신경망막염환자에서유리체강내베바시주맙주사시행후호전된증례 1 예를경험하였기에보고하고자한다. 증례요약 : 특별한병력이없는 56 세남자가 3 일전부터발생한우안시야장애로내원하였다. 우안최대교정시력 0.5, 상대적구심성동공운동장애및안구이동통, 유리체강의염증세포, 안저검사상시신경유두부종및출혈, 성상경성삼출물이있었다. 색각검사상전색약, 형광안저촬영상시신경유두과형광, 빛간섭단층촬영상시신경유두부종, 황반부장액성망막박리, 시야검사상하측수평시야결손을나타냈다. 유리체강내베바시주맙주사치료및스테로이드안약치료를하였고, 치료 6 일째증상호전및시신경유두부종의호전, 망막하액의감소가관찰되었다. 6 주후시력 1.0 으로회복되고, 9 주후시야검사상비특이적암점외이상소견보이지않았다. 결론 : 장액성망막박리를동반한 Leber 특발성성상신경망막염환자에서유리체강내베바시주맙주입술이치료로서사용될수있다. < 대한안과학회지 2014;55(10): > 1566

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<32332D30332D323328C3D6B0E6BDC42DBDC5C1F8C8F E687770> 대한안과학회지 2013 년제 54 권제 8 호 J Korean Ophthalmol Soc 2013;54(8):1298-1302 pissn: 0378-6471 eissn: 2092-9374 http://dx.doi.org/10.3341/jkos.2013.54.8.1298 = 증례보고 = 유리체강내베바시주맙주입술을이용한당뇨병유두병증의치료 1 예 신진희 최경식 순천향대학교의과대학안과학교실