대한안과학회지 2015 년제 56 권제 4 호 J Korean Ophthalmol Soc 2015;56(4):643-649 ISSN 0378-6471 (Print) ISSN 2092-9374 (Online) http://dx.doi.org/10.3341/jkos.2015.56.4.643 Case Report 푹스홍채이색섬모체염환자에서방수유출장치삽입술후발생한각막부전 Corneal Endothelial Cell Loss after Tube Shunt Surgery in Fuch's Heterochromic Iridocyclitis 이진아 조양경 나태윤 최진아 Jin Ah Lee, MD, Yang Kyung Cho, MD, PhD, Tae Yoon La, MD, PhD, Jin A Choi, MD, PhD 가톨릭대학교의과대학성빈센트병원안과및시과학교실 Department of Ophthalmology and Visual Science, St. Vincent's Hospital, The Catholic University of Korea College of Medicine, Suwon, Korea Purpose: To report a case of corneal failure after implantation of the Ahmed glaucoma valve occurring in a patient diagnosed with Fuchs heterochromic iridocyclitis. Case summary: A 53-year-old male who complained of ocular pain and suddenly decreased visual acuity in his right eye visited our clinic. His visual acuity was 0.15 and intraocular pressure (IOP) was 55 mm Hg. The slit-lamp examination revealed edematous cornea, fine round or stellate keratic precipitates connected with fine filaments on the endothelium and depigmentation of the iris. The corneal endothelial cell density was 2,958 cells/mm 2. There was no specific finding in his left eye. The IOP did not improve with medical treatment, therefore, an Ahmed glaucoma valve was implanted in his right eye. At every follow-up exam the tube was well positioned and the IOP was maintained between 8 and 13 mm Hg. Eight months postoperatively, the patient complained of decreased visual acuity and the cell density was decreased to 1,408 cells/mm 2. Posterior subcapsular cataract opacity was observed as well as progression of depigmentation and distortion of the iris. Seventeen months after the surgery, the cell density was 700 cells/mm 2. On follow-up examination, his visual acuity was decreased to FC10 cm with the cataract progressing, therefore cataract surgery was performed. One month postoperatively, his vision improved to 0.1. However, the visual acuity deteriorated due to progression of the corneal edema and penetrating keratoplasty was performed. Conclusions: Aggravation of the corneal complication after Ahmed glaucoma valve implantation should be considered in patients with Fuchs heterochromic iridocyclitis-induced glaucoma. J Korean Ophthalmol Soc 2015;56(4):643-649 Key Words: Ahmed glaucoma valve surgery, Corneal endothelial cell density, Corneal failure, Fuchs heterochromic iridocyclitis, Uveitic glaucoma Received: 2014. 9. 27. Revised: 2014. 12. 30. Accepted: 2015. 3. 24. Address reprint requests to Jin A Choi, MD, PhD Department of Ophthalmology, College of Medicine, The Catholic University of Korea, St. Vincent's Hospital, #93 Jungbu-daero, Paldal-gu, Suwon 442-723, Korea Tel: 82-31-249-7343, Fax: 82-31-249-7340 E-mail: jinah616@hanmail.net * This study was presented as a poster at the 112th Annual Meeting of the Korean Ophthalmological Society 2014. 포도막염환자의 20-40% 에서안압상승이동반된다고알려졌으며, 1,2 포도막염에의한속발성녹내장은기존의섬유주절제술로잘반응하지않는다. 3,4 이에대한대안으로많은경우에서방수유출장치삽입술이시행되고있고, 수술후안압조절효과가기존섬유주절제술에비해더좋은것으로알려졌다. 5-7 하지만방수유출장치삽입술시행후, 연간 12% 에서합병증이발생하고, 8 저안압증, 좁은전방, 전방출혈, 백내장, 안내염, 만성적인각막내피세포손 c2015 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. 643
- 대한안과학회지 2015 년제 56 권제 4 호 - 상과이로인한각막부전등이발생할수있다. 9 특히각막관련합병증이 8-29% 의환자에서일어나고, 10 수술후 1년째 15.3%, 수술후 2년째 18.6% 의각막내피세포감소를보인다. 9 이러한각막합병증은환자가수술전에각막질환을가지고있거나각막이식을받았던경우에더흔히발생하는것으로보고되고있고, 그외에도이전수술력, 밸브와각막의접촉또는거리, 이물반응에의한손상, 전방의염증, 수술전높은안압이유지된기간, 안약보존제의독성등다양한원인이있다. 8,11-18 푹스홍채이색섬모체염 (Fuchs heterochromic iridocyclitis, FHIC) 은전체포도막염환자의 3% 를차지하는만성적인염증성질환으로경한염증과전형적인각막후면침착물, 홍채탈색을보인다. 19 FHIC의진행은녹내장과백내장의발병과관계가있는데, 20 FHIC 속발녹내장은영구적이고일반적으로약물치료에반응하지않아수술을요하는경우가많다. 21 FHIC 속발녹내장환자에서방수유출장치삽입후각막내피세포에대한영향에대해서는아직연구가되어있지않은상태이나수술로인한스트레스에취약할가능성이있다는보고가있다. 22 저자들은 FHIC로인한속발성녹내장환자에서방수유출장치삽입술후홍채의변형및탈색, 후낭하혼탁이진 행되고기존보고들보다심하게각막내피세포가 70-80% 감소한 1예를경험하여이를보고하고자한다. 증례보고 53세남자환자가 1주전부터우안의시력저하와불편감을주소로본원에내원하였다. 우안나안시력은 0.15, 좌안나안시력은 0.8이었고우안의안압이 55 mmhg 로상승되어있었다. 세극등현미경검사상우안의결막은충혈되어있었고각막부종및미세수포가관찰되었다. 각막후면에수개의별모양의각막후면침착물 (stellate keratic precipitate) 이있었고이들은서로가늘게연결되어있는양상을보였다 (Fig. 1A). 전방은깊었으나각막미세수포로인하여뿌옇게보여세포가관찰되지않았다. 홍채는아래쪽이경하게탈색되어있었고후유착 (posterior synechiae) 은관찰되지않았으며전방각검사상매우증가된섬유주색소침착이관찰되었으나, 주변홍채전유착 (peripheral anterior synechiae) 은관찰되지않았다 (Fig. 1B). 안저검사에서는시신경부종이관찰되었다 (Fig. 1C). 좌안의각막과홍채에서는특이소견이없었다. 우안의각막내피세포는 2,958 개 /mm 2, 좌안의각막내피세포는 2,364개 /mm 2 로정상소견 A B C Figure 1. Preoperative images: The slit lamp exam shows (A) fine round or stellate keratic precipitates and fine filaments on the endothelium between the keratic precipitates (B) patchy loss of the iris pigment epithelium. (C) The funduscopy shows swollen optic disc. 644
- 이진아외 : 푹스홍채이색섬모체염에서의각막부전 - A B C Figure 2. After the Ahmed valve implantation: (A) the valve tip was well positioned at superotemporal side (POD 1 day) (B) the slit lamp shows depigmented atrophic iris and posterior synechiae (POD 8 months) (C) before PPKP: Severely edematous cornea and corneal haziness are shown. POD = Postop date; PPKP = penetrating keratoplasty. 을보였다. 이를바탕으로포도막염속발녹내장, 푹스홍채이색섬모체염진단하에최대약물치료로안압조절이되지않아아메드밸브삽입술을시행하였다. Ahmed glaucoma valve (FP-7, New World Medical, Rancho Cucamonga, CA, USA) 는구후마취후에상이측에삽입하였다. 상이측결막을원개기저로절개한후 ahmed glaucoma valve를공막에 5-0 Ethibond로고정하고, 23G 바늘을이용하여윤부에서 1 mm 후방에전방천자를시행한후실리콘관을홍채와평행한방향으로삽입하였다. 실리콘관을덮기위해 3.5 3.5 mm 크기의공막편을이식하였다. 수술후 moxifloxacin (Vigamox, Alcon, Fort Worth, Texas, USA) 와 dexamethasone (Maxidex, Alcon, Fort Worth, Texas, USA) 점안약을 1일 6회점안으로시작하여염증에따라증감하였다. 수술후 1일째우안시력은 0.32였고안압은 18 mmhg이었다. 튜브는상이측에잘위치해있었고세포는 1+ 로관찰되었다 (Fig. 2A). 수술후 1달째시력은 0.6까지호전되었고안압은 8 mmhg 이었다. 그러나여전히전방세포는 1+ 로관찰되었고후유착 (posterior synechiae) 이보였다. 유리체의염증소견은관찰되지않았다. 이후시력은 0.63-0.8, 안압 8-13 mmhg 로유지되었고전방내염증도변화를보이지않았다. 수술후 8개월째갑자기우안나안시력이 0.32로저하되었고안압은 8 mmhg, 각막내피세포밀도는 1,408개 /mm 2 로확인되었다. 전방염증은 1+ 였고, 홍채탈색및변형이하이측방향에서진행되었고, 후피막하수정체혼탁 (posterior subcapsular opacity) 이심해진양상이었다 (Fig. 2B). 안저검사상이상소견은없었다. 수술후 17개월째시력은안전수동으로저하되었고, 백내장이더욱진행한양상및전방염증 2+, 심한후유착을보였다. 우안의각막내피세포는 638개 /mm 2 로더감소한소견을보였다. 아메드밸브삽입술후 35개월째, 우안백내장수술을시행하였다. 백내장수술은 temporal clear corneal incision으로초음파유화술을시행한후, 인공수정체는접힘형연성인공수정체 (YA60BBR, Hoya Co., Tokyo, Japan) 를삽입하였다. 수술후 moxifloxacin (Vigamox, Alcon, Fort Worth, Texas, USA) 과 dexamethasone (Maxidex, Alcon, Fort Worth, Texas, USA) 점안약을 1일 4회점안으로시작하여경과에 645
- 대한안과학회지 2015 년제 56 권제 4 호 - 따라횟수를증감하였다. 백내장수술시행후 3주째시력은 0.1까지호전되었고안압은 7-8 mmhg 로유지되었다. 백내장수술후 3개월째시력이다시 FC50 cm까지감소되어있었고각막의부종및혼탁이관찰되었다 (Fig. 2C). 이에백내장수술후 6개월째전층각막이식 (penetrating keratoplasty) 을시행하였다. 각막이식은 7.5 mm 바론공여각막천공기 (Katena Products Inc.) 를이용하여공여각막을절제하고수여각막을 7.0 mm 헤스버그바론진공각막원형절제기 (Katena Products Inc., Denvile, New Jersey, USA) 를이용하여떼어낸후, 10-0 나일론으로 16 방향에서단순봉합을시행하여공여각막을고정하였다. 수술후 Moxifloxacin (Vigamox, Alcon, Fort Worth, Texas, USA) 과 Dexamethasone (Maxidex, Alcon, Fort Worth, Texas, USA) 점안약을 1일 6회점안으로시작하여경과에따라횟수를증감하였다. 수술후교정시력은 0.08, 안압은 7 mmhg이며전방염증은관찰되지않은상태이다. 고찰 포도막염속발녹내장은젊은환자에게서많이생기고발병기전이다양하기때문에치료가어려워결국수술적치료까지요하는경우가많다. 23 기존에는섬유주절제술을일반적으로시행하였으나난치성녹내장의경우방수유출장치삽입술이흔히시행되며, 속발성녹내장환자에게방수유출장치삽입술을시행하고 1년경과관찰한결과 94% 에서성공률을보이고, 6 수술후 wound leaking의합병증도기존의섬유주절제술보다는적다. 24 하지만저안압증, 얇은전방, 전방출혈, 튜브와각막의접촉, 백내장, 안내염, 각막내피세포감소그리고각막부전등의많은합병증이발생할수있다. 25 이중각막관련합병증이 8-29% 의환자에서일어나는데, 10 아메드밸브수술후각막내피세포의평균소실률은 1달째 5.8%, 6달째 11.5%, 12달째 15.3%, 18개월째 16.6%, 24개월에 18.6% 라는보고가있다. 9 또다른논문에서는아메드밸브수술후 12개월째평균 14.8% 의각막내피세포감소율을보였다. 13 각막내피세포감소를일으키는원인은아직명확히밝혀진바는없지만이유를설명하려는다양한이론들이있다. 튜브주변의 jet flow로인한손상, 수술후눈을깜박거리거나비비는경우등간헐적인각막 튜브의접촉으로인한기계적인손상, 튜브와포도막의접촉으로인한전방에서의염증, 그리고실리콘튜브에대한이물반응으로인한손상, 실리콘관의첨단과각막사이의거리, 안약점안시안약의보존제의독성으로인한손상, 수술전높은안압과그상승기간으로인한각막내피세포의직접적인압박또는저산소증으로인한손상, 기 존의각막질환및각막이식수술력, 수술후얕은전방등이제시되고있다. 8,11-18 이번증례에서도물론여러가지원인이각막내피세포감소에작용하였을것이다. 하지만이번증례는 2년내에 70-80% 의각막내피세포감소를보여 24개월에 18.6% 의소실률을보였다는것보다훨씬큰소실률을보였다. 이에위에언급되었던일반적인요인외에다른원인이추가로작용했을가능성이있을것으로보인다. FHIC는전체포도막염환자의 3% 를차지하는만성적인염증성질환으로다음과같은임상적인양상을근거로진단을내린다. 26,27 1) 경하지만지속적인앞포도막염 (chronic low grade, mainly anterior uveitis), 2) 급성악화의부재 (absence of significant acute exacerbations), 3) 미만성의별모양각막후면침전물 (diffusely spread stellate keratic precipitates), 4) 홍채탈색소및양안의불균형적인홍채색깔 (diffuse iris atrophy with or without obvious heterochromia), 5) 홍채후유착의부재 (lack of posterior synechiae) 가현재널리사용되고있는진단기준이다. 28 하지만 Yang et al 19 에따르면모든진단기준을다부합하지않아도주요임상양상에따라진단내릴수있고인종간에도 FHIC의특징에차이가있다고한다. 본증례의환자는위소견들을대부분만족하였다. 초진시에급성악화로보이는것은당시전방염증이경한것으로보아, 전방염증의급성악화로인한소견이아닌안압상승으로인한소견으로판단된다. 또한녹내장수술후지속적으로관찰된경한전방염증, 전형적인별모양각막후면침착물이전반적으로분포되어있는것, 아메드밸브위치와먼부분의전반적인홍채위축및탈색그리고후낭하혼탁타입 (posterior subcapsular opacity) 의백내장을보였고, 반대눈의홍채와각막에는특이소견이없었다. 이는 Yang et al 19 논문에서보였던것과같은동양인의 FHIC 특징을보인것이다. FHIC의진행은녹내장과백내장의발병과관계가있는데, 20 만성적인염증과스테로이드제를이용한치료가이러한속발성녹내장발생에영향을주는중요한요인이다. 29 FHIC 환자에서속발성녹내장의유발률은 6.5-59.0% 로, 29 이렇게유병률의범위가넓은것은각연구마다녹내장에대한정의가다르기때문에발생한것으로보이며, 30 일반적으로녹내장발생률이약 20% 인것으로알려졌다. 31 FHIC 속발녹내장은영구적이고일반적으로약물치료에반응하지않는다. 21 FHIC로인해발생한백내장은처음에는후낭하혼탁으로나타나서점점전층에걸친혼탁 (total opacification) 으로진행한다. 80% 이상의환자에게서백내장이나타나며많은경우가진단시관찰된다. 29 본증례에서는아메드밸브삽입후, 지속적인전방염증, 각막 튜브접촉 (cornea-tube touch) 이없고눈을비비는습 646
- 이진아외 : 푹스홍채이색섬모체염에서의각막부전 - A-1 A-2 B C D Figure 3. Remarkable changes of corneal endothelial cell density: (A) before Ahmed valve implantation (A-1: right eye, A-2: left eye) (B) POD 7 months after Ahmed valve implantation (C) POD 13 months after Ahmed valve implantation (D) before cataract surgery. POD = Postop date. 관또는저안압이없는상태에서심각한각막내피세포감소 (Fig. 3) 및술후 8개월경부터홍채의탈색소진행, 홍채모양변형 (iris distorsion) 및후낭하혼탁의진행양상을보였다. 이를통해 FHIC에의해백내장과녹내장이발생하였다고추정할수있었다. FHIC의원인은확실히알려져있지않다. 면역학적원인에의한염증외에 herpes simplex virus, toxoplasma gondii 등의감염도병의원인으로작용할것으로알려졌다. 32 최근에는 FHIC의임상양상을보이는환자의방수에서 cytomegalovirus (CMV) 가동정되는예가보고되고있으며, CMV는바이러스로인한앞포도막염의중요인자로대두되고있다. 33 FHIC에서내피세포가손상되어있다고알려졌으며, 22 CMV에의한포도막염은각막내피세포염 (corneal endothelitis) 을유발하는것으로알려졌다. 34 따라서 FHIC 647
- 대한안과학회지 2015 년제 56 권제 4 호 - 와 CMV와의연관성은 FHIC에서각막내피손상이가속화되는현상과관련이있다하겠다. FHIC 환자의각막내피세포는수술로인한스트레스에취약할가능성이있다는보고가있다. 22 실제로 FHIC 환자중백내장수술을받지않은경우, 5% 정도의각막내피세포감소율을보였으나백내장수술을받은경우에서는각막내피세포수가 45-50% 나감소한경우가보고된바있다. 22 그러나 FHIC 환자에서방수유출장치삽입후각막내피세포에대한영향에대해서는아직연구가되어있지않은상태이다. 이에본증례에서는방수유출장치삽입술후기존에알려진원인외에각막내피세포감소를가속화한원인이 FHIC라판단하였다. FHIC로인한속발성녹내장환자에서방수유출장치삽입술후각막내피세포가심각하게감소하였던 1예를경험하였기에이를보고하는바이다. FHIC를보이는환자에서방수유출장치삽입술시각막합병증이가속화될수있다는점을알고여러요인들을면밀히살펴봐야하겠다. REFERENCES 1) Panek WC, Holland GN, Lee DA, Christensen RE. Glaucoma in patients with uveitis. Br J Ophthalmol 1990;74:223-7. 2) Foster CS, Havrlikova K, Baltatzis S, et al. Secondary glaucoma in patients with juvenile rheumatoid arthritis-associated iridocyclitis. Acta Ophthalmol Scand 2000;78:576-9. 3) Allen RC, Bellows AR, Hutchinson BT, Murphy SD. Filtration surgery in the treatment of neovascular glaucoma. Ophthalmology 1982;89:1181-7. 4) Schwartz AL, Anderson DR. Trabecular surgery. 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- 이진아외 : 푹스홍채이색섬모체염에서의각막부전 - = 국문초록 = 푹스홍채이색섬모체염환자에서방수유출장치삽입술후발생한각막부전 목적 : 푹스홍채이색섬모체염으로인한속발녹내장환자에서방수유출장치삽입술후각막내피세포부전이온증례를보고하고자한다. 증례요약 : 53 세남자가우안통증및시력저하를주소로내원하였다. 시력은 0.15 이었고, 안압은 55 mmhg 이었다. 별모양의각막후면침착물이서로가늘게연결되어있었고각막부종과홍채의부분적탈색이있었다. 각막내피세포밀도는 2,958 개 /mm 2 였다. 좌안은특이소견은없었다. 약물치료로안압조절이안되어아메드밸브삽입술을시행하였다. 수술후튜브는잘위치해있었고안압은 8-13 mmhg 로유지되었으나, 수술후 8 개월째환자가시력저하를호소하였고후피막하수정체혼탁이심해지며, 홍채변형및탈색이진행되었다. 각막내피세포밀도는 1,408 개 /mm 2 로감소하였다. 수술후 17 개월째각막내피세포밀도 700 개 /mm 2 로더욱감소하였고, 경과관찰하다백내장이더욱진행되면서시력이 FC10 cm 까지저하되어백내장수술을하였다. 백내장수술후 1 달째시력이 0.1 이었으나각막부종이심해지면서다시시력이저하되어각막이식을시행하였다. 결론 : 푹스홍채이색섬모체염환자에서방수유출장치삽입술시각막합병증을가속화시킬수있어주의가필요하겠다. < 대한안과학회지 2015;56(4):643-649> 649