대한안과학회지 2018 년제 59 권제 5 호 J Korean Ophthalmol Soc 2018;59(5):491-495 ISSN 0378-6471 (Print) ISSN 2092-9374 (Online) https://doi.org/10.3341/jkos.2018.59.5.491 Case Report 복막결핵환자에서발생한합병증을유발한홍채낭종의자발적회복 1 예 Case of Spontaneous Recovery of an Iris Cyst in a Patient with Peritoneal Tuberculosis 윤요셉 이승환 민정기 이창규 Yo Sep Yoon, MD, Seunghwan Lee, MD, Jung Kee Min, MD, Chang Kyu Lee, MD 울산대학교의과대학울산대학교병원안과학교실 Department of Ophthalmology, Ulsan University Hospital, University of Ulsan College of Medicine, Ulsan, Korea Purpose: We report a case of spontaneous recovery of an iris cyst with only tuberculosis medication and conservative eye drops when uveitis and angle closure occurred because of a cyst in a patient with peritoneal tuberculosis. Case summary: 49-year-old female who was diagnosed with iritis and treated with steroid eye drops visited our clinic because of decreased visual acuity 1 month prior. There were anterior chamber inflammation cells and an iris cyst completely obstructing the anterior chamber at 12 o'clock. t the time, the patient had been diagnosed with peritoneal tuberculosis in the Department of Internal Medicine and Gynecology and had been treated with surgery and medication. The patient had no past history of glaucoma, but when the iris cyst developed, the intraocular pressure increased to 29 mmhg and anterior inflammatory cells were seen in the range of +1 to +2. The primary lesion of tuberculosis improved and the iris cyst disappeared with treatments involving medication for tuberculosis, steroid eye drops, and glaucoma eye drops, without invasive treatments such as alcohol curettage, laser treatment, or cyst resection. Conclusions: If an iris cyst is a new lesion of the eye, it is necessary to identify the pattern and cause of the iris cyst first, and if a secondary benign iris cyst is suspected, the primary treatment of the causative disease is necessary rather than prompt invasive treatment. J Korean Ophthalmol Soc 2018;59(5):491-495 Keywords: Glaucoma, Iris cyst, Tuberculosis 홍채종양은양상에따라낭성또는고체병변으로크게분류되며낭성병변은위치에따라홍채색소상피낭종과실질낭종으로분류할수있다. 1-3 일반적으로홍채색소상피낭 Received: 2017. 12. 21. Revised: 2018. 2. 11. ccepted: 2018. 4. 19. ddress reprint requests to Chang Kyu Lee, MD Department of Ophthalmology, Ulsan University Hospital, #877 angeojinsunhwando-ro, Dong-gu, Ulsan 44033, Korea Tel: 82-52-250-8757, Fax: 82-52-250-7174 E-mail: coolleo7@naver.com * Conflicts of Interest: The authors have no conflicts to disclose. 종은별다른합병증을야기하지않으므로무의미하게남아있는경우가많지만홍채실질낭종의경우위치, 크기, 합병증정도에따라낭종에대한적극적인시술이필요한경우가많다. 4 홍채낭종은원인에따라일차성및이차성으로분류한다. 특별한원인없이발생한경우를일차성홍채낭종이라하고, 외상, 수술, 약물, 종양, 결핵등의원인으로발생한것이이차성홍채낭종이다. 2-6 결핵은활동성결핵환자의결핵균에의해감염되어발생하는질환으로공기를매개로감염된다. 따라서폐에잘 c2018 The Korean Ophthalmological Society This is an Open ccess article distributed under the terms of the Creative Commons ttribution 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. 491
- 대한안과학회지 2018 년제 59 권제 5 호 - 생기며, 폐이외에도흉막, 림프절, 척추, 뇌, 신장, 위장관등다양한장기들을침범할수있고안구에도결핵이침범하여문제를일으킬수있다. 7,8 본증례에서는복막결핵외에평소다른전신적또는안과적과거력이없는환자에서홍채실질낭종이발생하였지만외과적결핵병변절제, 내과적결핵치료및안과적대증치료만으로홍채낭종이자발적으로회복되어이를보고하고자한다. 증례보고 49세여자환자가한달간지속된우안시력감소를주소로본원으로의뢰되었다. 환자는 1개월전부터포도막염의심하에개인병원안과에서 1% prednisolone acetate eye drop (PredForte, llergan, Inc., Irvine, C, US) 하루 3 회점안중이었다. 본원안과처음방문당시나안시력우안 8/20, 좌안 10/20으로측정되었고교정시력은양안 20/20으로측정되었으며, 안압은골드만압평안압계로우안 20 mmhg, 좌안 17 mmhg로측정되었다. 세극등현미경검사상우안각막에표층점상각막염소견이있었고전방내 염증세포 +1~+2 소견을보여스테로이드안약을지속적으로투여하였다. 과거력상고혈압, 당뇨등의특별한질환은없었고흉부엑스레이검사상폐에는이상소견이보이지않았다. 하지만본원안과내원 6개월전부터복부팽만감이있었고 2개월전에는 15일이상생리가지속되어, 본원안과에처음방문할당시산부인과에도방문하여초음파검사, 복수천자등산부인과기초검사를실시한결과복막결핵의증, 난소암의증으로배전체자궁절제술, 난소절제술, 복막유착박리술을시행하게되었다. 이때시행한조직검사와 cid-fast acilli stain에서복막결핵으로진단되었고 (Fig. 1), 감염내과와협진하에결핵약물치료를하였다. 본원산부인과에서실시한수술 2주후안과추적검사에서초기세극등현미경검사에서보이지않던우안전방 11 시방향에서 1시방향으로홍채실질낭종이관찰되었다 (Fig. 2). 이때안압은골드만압평안압계로우안 29 mmhg, 좌안 17 mmhg로측정되었고. 세극등현미경검사상전방내염증세포 +2 소견을보였고안저검사상이상소견은없어복막결핵과이를제거하는복막수술로인한결핵균파종으로추정되는전방포도막염과동반된이차성결핵성홍채실질낭종의증으로의한안압상승으로진단후전신적으로는항결핵제 Isoniazid + Rifampin + Pyrazinamide + Ethambutol 치료를지속하였고안압과염증조절을위해우안 Fluorometholone eye drop (Fluorometholon, Santen, Osaka, Japan) 4회, 사이클로스포린점안액 0.05% ( 싸이포린엔 0.05%, Taejoon, Seoul, Korea) 2회, brimonidine tartrate/timolol maleate ophthalmic solution 0.2%/0.5% (COMIGN, llergan, Irvine, C, US) 를 2회점안하였다. 홍채실질낭종에대한적극적인중 Figure 1. Histopathology of biopsy () and computed tomography image of abdomen and pelvis with enhancement (). () Granulomatous inflammation with caseation necrosis is observed in the biopsy material taken from peritoneum (in the circle) (hematoxylin and eosin [H&E] stain 100). () Multiple irregular abscess-like cavities (in the circle) existed in uterine corpus, which indicate bilateral tuberculosis salpingitis with tuberculosis peritonitis and myositis. Figure 2. First slit lamp photography of the iris cyst. Cystic type of iris mass (circle) was shown at superior portion of anterial chamber and some iris pigmentation was also shown in front of the lens (arrows). 492
- 윤요셉외 : 안구결핵성홍채낭종의자발적회복 - Figure 3. One month later pictures. Follow up slit lamp photography () and gonioscope photography (). The iris cyst (in the circle) was slightly decreased compare to the initial finding of the slit lamp examination. 재적시술없이안약치료만으로 1달경과후크기가감소하는것을확인할수있었으며 (Fig. 3), 안압은골드만압평안압계로양안 16 mmhg로안정적으로유지되는것을관찰할수있었다. 본원안과에서치료 2달경과후홍채실질낭종은거의사라지고홍채침착으로흔적만남아있는모습을확인하였고 (Fig. 4), 나안시력우안 20/20, 좌안 18/20 으로회복하였으며안압도골드만압평안압계로우안 13 mmhg, 좌안 19 mmhg로정상범위소견을보였다. 완치후 6개월간복막결핵과홍채낭종은재발하지않고안정적으로유지되고있다. 고 찰 홍채는조직학적으로전경계층, 홍채실질, 상피층으로구분된다. 전경계층은결체조직과홍채간질의멜라닌세포가치밀하게농축되어이루어져있고, 홍채실질은섬유아세포, 색소세포및교원섬유, 혈관등이풍부하며, 상피층은두층의상피세포로구성된다. 9 홍채낭종은 1869년 Hulke에의해처음보고되었다. 10 일반적으로발생원인에따라일차성과이차성낭종으로분류하며일차성낭종은별다른이유없이발생하며선천적 Figure 4. Two months later pictures. Slit lamp photography () and gonioscope photography (). The iris cyst (in the circle) was almost disappeared and pigmented lesion was remained by slit lamp examination () and gonioscopic examination (). 으로생기는경우가많다. 반면에이차성홍채낭종은안구천공, 안구내종양, 눈수술, 감염, 결핵, 축동제의장기간사용등이차적원인으로발생한다. 2-6,11 홍채낭종은발생위치에따라홍채전반부의대부분을차지하는실질에서낭종이생기는홍채실질낭종과홍채후반부의상피층에서낭종이생기는홍채색소상피낭종으로나눌수있다. 2-4 이중홍채실질낭종은홍채앞경계층을침범하여전방내까지확장되는반투명한홍채덩어리의형태를가지는것이특징이다. 1-4 대부분홍채색소상피낭종은보통안정적으로유지되어특별한치료가필요없는경우가많은반면, 홍채실질낭종의경우크기가증가하면시력저하, 광시증, 안구통증과같은증상이발생할수있다. 또한이차적으로폐쇄각녹내장, 고원홍채증후군, 색소분산증후군, 각막부종, 백내장, 수정체아탈구, 홍채염등의합병증을일으킬수있다. 12 이러한낭종에의한합병증은 10세이전의어린이에게선천적으로홍채실질낭종이발생한경우심하며시력예후도안좋은것으로알려져있다. 6 따라서홍채실질낭종에의한여러가지안구내문제발생시제거를위한노력이필요하며, 이전엔외과적절제및레이저치료를시도하였지만재발과합병증이많아최근에는 Shields 등이제시한낭종내알코올주입술로인한낭종경화술이안전하고 493
- 대한안과학회지 2018 년제 59 권제 5 호 - 효과적인방법으로소개되고있다. 12,13 일차안구결핵은안구가결핵균이우리몸에들어오는일차적매개체가되는것을말하며, 이차안구결핵은우리몸다른부분에결핵균이있고이결핵균이인접한구조물을통해이동하거나혈액학적으로이동하여눈으로결핵균이들어오는것을말한다. 이때안구내증상으로는염증을유발할수있고이러한염증은지방침착물성각막후면침착, 홍채육아종, 홍채뒤유착, 유리체염, 혈관염, 망막부종, 안내염등의형태로나타난다. 14,15 특히홍채결절로나타나는안구결핵의경우는각막을침범하는전방포도막염의형태로잘나타나지만이러한안구내부작용은전신적인항결핵치료에비교적반응이좋은것으로되어있다. 따라서이차적안구결핵에의한홍채낭종의치료는결핵의전신치료만으로충분한경우가많다. 16,17 안구결핵의진단은방수의 polymerase chain reaction이나조직검사등으로 Mycobacterium tuberculosis를확인하는것이확실한방법이기는하나위음성이많으며모든경우조직검사를할수없는어려움이있어결핵이빈번한나라에서는폐나전신적인결핵이있으면서위에서언급한안구내염증이나홍채결절등의증상이있으면안구결핵으로의심하고결핵균을치료하는것이임상에서는일반적이다. 17,18 본증례는전방내염증과홍채실질낭종의증으로인한이차적안압상승이발생한경우이다. 일반적으로소아에게생긴일차성홍채낭종이거나홍채실질낭종에의해합병증이발생한경우는적극적인수술이나시술이필요할수있으나, 본경우는환자가성인이었으며한달간의전방내염증과복막수술이후급격히생긴것으로보아이차적낭종으로생각할수있었다. 그리고전방내염증이있었던시기에결과적으로복막결핵이있었으며복막결핵을수술적으로제거후급속도로낭종이발생한것으로보아복막결핵의일차병소가수술에의한파종으로이차적홍채낭종의발생을일으켰다고의심할수있었다. 이것은다른전신검사상이상소견을관찰할수없었으며이후결핵치료로낭종의자발적관해가이루어진것을보아도결핵이원인이되어이차적홍채실질낭종이발생하였음을짐작할수있다. 하지만방수나홍채실질낭종조직검사의결과가없어서낭종의원인을확진할수없는것이이번증례의한계점이다. 그렇지만병변이홍채실질낭종이고전신적인결핵의치료와안과적대증적인치료만으로호전을보였기때문에진단을위해침습적인시술의필요성을느끼지못하였으며또한이러한진단을위한시술등이환자에게안구내출혈, 결핵균의파종등다른합병증을유발할수도있어시술없이지켜보았다. 그리고이차적인안구내결핵의 경우, 특히홍채낭종이발생한경우에는비록염증의소견이있지만일차병변인결핵을전신적으로치료하는것만으로안구내합병증이좋아지고 16,17 이러한현상은 Saricaoğlu et al 6 의보고에서도동일하게관찰된다. 24세남자환자가폐결핵에의한이차적인안구내결핵이단일홍채낭종으로나타났고전신적인결핵치료만으로 2달후홍채낭종이완전한관해를이루었다. 따라서본증례에서도결핵에대한전신적인치료를우선하였으며안과적치료는안구내증상에맞추어대증적치료를하면서홍채실질낭종의형태변화를면밀히관찰하였던것이다. 결론적으로홍채낭종이안구의병변으로발생한경우침습적인치료를즉각적으로시행하기보다는우선홍채낭종의양상과원인질환파악이필요하며, 특히이차적홍채낭종이의심되는경우원인질환에대한일차적치료를우선하며안과적으로는증상에맞게대증치료를하면서경과를관찰하는것이필요하리라사료된다. REFERENCES 1) Shields CL, Kancherla S, Patel J, et al. Clinical survey of 3680 iris tumors based on patient age at presentation. Ophthalmology 2012;119:407-14. 2) Shields J. Primary cysts of the iris. Trans m Ophthalmol Soc 1981;79:771-809. 3) Shields J, Shields CL, Lois N, Mercado G. Iris cysts in children: classification, incidence, and management. r J Ophthalmol 1999; 83:334-8. 4) Lois N, Shields CL, Shields J, Mercado G. Primary cysts of the iris pigment epithelium. Clinical features and natural course in 234 patients. Ophthalmology 1998;105:1879-85. 5) Shields J, Klinc MW, ugsburger JJ. Primary iris cyst: a review of the literature and report of 62 cases. r J Ophthalmol 1984; 68:152-66. 6) Saricaoğlu MS, Sengun, Guven D, Karakurt. Ocular tuberculosis with angle granuloma. Eye (Lond) 2004;18:219-20; discussion 220-1. 7) Thompson MJ, lbert DM. Ocular tuberculosis. rch Ophthalmol 2005;123:844-9. 8) Yeh S, Sen HN, Colyer M, et al. Update on ocular tuberculosis. Curr Opin Ophthalmol 2012;23:551-6. 9) Shin JH, Park HY, Cho SJ. Free-floating pigmented iris cyst in the anterior chamber. J Korean Ophthalmol Soc 2008;49:1862-66. 10) Hulke JW. Report on cases of cysts of the iris. Roy Lond Ophth Hosp Rep 1869;6:12. 11) Lee SS, Yoo SJ, Kim Y. Laser treatment for recurrent secondary iris cyst. J Korean Ophthalmol Soc 2004;45:146-51. 12) Shields CL, repalli S, Lally E, et al. Iris stromal cyst management with absolute alcohol induced sclerosis in 16 patients. JM Ophthalmol 2014;132:703-8. 13) Wiwatwongwana, Ittipunkul N, Wiwatwongwana D. b externo laser photocoagulation for the treatment of spontaneous iris stromal cyst. Graefes rch Clin Exp Ophthalmol 2012;250:155-6. 494
- 윤요셉외 : 안구결핵성홍채낭종의자발적회복 - 14) odaghi, LeHoang P. Ocular tuberculosis. Curr Opin Ophthalmol 2000;11:443-8. 15) Sheu SJ, Shyu JS, Chen LM, et al. Ocular manifestations of tuberculosis. Ophthalmology 2001;108:1580-5. 16) Rosen PH, Spalton DJ, Graham EM. Intraocular tuberculosis. Eye (Lond) 1990;4:486-92. 17) iswas J, Madhavan HN, Gopal L, adrinath SS. Intraocular tuberculosis. Clinicopathologic study of five cases. Retina 1995; 15:461-8. 18) lvarez GG, Roth VR, Hodge W. Ocular tuberculosis: diagnostic and treatment challenges. Int J Infect Dis 2009;13:432-5. = 국문초록 = 복막결핵환자에서발생한합병증을유발한홍채낭종의자발적회복 1 예 목적 : 복막결핵환자에서발생한홍채낭종으로인해포도막염및폐쇄각성안압상승이발생했을때결핵치료및보존적치료만으로홍채낭종의자발적회복을보인증례가있어보고하고자한다. 증례요약 : 49 세여자환자로본원내원한달전우안시력저하가있어일차병원안과에서우안홍채염진단하에스테로이드치료중전방내염증세포와 12 시방향으로전방각을완전히막는홍채낭종이발생하여내원하였다. 이때환자는본원내과와산부인과에서복막결핵으로진단후수술과약물치료중이었다. 환자는과거력상녹내장은없었지만홍채낭종발생시우안안압 29 mmhg 까지상승하였고전방내염증세포가 +1~+2 정도보였다. 고식적인낭종내알코올주입, 레이저치료, 절제술등과같은침습적인치료없이앓고있던결핵에대한약물치료와함께안압약과스테로이드안약치료만으로결핵의일차병변의호전과함께홍채낭종도완전히없어졌다. 결론 : 홍채낭종이안구의병변으로발생한경우침습적인치료를즉각적으로시행하기보다는우선홍채낭종의양상과원인질환파악이필요하며이차적양성홍채낭종이의심되는경우원인질환에대한일차적치료가필요할것으로생각된다. < 대한안과학회지 2018;59(5):491-495> 495