= 증례보고 = 눈물샘길질환의임상적고찰 대한안과학회지 2014 년제 55 권제 1 호 J Korean Ophthalmol Soc 2014;55(1):7-12 pissn: 0378-6471 eissn: 2092-9374 http://dx.doi.org/10.3341/jkos.2014.55.1.7 신선애 최창욱 김상덕 원광대학교의과대학안과학교실, 원광의과학연구소 목적 : 임상적으로흔하지않으며다른질환으로오인하기쉬운눈물샘길질환환자들을대상으로임상양상과치료방법을알아보고자하였다. 대상과방법 : 2007 년 3 월부터 2013 년 4 월까지본원에서눈물샘길질환으로진단받고치료했던환자 11 명, 11 안 ( 남자 5 명, 여자 6 명 ) 을대상으로의무기록을통한후향적연구를통해임상증상및검사소견, 치료방법, 재발여부를조사하였다. 결과 : 총 11 명의눈물샘길질환환자중 4 명은눈물소관낭포 (dacryops), 7 명은눈물길결석 (dacryolith) 에의한눈물샘세관염 (lacrimal gland ductulitis) 이었다. 평균연령은 47.9 세 (30-80 세 ) 였으며, 눈물샘세관염환자들은내원전몇개월동안결막염또는다래끼로치료받은경험이있었다. 발생부위는우안 4 명, 좌안 7 명이었으며, 증상은가쪽눈구석불편감, 농성분비물, 종괴순이었고, 눈물소관낭포 2 명과눈물샘세관염 1 명에서조직검사를시행하였으나특징적인소견을보이지않았다. 치료는눈물소관낭포는조대술 (marsupialization) 을시행하였고, 눈물샘세관염은위상피절제및결석제거술 (excision and dacryolith curettage) 로결석및박혀있는속눈썹을제거하고점안및경구항생제를투여하였으며, 약 2 개월의경과관찰에서모두호전되었다. 결론 : 눈물샘길질환은결막염, 다래끼, 안와낭종등으로오인될수있어정확한진단과치료가필요하다. < 대한안과학회지 2014;55(1):7-12> 눈물샘길질환은임상적으로흔하지않아결막염, 다래끼, 안와낭종등으로오인될수있으므로정확한진단과치료가필요하다. 눈물소관낭포는눈물샘조직이존재하는어느곳에서나발생할수있으며, 위치에따라안검엽, 안와엽, 부눈물샘, 이소성낭종으로나눌수있다. 1,2 눈물소관낭포는일반적으로통증이나압통이없이상안검의외측에서부드럽게촉지되는유동성의종괴로발견되지만염증이나감염동반시빠르게성장하거나통증을유발할수있으며, 안검외반, 안검하수, 안구돌출, 복시등의증상을호소할수있다. 1-9 눈물길결석은눈물주머니, 코눈물관, 눈물소관, 눈물샘관등눈물분비및배출기관에서발생할수있으며, 이중특히눈물샘관에발생한결석은매우드문것으로알려졌다. 10-12 눈물샘관에발생한결석은무증상인경우도있으나, 통증, 눈부심, 눈물흘림, 심한경우안구돌출을일으 Received: 2013. 8. 16. Revised: 2013. 8. 28. Accepted: 2013. 11. 20. Address reprint requests to Sang Duck Kim, MD, PhD Department of Ophthalmology, Wonkwang University Hospital, #895 Muwang-ro, Iksan 570-711, Korea Tel: 82-63-859-1370, Fax: 82-63-855-1801 E-mail: sangduck@wonkwang.ac.kr * This study was presented as a narration at the 106th Annual Meeting of the Korean Ophthalmological Society 2011. * This study was supported by research fund of Wonkwang University in 2013. 킬수있다. 10-13 저자들은임상적으로눈물샘길질환으로진단받고치료받은 11명 (11안) 의환자들을통하여임상증상, 검사소견, 치료방법과결과에대해알아보고자하였다. 대상과방법 2007년 3월부터 2013년 4월까지본원안과를방문하여눈물샘길질환으로진단받은 11명, 11안을대상으로임상증상, 검사소견, 치료방법과결과에대해알아보고자하였다. 가쪽눈구석불편감, 종괴, 농성분비물등의증상을주소로내원하여눈물샘세관염으로진단받은 7안은위상피절제및결석제거술 (excision and dacryolith curettage) 을시행하였고, 눈물소관낭포로진단받은 4안중 3안은조대술 (marsupialization) 을시행하였으며, 1안은수술을권유하였으나환자가특별한증상이없어치료를원하지않아시행하지않았다 (Table 1). 수술방법은결막낭내에 0.5% proparacaine hydrochloride (Alcon, Fort Worth, TX, USA) 로점안마취하고 1:100,000 epinephrine이혼합된 2% lidocaine 국소마취제를가쪽눈구석주변과위쪽결막낭에주입하였다. 국소마취후윗눈꺼풀을뒤집고결막수술가위로위상피성조직을절제한후 curette을이용하여내용물을제거하였다. www.ophthalmology.org 7
- 대한안과학회지 2014 년제 55 권제 1 호 - Table 1. Summary of management of 11 cases of lacrimal gland ductal disease No Age Duration Signs & Symptoms (years)/sex (months) Diagnosis Treatment 1 35/M Right upper lid mass 1 Dacryops Marsupialization & biopsy 2 36/M Left upper lid mass 2 Lacrimal gland ductulitis Excision & dacryolith curettage & biopsy 3 31/F Right upper outer lid mass with foreign 2 Lacrimal gland ductulitis Excision & dacryolith curettage body sensation 4 55/M Left eyelid swelling with pus like 3 Lacrimal gland ductulitis Excision & dacryolith curettage discharge 5 66/F Left mass of lateral canthus 1 Dacryops Marsupialization & biopsy 6 30/F Left eye discharge, some tearing 1 Lacrimal gland ductulitis Excision & dacryolith curettage 7 72/M Left eye foreign body sensation 1 Dacryops Observation 8 51/F Left eyewax, itching sensation 3 Lacrimal gland ductulitis Excision & dacryolith curettage 9 34/F Left eye foreign body sensation, 2 Lacrimal gland ductulitis Excision & dacryolith curettage tenderness 10 80/M Right eye foreign body sensation 2 Dacryops Marsupialization 11 34/F Right pus like discharge 2 Lacrimal gland ductulitis Excision & dacryolith curettage M = male; F = female. 찰하였다. 결 과 Figure 1. Post-operative photograph of marsupialization of right dacryops of 38-year-old man. The cyst walls marsupialized and the edges of the cyst lining were coapted to the edges of conjunctiva with 8-0 vicryl sutures. 조대술은절개수술과마찬가지의방법으로국소마취를시행한후윗눈꺼풀을뒤집고 11번 mass로낭종벽을눈꺼풀테와평행하게절개하였다. 그런다음낭종벽을부드럽게잡고낭종벽일부를절제한후낭종내의내용물을배출시키고낭종의개구부를남겨놓은채절제부위의양쪽가장자리를 8-0 vicryl을이용하여결막에봉합하였다 (Fig. 1). 이중눈물소관낭포 2명과눈물샘세관염 1명에서조직검사를시행하였다. 수술후항생제점안액 (Cravit, 0.5% levofloxacin, Santen) 과스테로이드점안액 (Ocumetholone, 0.1% fluorometholone, Samil) 을하루 4회점안하였으며, 경구용광범위항생제를 2주동안복용하게하였다. 수술후 3일, 10일, 1개월, 2개월순으로외래진료에서세극등현미경을이용하여경과관 눈물샘길질환으로진단받은환자 11명 11안중 10안을대상으로수술이이루어졌으며, 성별분포는남자 5명, 여자 6명이었다. 연령은 30세에서 80세까지로평균 47.9세였으며, 발생부위는우안 4안 (36.4%), 좌안 7안 (63.6%) 으로모두상외측원개결막부위였다. 증상은가쪽눈구석불편감이 4안, 농성분비물이 4안, 특별한증상없이종괴가관찰된경우가 3안이었고, 증상의기간은 1개월에서 3개월 ( 평균 2개월 ) 이었다. 이중 5안 (45.4%) 에서는본원내원전결막염혹은다래끼로오인하여항생제점안 (2안), 또는절개및배농술 (3안) 을시행받은경험이있었다. 눈물소관낭포 2안과눈물샘세관염 1안에서조직검사를시행하였는데눈물소관낭포 1안에서는정상눈물소관, 1안은단순낭종, 눈물샘세관염 1안은위상피성과형성소견을보였다. 수술후약 2개월의경과관찰후눈물소관낭포또는눈물샘세관염의재발은없었으며, 증상은모두호전되었다 (Fig. 2, 3). 모든환자에서수술후안구건조증등의특별한합병증은발생하지않아만족스러운수술결과를보였다. 고찰 눈물샘은안와내위쪽가의약간움푹패인눈물샘오목에위치하며위눈꺼풀올림근널힘줄의가쪽뿔에의하여안와엽과안검엽으로나뉘며안와엽은안검엽보다크기가크다. 각부분은소엽들로이루어져있으며소엽샘꽈리에서의분 8 www.ophthalmology.org
- 신선애외 : 눈물샘길질환의임상적고찰 - A B C Figure 2. (A) Pre-operative photograph of dacryops of left lacrimal gland of 66-year-old female presenting with cystic mass at the left superotemporal conjunctival cul-de-sac. (B) Post-operative (3 days after marsupialization) photograph. (C) Post-operative (1 month after marsupialization) photograph. 비물은먼저미세한소엽사이세관을통과해마침내분비관에이른다. 안와엽의소관은안검엽의소관과합쳐져상측원개결막에있는결막낭으로들어간다. 14 눈물소관낭포는눈물샘조직이존재하는곳이면어디든지발생할수있으며, 1,2,6-8 1803년 dacryops 로명명된이후국내외여러학자들에의해보고되어왔다. 15 Duke-Elder 15 는눈물소관낭포를안검엽의단순낭종, 안와엽낭종, 기생낭종, 유피낭종으로분류하였고, Bullock et al 2 은안검엽낭종 ( 단순낭종 ), 안와엽낭종, Krause와 Wolfring 샘에생기는부눈물샘낭종, 이소성눈물샘에생기는낭종으로분류하였다. 눈물소관낭포는대부분안검엽과안와엽이있는상외측원개결막부위에서발생하며, Wolfring샘이있는상안검검판상연의내측이나하안검검판하연의내측, Krause샘이있는원개결막부위에발생할수있고, 이소성낭종은눈주위의어느부위에서나발생할수있다. 16-18 저자의경우에도총 11안중 11안모두상외측원개결막부위에발생 하였으며이는해부학적위치로볼때안검엽또는안와엽눈물소관낭포로생각된다. 눈물소관낭포의발생원인으로 Bullock et al 2 은안검엽낭종의경우주로염증또는외상에의해, 안와엽낭종은대부분선천성으로, 부눈물샘낭종은안검엽낭종과마찬가지로주로외상이나감염에의해, 이소성낭종은만성적염증과연관되어발생한다고하였고, Hornblass and Herschorn 6 은감염, 외상또는 Stevens-Johnson syndrome과같은염증으로인해발생한다고하였다. 저자의경우에는총 11안중 2안 (18.1%) 에서이전의눈수술병력이있었으며, 5안 (45.4%) 에서결막염혹은다래끼로항생제점안이나절개및배농술로치료받은병력이있었다. 눈물소관낭포의발생기전은정확하게밝혀진바가없다. 초기보고에서는염증이나외상으로인해생긴눈물샘의잔존물이나반흔에의해배출소관이기계적으로막힘으로써낭종이형성된다고하였으나, 1,2 눈물배출계는정상적으 www.ophthalmology.org 9
- 대한안과학회지 2014 년제 55 권제 1 호 - A B C Figure 3. (A) Pre-operative photograph of a cystic mass with concretions at the left lateral canthal angle illustrated in 30-year-old female. (B) Macroscopic view of dacryolith and hair removed from the lacrimal gland ductule during the operation. (C) Post-operative (1 month after excision and dacryolith curettage) photograph. 로분비압이낮으며, 실험이나수술적으로눈물소관이막혔을경우근위부눈물샘조직이위축되며분비가중단됨을발견하였다. 2,5 Weatherhead 16 나 Bay and Roh 1 는눈물소관상피하형질세포에서능동적으로분비된 IgA가낭종내에축적되어발생한삼투압이낭종형성의동력이라고하였다. 눈물길결석은눈물주머니, 코눈물관, 눈물소관, 눈물샘관등눈물분비및배출기관에서발생할수있으며, 다른눈물배출계에서발생하는결석은비교적흔하지만, 눈물샘관에만발생하는결석은매우드물다. 10-12 눈물샘관결석의발생기전은명확하게밝혀지지않았지만 Duke-Elder 15 는감염이나독성상태에서박리된세포의핵주변에서발생한다고하였고, Baratz et al 19 은눈물낭의속눈썹이초기염증반응과결석형성의병소로작용한다고하였으며, Baker and Bartley 11 는정상눈물분비의구성요소인알부민이응축물로작용한다고하였다. 눈물소관낭포는일반적으로증상이없이상안검의외측 에서촉지되는종괴로발견되지만 2,5 울거나추위에노출되었을때크기가커질수있으며, 외부에서압력이가해질경우갑작스러운눈물과함께크기가작아지기도한다. 2,3,7 염증이나감염동반시빠르게성장하고통증을유발하거나누공을형성할수있으며, 낭종의크기가클경우안검외반, 안검하수, 안구돌출, 복시등의증상을호소할수있다. 2,3,5 눈물소관낭포와달리눈물샘관결석환자들은대부분통증, 눈부심, 눈물흘림등의증상을호소하며눈물샘의크기가커질경우안구돌출도있으킬수있다. 10,11 저자의경우총 11안의환자중가쪽눈구석불편감을호소한경우가 4안 (36.3%), 농성분비물이 4안 (36.3%), 특별한증상없이종괴만관찰된경우가 3안 (27.2%) 이었다. 눈물소관낭포의조직학적소견은다양하며낭종벽은주로편평혹은원주상피세포로구성된위중층비각화상피세포 (pseudostratifiednonkeratinizing epithelial cells), 6 내측의입방세포층과외측의근상피세포층, 5 두층의비섬모입 10 www.ophthalmology.org
- 신선애외 : 눈물샘길질환의임상적고찰 - 방세포층 16 등다양하게보고되었으며, 대부분두층으로구성되어있으며, 만성화된낭종에서는단층으로구성되기도한다. 6 Jakobiec et al 20 은최근연구에서정상눈물샘소관과눈물샘소관낭포에는면역조직학적으로근상피세포가존재하지않으며, 따라서눈물소관낭포의발생이눈물소관의신경근수축의실패와관련된다는가설은맞지않다고보고하였다. 저자의경우조직검사를시행한눈물소관낭포 2안과눈물샘세관염 1안중, 눈물소관낭포 1안에서는정상눈물소관, 나머지 1안은입방상피세포로구성된단순낭종, 눈물샘세관염 1안은위상피성과형성소견을보였다. 치료로는눈물소관낭포는주사침배액술이나완전절제술또는조대술을시행할수있다. 주사침배액술이나인위적인파열술및부분적절제술은재발이흔하고, 완전절제술의경우주위정상눈물샘조직이손상될경우눈물샘분비능의변화로안구건조증을초래할수있다. 1-9 반면조대술은낭종의완전절제술로인한이차적인눈물샘분비능의저하의위험성을줄이면서비교적양호한치료성적이보고되어 1,3-6,21 저자의경우눈물소관낭포의치료로조대술을시행하였다. 눈물길결석에의한눈물샘세관염의치료로는위상피절제후결석과내용물을제거하였는데 6예중 3예에서속눈썹이함께발견되었다 (Fig. 3). 수술후모든예에서재발은없었으며안구건조증등의불편감도보이지않았다. 눈물샘길질환은임상적으로흔하지않지만눈꺼풀또는눈물샘와에생긴낭종의감별진단에고려해보아야할중요한질환으로 5 임상특징, 해부학적위치및검사를통해확진할수있다. 눈물소관낭포의자연관해는보고되지않았으며, 6 환자들은만성결막염, 다래끼등으로오인하여오랫동안치료받고불편함을호소하는경우가많다. 눈물소관낭포또는눈물샘세관염은국소마취를통한비교적간단한수술적방법으로완치될수있으므로임상적으로눈물샘길질환이의심되는환자에서빠르고정확한진단과치료가필요할것으로생각된다. REFERENCES 1) Bay SW, Roh JH. Clinical characteristic of lacrimal ductal cyst. J Korean Ophthalmol Soc 2001;42:803-9. 2) Bullock JD, Fleishman JA, Rosset JS. Lacrimal ductal cysts. Ophthalmology 1986;93:1355-60. 3) Salam A, Barrett AW, Malhotra R, Olver J. Marsupialization for lacrimal ductular cysts (dacryops): a case series. Ophthal Plast Reconstr Surg 2012;28:57-62. 4) Harris GJ. Marsupialization of a lacrimal gland cyst. Ophthalmic Surg 1983;14:75-8. 5) Smith S, Rootman J. Lacrimal ductal cysts. Presentation and management. Surv Ophthalmol 1986;30:245-50. 6) Hornblass A, Herschorn BJ. Lacrimal gland duct cysts. Ophthalmic Surg 1985;16:301-6. 7) Park JH, Chun JS, Moon NJ, Beun DS. A case of lacrimal gland duct cyst. J Korean Ophthalmol Soc 1994;35:1476-80. 8) Lee SH, Lew H, Yun YS. A case of lacrimal ductal cyst with dacryolith. J Korean Ophthalmol Soc 2004;45:131-4. 9) Jeung YW, Rho JH. A case of lacrimal gland duct cyst associated with ptosis. J Korean Ophthalmol Soc 1997;38:1072-6. 10) Zafar A, Jordan DR, Brownstein S, Faraji H. Asymptomatic lacrimal ductule dacryolithiasis with embedded cilia. Ophthal Plast Reconstr Surg 2004;20:83-5. 11) Baker RH, Bartley GB. Lacrimal gland ductule stones. Ophthalmology 1990;97:531-4. 12) Kwon YD, Lim JW. An eyelash-induced lacrimal gland stone. J Korean Ophthalmol Soc 2010;51:1139-41. 13) Halborg J, Prause JU, Toft PB, et al. Stones in the lacrimal gland: a rare condition. Acta Ophthalmol 2009;87:672-5. 14) Bron AJ. Lacrimal streams: the demonstration of human lacrimal fluid secretion and the lacrimal ductules. Br J Ophthalmol 1986; 70:241-5. 15) Duke-Elder S. System of ophthalmology. Part II. The ocular adnexa. Lacrimal, orbital and para-orbital diseases. 1st ed. vol. 13. London: Henry Kimpton, 1974;638-43. 16) Weatherhead RG. Wolfring dacryops. Ophthalmology 1992;99: 1575-81. 17) Woo KI, Kim YD. Cyst of accessory lacrimal gland. Korean J Ophthalmol 1995;9:117-21. 18) Kacerovska D, Michal M, Ricarova R, et al. Apocrine secretion in lacrimal gland cysts (dacryops): a common but underrecognized phenomenon. J Cutan Pathol 2011;38:720-3. 19) Baratz KH, Bartley GB, Campbell RJ, Garrity JA. An eyelash nidus for dacryoliths of the lacrimal excretory and secretory systems. Am J Ophthalmol 1991;111:624-7. 20) Jakobiec FA, Zakka FR, Perry LP. The cytologic composition of dacryops: an immunohistochemical investigation of 15 lesions compared to the normal lacrimal gland. Am J Ophthalmol 2013; 155:380-96.e1. 21) Eifrig CW, Chaudhry NA, Tse DT, et al. Lacrimal gland ductal cyst abscess. Ophthal Plast Reconstr Surg 2001;17:131-3. www.ophthalmology.org 11
- 대한안과학회지 2014 년제 55 권제 1 호 - =ABSTRACT= Clinical Evaluation of Lacrimal Gland Ductal Disease Seon Ae Shin, MD, Chang Wook Choi, MD, Sang Duck Kim, MD, PhD Department of Ophthalmology, Wonkwang University School of Medicine, Institute of Wonkwang Medical Science, Iksan Korea Purpose: To investigate the clinical characteristics and management of lacrimal gland ductal disease, a rare disease often mistaken for other diseases. Methods: A retrospective chart review of 11 patients (11 eyes, 5 males, 6 females) diagnosed with lacrimal ductal disease between March 2007 and April 2013 was performed. Results: Among 11 eyes in 11 patients, 4 were diagnosed with dacryops and 7 with lacrimal gland ductulitis initiated by dacryolith. The mean age of the subjects was 47.9 years (range, 30-80 years). Lacrimal gland ductulitis patients received treatment for conjunctivitis or hordeolum for several months. Four cases involved the right eye and 7 cases involved the left eye. Symptoms included foreign body sensation, pus-like discharge and palpable mass. Biopsy was performed in 3 cases and showed no specific findings. Patients with dacryops underwent marsupialization, whereas patients diagnosed with lacrimal gland ductulitis underwent excision and dacryolith curettage. During the 2-month follow-up period, all cases showed no signs of recurrence or complications. Conclusions: Lacrimal gland ductal disease can be mistaken for other diseases such as conjunctivitis, hordeolum, or orbital cyst, thus requiring accurate diagnosis and appropriate management. J Korean Ophthalmol Soc 2014;55(1):7-12 Key Words: Dacryolith, Dacryops, Lacrimal gland ductulitis, Marsupialization Address reprint requests to Sang Duck Kim, MD, PhD Department of Ophthalmology, Wonkwang University Hospital #895 Muwang-ro, Iksan 570-711, Korea Tel: 82-63-859-1370, Fax: 82-63-855-1801, E-mail: sangduck@wonkwang.ac.kr 12 www.ophthalmology.org