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대한안과학회지 2018 년제 59 권제 6 호 J Korean Ophthalmol Soc 2018;59(6):589-593 ISSN 0378-6471 (Print) ISSN 2092-9374 (Online) https://doi.org/10.3341/jkos.2018.59.6.589 Case Report 인공수정체혼탁으로오인된지연낭폐쇄증후군 1 예 A Case of Late-onset Capsular Block Syndrome, Resulting in the Misdiagnosis of Intraocular Lens Opacity 안민원 김소희 이종수 Min Won Ahn, MD, So Hee Kim, MD, Jong Soo Lee, MD, PhD 부산대학교의과대학부산대학교병원안과학교실 Department of Ophthalmology, Pusan National University Hospital, Pusan National University School of Medicine, Busan, Korea Purpose: We report a case of late-onset capsular block syndrome, which resulted in a misdiagnosis of intraocular lens (IOL) opacity. Case summary: A 59-year-old man visited our clinic with reduced visual acuity in the right eye from 1 year prior. He had undergone uncomplicated bilateral cataract surgery by phacoemulsification with IOL implants at another hospital 10 years before. There was no specific history with the exception of hypertension. After being diagnosed in the ophthalmology clinic with IOL degeneration and opacity in the right eye, he was referred to our hospital for IOL replacement. Upon examination, his right uncorrected visual acuity was 0.06 and intraocular pressure was 22 mmhg. The refractive error could not be checked due to IOL opacity. Slit-lamp microscopy revealed a cloudy, milky IOL. Anterior-segment optical coherence tomography of the right eye showed retention of a highly reflective material in the lens capsule behind the IOL. Posterior capsule enlargement of the right eye was confirmed on ultrasound biomicroscopy. After neodymium-doped yttrium aluminium garnet (Nd:YAG) laser capsulotomy was performed, the homogeneous space disappeared and the eye recovered normal visual acuity. Conclusions: Capsular block syndrome is a rare complication that can occur shortly (1 day to 2 days) after cataract surgery. Late-onset capsular block syndrome, which occurs 10 years after surgery differs from typical clinical manifestations. Thus, capsular block syndrome is an important consideration upon the presentation of opacification due to IOL degeneration. J Korean Ophthalmol Soc 2018;59(6):589-593 Keywords: Capsular block syndrome, Intraocular lens opacification, Misdiagnosis, Neodymium-doped yttrium aluminium garnet (Nd:YAG) laser Received: 2018. 2. 8. Revised: 2018. 3. 29. Accepted: 2018. 5. 25. Address reprint requests to Jong Soo Lee, MD, PhD Department of Ophthalmology, Pusan National University Hospital, #179 Gudeok-ro, Seo-gu, Busan 49241, Korea Tel: 82-51-240-7321, Fax: 82-51-242-7341 E-mail: jongsool@pusan.ac.kr * This study was supported by a grant from University Research Park Project of Busan National University funded by Busan Institute of S&T Evaluation and Planning (No.201726550002). * Conflicts of Interest: The authors have no conflicts to disclose. 낭폐쇄증후군은백내장수술시전낭원형절개를하고후방인공수정체를낭내삽입한경우에발생하는드문합병증이다. 1 전낭원형절개면과인공수정체광학부와접촉및수정체낭의팽창과수정체낭내액화된물질의축적을특징으로한다. 대표적인임상양상으로는인공수정체의광학부및홍채의전방이동, 전방의얕아짐, 예기치않은근시발생, 광학부와후낭사이의혼탁물에의한공간증가, 안압상승등이있다. 발생시기에따라낭폐쇄증후군은백내장수술중수력분리술후수정체핵탈구에의한수술중낭폐쇄증후군, 수술 c2018 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. 589

- 대한안과학회지 2018 년제 59 권제 6 호 - 직후발생하는초기낭폐쇄증후군과액화된후발백내장이동반되는지연낭폐쇄증후군으로나눌수있다. 1 낭폐쇄증후군은대부분수술직후에발생하지만지연성으로수술후 3.8-8.5년후에도발생한다. 1-3 그러나후방인공수정체가낭내삽입되는백내장수술후 10년이상지연낭폐쇄증후군은보고되지않았다. 낭폐쇄증후군의발생기전은아직불명확하고, 발생시기나형태에따라주요원인이다르기에치료법또한적절하게적용해야한다. 4 수술후초기에발생하는경우근시로의이행이나인공수정체및홍채의전방이동, 인공수정체와수정체후낭사이의공간증가등특징적인형태와임상양상으로쉽게진단이이루어진다. 그러나지연성으로발생하는경우특징적인근시로의이행이나인공수정체의전방이동에따른형태학적소견이쉽게관찰되지않을경우, 본증례와같이친수성아크릴인공수정체의칼슘침착으로인한인공수정체의광범위한혼탁으로오인할수있어문헌고찰과함께증례를보고하고자한다. A B 증례보고 59세남자환자가 1년전부터진행된우안시력저하를주소로인근안과에서인공수정체혼탁의의심소견이관찰되어인공수정체교체를위해본원으로전원되었다. 과거력에서 10년전개인안과의원에서양안수정체유화술및후방인공수정체수정체낭내삽입술을시행받았다. 내원당시나안시력은 0.06이었으며, 교정은되지않았다. 안압은 22 mmhg로약간상승되어있었지만안압하강제를사용할정도는아니었다. 우안의굴절력은인공수정체부위의혼탁때문에측정되지않았다. 세극등현미경검사에서산동전에는인공수정체의광범위혼탁처럼보였으나, 산동후에는전낭원형절개면이섬유화되어인공수정체광학부와유착되어있었으며혼탁한유백색물질이인공수정체뒤쪽수정체낭내주로하부의공간에서관찰되고, 후낭이팽창되어있었다 (Fig. 1A). 전안부빛간섭광학단층촬영에서인공수정체뒤쪽으로수정체낭내고반사물질의저류가관찰되었고 (Fig. 2A), 초음파생체현미경검사에서는수정체후낭의팽창을확인하였다 (Fig. 2B). 인공수정체의광범위혼탁이아닌지연낭폐쇄증후근으로진단하고 Neodymium-doped yttrium aluminium garnet (Nd:YAG) 레이저후낭절개술을시행하였다. 수정체낭내유백색물질에의해후낭시야확보가어려웠지만비교적시야가확보되는수정체낭의주변부를중심으로먼저, 수정체후낭을절개하여혼탁액체물을유리체강으로배출시킨후시야가확보되면서십자형절개방식으로성공적인레이저후낭절개술을시행하였다. 시행직 Figure 1. Slit lamp photography of the right eye. (A) The anterior capsular incision was fibrous and adhered to the optic part of intraocular lens (IOL), cloudy milky material was observed in the intraocular lens space behind the IOL. And the posterior capsule was swollen. (B) After the neodymium-doped yttrium aluminium garnet (Nd:YAG) capsulotomy, milky white material was not observed behind the intraocular lens space and the uncorrected visual acuity was 1.0. 후즉시우안의나안시력은 0.7로상승되었다. 이차적인염증발생방지를위해플루오로메톨론 0.1% (Ocumetholone, Samil, Seoul, Korea), 항생제점안액을하루 3회처방하였다. Nd:YAG 레이저후낭절개술후 2일째나안시력은 1.0으로상승되었고, 안압상승이나인공수정체손상, 탈구와같은합병증은없었다 (Fig. 1B). 약 1달후외래추적관찰에서는나안시력 1.0과정상의안압소견을얻었다. 고 찰 낭폐쇄증후군은 Davison 5 과 Holtz 6 가처음보고하였고, 1-1.6% 의빈도로대부분술후 1-2주째발생한다. 그특징으로백내장수술후전낭원형절개면경계와인공수정체광학부사이의접촉및폐쇄에따른인공수정체와후낭사이의폐쇄적공간, 액체의고임에따른후낭팽창을들었다. 국내보고로는 0.73-1.45% 의발생빈도로보고되고술후 2 주이내에발생한초기낭폐쇄증후군이보고되고있지 590

- 안민원외 : 지연낭폐쇄증후군 1 예 - A B Figure 2. Anterior optical coherence tomography and ultrasound biomicroscopy. (A) Anterior segment optical coherence tomography of the right eye showed retention of highly reflective material in the lense capsule behind the intraocular lens. Arrow showed Retention of highly reflective material in the capular bag. (B) Posterior capsule enlargement of right eye was confirmed on ultrasound biomicroscopy. Arrow showed capsular bag distension. IR = infrared; ART = automatic real-time tracking; OCT = optical coherence tomography; HR = high resolution. 만, 4,7,8 아직지연낭폐쇄증후군의임상적보고는없고증례보고만있으며, 수술후 10년이상발생한보고는없다. 낭폐쇄증후군은백내장수술시전낭원형절개를하고후방인공수정체를낭내삽입한경우에발생하는합병증으로알려져있지만, 통조림따기낭절개를통한백내장낭외적출술시행후, 2 인공수정체섬모체고랑삽입술시에도발생한보고가있다. 9,10 이같은경우는전낭절개면이인공수정체광학부전면또는후면에의해완전히폐쇄되어발생한다. Miyake et al 1 이발생시기에따라낭폐쇄증후군을수술중발생, 수술후초기발생과지연성발생 3가지로분류하였다. 수술중발생하는경우는수력분리술시많은양의평형염액을빠르게주입하여수정체핵탈구로전낭원형절개면이수정체핵과폐쇄되어수정체낭에평형염액이갇히는형태로후극백내장이나긴안축장을가진눈과관련성이있다. 합병증으로후낭파열과이로인한수정체의유리체강내로의후방편위가발생할수있다. 유리체절제술로치료할수있으며, 예방을위해서는수력분리술시조심스럽게천천히진행해야한다. 수술후초기에발생하는낭폐쇄증후군은대부분수술후 1일에서 2주이내에발생하는경우로인공수정체후방에잔여점탄물질이남아유발하는것으로알려져있다. Sugiura et al 11 이초기낭폐쇄증후군을대상으로수정체낭내액체를분석한결과수술중사용된점탄물질과거의동일한성분이라는것을보고하여완전히제거되지못한점 탄물질이초기낭폐쇄증후군의중요한요인이라고제시하였다. 불완전하게제거된점탄물질이나잔여수정체세포와이에따른부산물들에의해, 수정체낭내삼투압이상승하여삼투압의차이를유발하고, 수정체낭이반투과성막으로작용하여수분을수정체낭내로끌어들여후낭팽창이일어난다. 이로인하여전방이얕아지고예기치않은근시가발생하며, 폐쇄각녹내장이합병증으로발생할수있다. Sorenson et al 12 은초기낭폐쇄증후군으로발생한근시가평균 -2.35디옵터(+0.13~-4.5D) 라고보고하고, Nd:YAG 레이저에의해전낭또는후낭절개술로수정체낭내저류된액체를배출하여치료하는것이가장간단한처치방법이다. 낭폐쇄증후군의예방을위해서는수술중수정체낭에존재하는점탄물질을완벽하게제거하는것이매우중요하다. 그러나 Kim and Shin 4 은 Miyake et al 1 분류와달리초기낭폐쇄증후군이잔여점탄물질이없는경우에도발생함을보고하고, 전방의염증반응과섬유성삼출물이전낭원형절개면과인공수정체의밀접한접촉으로발생하는경우는스테로이드안약점안만으로수술이나시술없이치료되기도한다고주장하였다. 수술후지연낭폐쇄증후군은수술후수개월에서수년이지나발생하는매우드문형태로, 국내보고는수술후 8.5년째까지, 2 국외보고는수술후 8년째까지 3 발생을보고하였다. 본증례는지금까지의보고보다긴, 수술후 10년째발생한첫증례이다. 초기낭폐쇄증후군과달리수정체낭내인공수정체후면으로혼탁한유백색물질의축적을특징으로한다. 전낭원형절개면과인공수정체광학부의섬유화로수정체낭이폐쇄된공간이되면, 인공수정체광학부와후낭사이에혼탁한유백색물질이서서히축적되어지연낭폐쇄증후군을유발한다. 지연낭폐쇄증후군은혼탁한유백색액체나후낭혼탁에의한시력저하로, Eifrig 13 는지연낭폐쇄증후군을 Lacteocrumenasia라는용어로명명하기도하였다. 혼탁한유백색물질을전기영동분석한결과, 알파- 크리스탈린 (alpha-crystallin) 의농도는높고상대적으로알부민농도는낮게나타났다. 이는수정체자체또는수정체상피세포로부터수정체낭내축적된물질이기인했다는것을의미하며, 감마글로불린이검출되지않는것은수정체낭내항원-항체반응의가능성이매우낮음을의미한다. Miyake et al 1 과 Kim and Shin 4 은혼탁한유백색물질의측적은수정체상피세포의증식이나가성화생 (pseudometaplasia) 의결과라고주장하기도한다. 지연낭폐쇄증후군에서는수술후초기낭폐쇄증후군과달리인공수정체와홍채의전방이동, 전방의얕아짐, 예기치않은근시, 안압상승등의특징적인임상양상이나타나지않는다. 따라서의심하지않는경우본증례처럼인공수 591

- 대한안과학회지 2018 년제 59 권제 6 호 - 정체광범위혼탁으로오인할수있다. 치료는 Nd:YAG 레이저전낭혹은후낭절개술을시도하며, 혼탁한유백색물질에의해레이저가어려운경우수술적치료를고려해볼수있다. 14 본증례는개인안과에서친수성아크릴인공수정체의칼슘침착으로인한광범위한인공수정체의혼탁에의한시력저하로의심하고인공수정체교체를위해본원으로의뢰되었다. 오인된이유를생각해보면, 본증례에서지연낭폐쇄증후군의특징적인소견인혼탁한유백색물질이홍채에가려져산동검사를시행하지않으면정확하게관찰하기어려운위치에축적되어있으면서초기에발생하는낭폐쇄증후군의전형적인임상소견도없었다는것이다. 또한수정체낭내전반적인혼탁한액체와인공수정체광학부와전낭원형절개면의섬유화로인해마치인공수정체자체의혼탁으로오인되었던경우였다. 백내장수술후지연성친수성아크릴인공수정체혼탁은평균적으로수술후 25.8-29.1개월정도에발생하며, 모든재질의인공수정체에서혼탁이발생할수있지만친수성아크릴인공수정체혼탁이가장많으며인공수정체교체가필요한심각한시력저하를가장많이유발한다. 15,16 아직까지혼탁이발생하는원인이밝혀지지는않았지만대부분칼슘침착에의한혼탁이며, 광학부표면또는표면아래나인공수정체재료내에서발생한다. 대표적인증상은시력저하이며, 당뇨, 녹내장, 포도막염, 백내장수술후과도한염증반응등이연관되어있다고알려져있다. 본증례환자는당뇨, 녹내장, 포도막염과거력이없었고, 발생시기또한수술후 10년이나경과된상황에서초음파생체현미경과전안부빛간섭단층촬영에서인공수정체에의한전방이동과이에따른전방각의얕은현상이있었고, 인공수정체후면으로수정체낭내혼탁된물질저류및후낭의팽창으로인공수정체혼탁과감별이가능하였다. 백내장수술후변성된인공수정체혼탁을감별하기위해서는먼저, 산동후세극등현미경검사로정밀하게검사하는것이필요하고, 확실한해부학적구조를파악하기위해서는전안부빛간섭단층촬영과초음파생체현미경검사가필요하다. 전안부빛간섭단층촬영이대부분의전안부구조를정밀하게평가할수있지만, 홍채의이중색소상피에의한빛차단효과로홍채후면의구조평가에는한계가있어인공수정체광학부, 지지부와그주변조직전체를파악하는데한계가있다. 17 반면초음파생체현미경검사의주된장점은전방각의효과적인평가와인공수정체광학부, 지지부를둘러싼모든구조물을보다쉽게평가할수있다는것이다. 18 본증례에서도두가지검사에서낭폐쇄증후군의전형적인특징인인공수정체후면으로수정체낭내물질 저류와이로인한후낭팽창을관찰하여지연낭폐쇄증후군을확진할수있었다. 본증례처럼지연성으로발생하는낭폐쇄증후군은인공수정체의광학부및홍채의전방이동, 전방의얕아짐, 예기치않은근시발생, 광학부와후낭사이의혼탁물에의한공간증가, 안압상승등의특징적인소견이관찰되지않기에진단이힘들수있다. 비록낭폐쇄증후군은드물지만모든형태의백내장수술후인공수정체를삽입하는경우에는발생할수있기에, 항상진단에각별한관심을가지는것이중요하다. REFERENCES 1) Miyake K, Ota I, Ichihashi S, et al. New classification of capsular block syndrome. J Cataract Refract Surg 1998;24:1230-4. 2) Heo JY, Ahn MD, Joo CK. Two cases of late postoperative capsular block syndrome. Korean J Ophthalmol 1999;13:105-9. 3) Rana M, Jiang L, Ilango B, Yang YC. Late-onset capsular block syndrome: unusually delayed presentation. Case Rep Ophthalmol 2013;4:299-302. 4) Kim HK, Shin JP. Capsular block syndrome after cataract surgery: clinical analysis and classification. J Cataract Refract Surg 2008;34:357-63. 5) Davison JA. Capsular bag distension after endophacoemulsification and posterior chamber intraocular lens implantation. J Cataract Refract Surg 1990;16:99-108. 6) Holtz SJ. Postoperative capsular bag distension. J Cataract Refract Surg 1992;18:310-7. 7) Choi BJ, Jin YB, Hur J. Postoperative capsular bag distension in cataract surgery. J Korean Ophthalmol Soc 1998;39:2031-7. 8) Lee SJ, Kim HY, Oum BS, Lee JE. Early capsular block syndrome after phacoemulsification with posterior chamber IOL insertion combined with vitrectomy. J Korean Ophthalmol Soc 2013;54:716-22. 9) Basti S, Nayak H, Mathur U. Capsular bag distension after optic captureof a sulcus-fixated intraocular lens. J Cataract Refract Surg 1999;25:293-5. 10) Geyer O, Goldstein M, Rothkoff L, Lazar M. Capsular bag distension associated with sulcus implantation of intraocular lenses. J Cataract Refract Surg 1998;24:1538-40. 11) Sugiura T, Miyauchi S, Eguchi S, et al. Analysis of liquid accumulated in the distended capsular bag in early postoperative capsular block syndrome. J Cataract Refract Surg 2000;26:420-5. 12) Sorenson AL, Holladay JT, Kim T, et al. Ultrasonographic measurement of induced myopia associated with capsular bag distention syndrome. Ophthalmology 2000;107:902-8. 13) Eifrig DE. Capsulorhexis-related lacteocrumenasia. J Cataract efractive Surg 1997;23:450-4. 14) Qu J, Bao Y, Li M, et al. Surgical management of late capsular block syndrome. J Cataract Refract Surg 2010;36:1687-91. 15) Neuhann IM, Werner L, Izak AM, et al. Late postoperative opacification of a hydrophilic acrylic (hydrogel) intraocular lens: a clinicopathological analysis of 106 explants. Ophthalmology 2004;111:2094-101. 592

- 안민원외 : 지연낭폐쇄증후군 1 예 - 16) Bompastor-Ramos P, Póvoa J, Lobo C, et al. Late postoperative opacification of a hydrophilic-hydrophobic acrylic intraocular lens. J Cataract Refract Surg 2016;42:1324-31. 17) Ramos JL, Li Y, Huang D. Clinical and research applications of anterior segment optical coherence tomography - a review. Clin Exp Ophthalmol 2009;37:81-9. 18) Kucukevcilioglu M, Hurmeric V, Erdurman FC, Ceylan OM. Imaging late capsular block syndrome: ultrasound biomicroscopy versus Scheimpflug camera. J Cataract Refract Surg 2011;37:2071-4. = 국문초록 = 인공수정체혼탁으로오인된지연낭폐쇄증후군 1 예 목적 : 인공수정체혼탁으로오인된지연낭폐쇄증후군 1 예에대한진단및치료경험을보고하고자한다. 증례요약 : 59 세남자가 1 년전부터진행된우안시력저하를주소로내원하였다. 고혈압외에는특이병력이없었으며, 10 년전타병원에서양안백내장수술을받았다. 안과개인의원에서우안인공수정체변성으로인한혼탁으로진단받고인공수정체교체를위해본원으로전원되었다. 내원당시나안시력은 0.06 이며, 안압은 22 mmhg, 굴절력은인공수정체부위의혼탁때문에측정되지않았다. 세극등현미경검사에서는인공수정체의부위가우유빛변성으로관찰되었다. 전안부빛간섭광학단층촬영에서인공수정체뒤쪽고반사물질의저류가관찰되었고, 초음파생체현미경검사에서는수정체후낭의팽창이확인되었다. Neodymium-doped yttrium aluminium garnet (Nd:YAG) 레이저후낭절개술을시행한후합병증발생없이나안시력이 1.0 으로호전되었다. 결론 : 낭폐쇄증후군은백내장수술후 1 일에서 2 주사이에드물게발생하는합병증인데, 술후 10 년이지나서도발생하는지연낭폐쇄증후군은전형적인임상양상과달리나타나기에인공수정체의변성으로인한혼탁과감별이매우중요하다. < 대한안과학회지 2018;59(6):589-593> 593