JOURNAL OF RETINA 2016;1(1): CASE REPORT ISSN 맥락막혈관병증에대한광역학치료후발생한광범위장액망막박리 Extensive Serous

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1 JOURNL OF RETIN 2016;1(1):46-50 CSE REPORT ISSN 맥락막혈관병증에대한광역학치료후발생한광범위장액망막박리 Extensive Serous Retinal Detachment after Photodynamic Therapy for Polypoidal Choroidal Vasculopathy 이택훈 1, 이상준 2, 남기엽 2 Taek Hoon Lee 1, Sang Joon Lee 2, Ki Yup Nam 2 1 대전이안과병원, 2 고신대학교의과대학안과학교실 1 Rhee's Eye Hospital, Daejeon, Korea 2 Department of Ophthalmology, Kosin University College of Medicine, usan, Korea Purpose: To report a case with extensive serous retinal detachment (SRD) after photodynamic therapy (PDT) for polypoidal choroidal vasculopathy (PCV) Case summary: 72 year old man visited our clinic complaining of visual disturbance in the left eye for 1 week. Visual acuity (V) was finger counting at 30 cm in the left eye. The fundus was invisible due to vitreous hemorrhage, and so vitrectomy was performed; atrophic scar on the posterior pole and extensive subretinal hemorrhage over the inferior retina were seen during surgery. One week after surgery, best corrected visual acuity was Three weeks after vitrectomy, large pigment epithelial detachment superior to the fovea and cystic lesions in the retina were seen on optical coherence tomography, and a branching vascular network and polyps were found on indocyanine green angiography. Thus, PDT was performed for PCV treatment. Two weeks after PDT, the patient complained of decreased vision and superior visual field defect. On fundus examination, extensive retinal detachment (RD) reaching below the fovea was found, but no retinal break or hole was found. Thus, intravitreal anti-vascular endothelial growth factor injection had been performed at 5 months. However, RD gradually progressed to fovea and subretinal fibrosis was developed. Surgical treatment with retinotomy, subretinal fluid drainage and fluid-gas exchange was performed. The retina was flattened after surgery and V recovered to 0.1. Conclusions: Transient SRD is often seen after PDT for age related macular degeneration or PCV, but extensive SRD is very rare. Surgical treatment may be another treatment option in such cases. Keywords: Polypoidal choroidal vasculopathy; Photodynamic therapy; Serous retinal detachment ddress reprint requests to Ki Yup Nam, MD Department of Ophthalmology, Kosin University Gospel Hospital, #262 Gamcheon-ro, Seo-gu, usan 49267, Korea Tel: , Fax: oksnam1231@daum.net Received: Revised: ccepted: Copyright 2016 The korean retina society This is an Open ccess article distributed under the terms of the Creative Commons ttribution Non-Commercial License ( which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

2 Lee TH, et al. PCV 에서 PDT 후발생한장액망막박리 서론 연령관련황반변성 (age related macular degeneration, MD) 에대한광역학치료 (photodynamic therapy, PDT) 의효과는 Verteporfin in Photodynmic Therapy Study (VIP), Treatment of ge Related Macular Degeneration with Photodynamic Therapy (TP) 연구등을통해이미널리알려져왔다 [1-3]. 또한결절맥락막혈관병증에대해서는 Ranibizumab과의비교연구인 efficacy and safety of verteporfin photodynamic therapy in combination with ranibizumab or alone versus ranibizumab monotherapy in patients with symptomatic macular polypoidal choroidal vasculopathy (EVEREST) 연구에서 PDT가결절을소실시키는데더효과가있음이밝혀졌다 [4]. PDT는광과민성을띠는 verteporfin을이용하는데, 정맥주사를통해신생혈관에도달하면내피세포의 low density lipid (LDL) 단백수용체와결합하게된다. 이부위에 689 nm의다이오드레이저를조사하면 verteporfin이활성화되는데이로인해활성산소와자유유리기가발생하고내피세포손상이일어나며혈관기저막노출및혈소판응집이뒤따라일어나혈관을폐쇄시킨다 [5]. 연령관련황반변성에대한 PDT의합병증은드문것으로알려져있다. VIP와 TP 연구환자들을대상으로 PDT의안전성에 대해서메타분석한연구에서는위약군과비교했을때전신적혹은안과적합병증의차이는없었다 [6]. 안과적으로가장흔한합병증은시력장애였는데대부분일시적이고경하였으며급성의심한시력저하는 % 에서나타났다. 심한시력저하의원인으로는망막하액의증가, 망막하출혈, 녹색의황반하병변등이확인되었는데망막하액증가의경우 3개월내에모든증례에서흡수되었다 [7]. 한편, Mennel et al. [8] 은연령관련황반변성에대한 PDT 후초기에일시적인장액망막박리가나타나며이는 PDT 후신생혈관누출증가및외측혈액망막장벽의파괴와관련이있다고하였다. 하지만지금까지연령관련황반변성에대한 PDT 후지속적인장액망막박리에대한보고는찾아보기어렵다. 저자들은결절맥락막병증에대한 PDT 후초기에광범위한장액망막박리가발생하고지속적으로유지되어수술적치료로망막을재유착시킨증례를경험하여이를보고하고자한다. 증례보고 72세남자환자가좌안의시력저하를주소로내원하였다. 내원당시시력은양안모두안전수지 30 cm로측정되었으며교정되지않았다. 과거력상약 5년전양안의연령관련황반변성및개 C Figure 1. Examinations 3 weeks after initial vitrectomy for breakthrough vitreous hemorrhage. () trophic lesions are over the posterior pole area and large retinal pigment epithelium detachment (PED) superior to fovea is also found on fundus examination. () Optical coherence tomography shows intraretinal cysts and large PED. (C) Indocyanine green angiography shows branching retinal vascular network and polyps. 47

3 JOURNL OF RETIN Figure 2. Fundus examination and optical coherence tomography (OCT) 2 weeks after photodynamic therapy. () On fundus examination, retina below the fovea seems to be slightly opaque and choroidal vessels are invisible. () OCT also shows inferior retinal detachment reaching the fovea. 방각녹내장으로본원에서진료받았으며당시최대교정시력은우안안저수지 30 cm, 좌안 0.25였다. 당시우안안저검사및빛간섭단층촬영 (optical coherence tomography, OCT) 에서는위축성반흔및큰망막내낭포로인한망막비후를보였으며좌안에서는후극부의광범위한망막색소상피박리 (pigment epitheli-소 al detachment, PED) 가관찰되었다. 인도시아닌그린혈관조영술 (indocyanine green angiography, ICG) 에서선형혈관및결절이관찰되어결절맥락막혈관병증 (polypoidal choroidal vascu - lopahty, PCV) 으로판단되어좌안에 anti-vascular endothelial growth factor (VEGF) 주입술을시행하며정기적인진료를시행하던중환자사정으로 2년간내원하지않았다. 당뇨병및고혈압과거력은없었으며특별한외상력도없었다. 전안부검사에서중등도의핵백내장이외에특이소견은보이지않았고안저검사에서는우안은초진시와큰차이를보이지않았으나좌안에는유리체출혈이관찰되고이로인해안저를확인할수없었다. 이에결절맥락막혈관병증과연관된유리체출혈 (breakthrough vitreous hemorrhage) 이라고판단한후유리체절제술, 수정체초음파유화술, 인공수정체삽입술, 유리체강내 bevacizumab 주입술을시행하였다. 수술중후극부의위축성병변및하부의광범위한망막하출혈을관찰할수있었으며특별한문제는발생하지않았다. 수술 2주후좌안의최대교정시력은 0.075였으며환자는유리체출혈발생전의시력과비슷하다고하였다. 수술 4주후시행한플루레신형광안저혈관조영술 (fluorescein angiography, F) 및 ICG에서방사상으로가지를치는선형혈관및결절, 이상혈관으로부터의누출이관찰되고 OCT에서황반부위쪽에큰망막색소상피박리및황반부의망막내낭성병변이관찰되었다 (Fig. 1). 이에결절맥락막혈관병증에대한치료로서 PDT 를시행하였다. 체표면적 (m 2 ) 당 6 mg의 verteporfin (Visudyne, Novartis G, asel, Switzerland) 을 10분간에걸쳐정맥주사하였고, 5분후에다이오드레이저를조사하였다. 결절과선형혈관을포함하여지름 4,500 μm의원영역에시행하였으며표준치료세기인 50 J, 600 mw의세기로 83초간조사하였다. PDT 1주후환자가좌안의시력이저하된것같다고호소하였다. 교정시력은 0.05였으며전안부에는특별한소견은보이지않았다. 안저검사에서큰변화는확인할수없었고 OCT에서는황반부위쪽 PED 근처로망막하액이고여있는것을확인할수있었다. PDT 후일시적인장액성망막하액은드물지않은소견이므로 1개월후다시경과관찰하기로하였으나시술 2주째환자가좌안상측시야가점점좁아진다고하여다시내원하였다. 안저검사에서하부의망막박리가황반부바로아래까지침범하여있는것이확인되었고이는황반부상측의 PED로부터황반부이측의망막하경로를통해연결되어있었다 (Fig. 2). 망막주변부에는열공은관찰되지않았다. 안저검사를누운자세와앉은자세에서시행한결과망막하액의이동을확인할수있었다. 이에장액망막박리로추정하고항혈관내피성장인자를유리체강내로주사하고경과관찰하였다. 1개월간격으로 bevacizumab 2회, aflibercept 2회주사후에도망막박리는호전되지않았다. 비록망막박리가황반부를침범하지는않았고시력은 0.1로유지되었으나망막박리가점차황반부로진행하는소견을보이고망막주름및망막하섬유성띠가발생하여 PDT 후 5개월째결국수술을결정하였다. 시신경비측에망막절개를하고망막하액을배액한후 C3F8 (18%) 을충전하였다. 수술중에도주변부망막의열공은발견하지못했다. 수술후 3개월째좌안안저는편평하게유지되었고최대교정시력은 0.1로회복되었다 (Fig. 3). 48

4 Lee TH, et al. PCV 에서 PDT 후발생한장액망막박리 Figure 3. Fundus examination () and optical coherence tomography () 3 months after reoperation. Retina is reattached well on fundus examination and optical coherence tomography. 고찰 기존의문헌들을살펴보면 MD 환자에서 PDT를시행한후초기에일시적인후극부의장액망막박리는드물지않은것으로여겨진다. Mennel et al. [8] 은 10명의전형맥락막신생혈관및잠복맥락막신생혈관환자에대하여 PDT를시행하고 2일, 7일째 OCT를시행하였다. 그결과 2일째모든환자에서후극부의장액망막박리가확인되었고중심망막두께가평균 626 ± 157 μm 로서시술전 217 ± 42 μm에비해증가하였으나 7일째에는망막하액이흡수되고중심망막두께가다시 240 ± 36 μm로감소하는경향을보였다. PCV에서도 PDT 후발생한장액성망막박리가발생한증례가보고된바있다. Tobita et al. [9] 은 PDT 후 7일째광범위한망막박리가발견되어테논낭하트리암시놀론주입술을시행하고 8주째망막하액이거의흡수된증례를보고하였다. Prakash and Han [10] 은 PCV에대한 PDT 후 2일째시력저하및하부의광범위한망막박리가발생한증례를보고하였다. 열공망막박리로의심하고공막돌륭술을시행하였으나수술중열공은발견할수없었다고하였다. 수술 3주후망막하액이흡수되고시력은회복되었으나 4개월후 PCV 재발로다시 PDT를시행하였고 2일후또망막박리가발생하였지만 2주째자연흡수되었다고보고하였다. PDT 후발생하는장액망막박리의기전은완전히밝혀지지는않았으나몇가지설명이가능하다. PDT에의해서신생혈관으로부터누출이일시적으로증가하거나망막색소상피의손상으로인해혈액망막장벽이파괴되어발생할수있다 [8]. 망막색소상피펌프기능저하및맥락막혈관투과성의증가나레이저를조사받은맥락막혈관에서의혈관내피성장인자증가도가능한기전으로설명된다 [10]. 본증례에서는기존보고들과비교하여몇가지차이점이있 다. 유리체절제술을시행한상태였고광범위한망막하출혈이 동반되어있었다. 유리체가제거된상태이므로유리체의망막 에대한압력이소실되고이미존재하고있던망막하출혈이망 막의유착력을저하시켜망막의박리를용이하게한것으로추 정해볼수있다. 따라서본증례에서는환자의유리체망막상태 로인해장액망막박리가기존의증례들에비해더광범위하게 발생하고장기적으로유지되었다고판단된다. PDT 후광범위한망막박리로인해유리체강내로접근하여망 막하액배액을통한망막유착술을시행한경우는지금까지보 고된적이없다. 이에보고하는바이며 PDT 를시행할때유리 체절제술후상태혹은광범위한망막하출혈이있는경우에는 삼출망막박리에대한주의를기울일필요가있다고생각한다. Conflicts of interest There are no conflicts of interest. References 1. Kaiser PK; Treatment of ge-related Macular Degeneration with Photodynamic Therapy (TP) Study Group. Verteporfin therapy of subfoveal choroidal neovascularization in age-related macular degeneration: 5-year results of two randomized clinical trials with an open-label extension: TP report no. 8. Graefes rch Clin Exp Ophthalmol 2006;244: ressler NM; Treatment of ge-related Macular Degeneration with Photodynamic Therapy (TP) Study Group. Photodynamic therapy of subfoveal choroidal neovascularization in age-related macular degeneration with verteporfin: two-year results 49

5 JOURNL OF RETIN of 2 randomized clinical trials-tap report 2. rch Ophthalmol 2001;119: ressler NM. Verteporfin therapy of subfoveal choroidal neovascularization in age-related macular degeneration: two-year results of a randomized clinical trial including lesions with occult with no classic choroidal neovascularization-verteporfin in photodynamic therapy report 2. m J Ophthalmol 2002;133: Koh, Lee WK, Chen LJ, et al. EVEREST study: efficacy and safety of verteporfin photodynamic therapy in combination with ranibizumab or alone versus ranibizumab monotherapy in patients with symptomatic macular polypoidal choroidal vasculopathy. Retina 2012;32: Kramer M, Miller JW, Michaud N, et al. Liposomal benzoporphyrin derivative verteporfin photodynamic therapy. Selective treatment of choroidal neovascularization in monkeys. Ophthalmology 1996;103: zab M, enchaboune M, linder KJ, et al. Verteporfin therapy of subfoveal choroidal neovascularization in age-related macular degeneration: meta-analysis of 2-year safety results in three randomized clinical trials: Treatment Of ge-related Macular Degeneration With Photodynamic Therapy and Verteporfin In Photodynamic Therapy Study Report no. 4. Retina 2004;24: rnold JJ, linder KJ, ressler NM, et al. cute severe visual acuity decrease after photodynamic therapy with verteporfin: case reports from randomized clinical trials-tp and VIP report no. 3. m J Ophthalmol 2004;137: Mennel S, Meyer CH, Eggarter F, Peter S. Transient serous retinal detachment in classic and occult choroidal neovascularization after photodynamic therapy. m J Ophthalmol 2005;140: Tobita H, Ohno-Matsui K, Ogawa M, et al. Transient serous retinal detachment after photodynamic therapy for polypoidal choroidal vasculopathy. Jpn J Ophthalmol 2008;52: Prakash M, Han DP. Recurrent bullous retinal detachments from photodynamic therapy for idiopathic polypoidal choroidal vasculopathy. m J Ophthalmol 2006;142: 국문초록 맥락막혈관병증에대한광역학치료후발생한광범위장액망막박리 목적 : 결절맥락막혈관병증에대한광역학치료후광범위한장액망막박리가발생하여수술로망막을재유착시킨증례를보고하고자한다. 증례요약 : 72세남자가좌안의유리체출혈로유리체절제술을시행받았고수술중후극부의위축성반흔및하부에광범위한망막하출혈이확인되었다. 시력은수술전안전수지 30 cm에서 로호전되었다. 3주후빛간섭단층촬영에서황반부상측의큰망막색소상피박리와망막의낭포성병변이관찰되고인도시아닌그린혈관조영에서선형혈관및결절이확인되어결절맥락막혈관병증으로판단하고광역학치료를시행하였다. 2주후상부시야결손및시력저하를호소하였고안저검사에서황반하부까지이르는광범위한망막박리가발견되었으나망막열공은보이지않았다. 이에장액망막박리로판단하고항혈관내피성장인자를주사하면서관찰하였다. 하지만망막박리가점차황반부로진행하여 5개월째망막절개및망막하액배액, 가스주입을포함한수술적치료를시행하였다. 이후망막은유착되었으며교정시력은 0.1 로회복되었다. 결론 : 결절맥락막혈관병증에서광역학치료후일시적인장액망막박리는종종발생하지만광범위한장액망막박리는드물며이경우수술적치료가도움이될수있다. 50

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