Case Report Korean J Otorhinolaryngol-Head Neck Surg 2019;62(2):125-30 / pissn 2092-5859 / eissn 2092-6529 https://doi.org/10.3342/kjorl-hns.2017.00479 Three Wall Orbital Decompression for Compressive Optic Neuropathy in Thyroid Ophthalmopathy Ji h Song 1, Joo Yeon Kim 1, Soo Jung Lee 2, and Jae Hwan Kwon 1 1 Department of Otolaryngology-Head and Neck Surgery, Kosin University College of Medicine, usan; and 2 Department of Ophthalomology, Inje University Haeundae Paik Hospital, usan, Korea 삼면안와감압술로치료한갑상샘눈병증에서발생한압박시신경병증 송지아 1 김주연 1 이수정 2 권재환 1 고신대학교의과대학이비인후과학교실, 1 인제대학교의과대학해운대백병원안과학교실 2 Received May 25, 2017 Revised July 7, 2017 ccepted July 17, 2017 ddress for correspondence Jae Hwan Kwon, MD, PhD Department of Otolaryngology- Head and Neck Surgery, Kosin University College of Medicine, 262 Gamcheon-ro, Seo-gu, usan 49267, Korea Tel +82-51-990-6247 Fax +82-51-245-8539 E-mail entkwon@hanmail.net Thyroid ophthalmopathy is an autoimmune disease that affect the orbital and periorbital soft tissue, characterized by bulging eye (exophthalmos) and compressed orbital structures, such as the optic nerves. The indications for surgical treatment for thyroid ophthalmopathy include decreased visual acuity caused by optic neuropathy, conjunctivitis and progressive facial deformity caused by exophthalmos. Orbital wall decompression by nasal endoscopy resulte in good cosmetic effects and visual recovery. alanced orbital decompression is considered to be a safe and effective surgery that can help avoid postoperative diplopia. We introduce three successful cases of orbital wall decompression for the treatment of thyroid ophthalmopathy. Korean J Otorhinolaryngol-Head Korean J Otorhinolaryngol-Head Neck Surg 2019;62(2):125-30 Neck Surg Key WordsZZ Graves opthalmopathy ㆍ Orbital decompression ㆍ Thyroid eye disease. 서론 갑상샘눈병증은자가면역기전에의한염증반응이일어나안와의구조물을침범하는질환으로외안근이나안와지방에림프구성침윤이발생하고부피가점점늘어나안구돌출이발생하며, 시신경등안와구조물을압박하게된다. 갑상샘눈병증에서안와감압술의적응증은압박시신경병증으로인한시력감소, 안구돌출에의한노출각막염, 심한안구돌출로인해얼굴의모습이변형된경우등이며, 수술의목적은안와지방을제거하거나안와주위벽을제거하여시신경에대한압력을완화하며시력을회복시키고안구돌출을호전시키는데있다. 1,2) 안와감압술의수술접근법으로결막경유법 (transconjunctival), 상악동경유법 (transantral), 내시경 적비내접근법 (endoscopic transnasal), 누구접근법 (transcaruncular) 등이있고, 현재까지는결막경유법과상악동경유법이가장많이시행되고있다. 1,3-5) 내시경적비내접근법은 1990 년 Kennedy 등 4,6,7) 이처음시도했으며, 외부에흉터를남기지않고하사근, 내안각인대및코눈물관, 안와아래신경등의주변구조물에손상을주지않으면서충분한안구돌출교정효과를얻을수있고안와첨의가장깊숙한부분까지접근이용이하여압박시신경병증을효과적으로회복시킬수있는것으로알려져있다. 6,7) 저자들은갑상샘눈병증에의한압박시신경병증으로시력감소및안구돌출이진행되던환자에서내시경을이용한비내접근법과결막경유법으로삼면안와감압술을시행한 3예를경험하였기에이를보고하고자한다. This is an Open ccess article distributed under the terms of the Creative Commons ttribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited. Copyright 2019 Korean Society of Otorhinolaryngology-Head and Neck Surgery 125
Korean J Otorhinolaryngol-Head Neck Surg 2019;62(2):125-30 증례 증례 1 52세여자환자가내원 1개월전부터서서히시작된복시, 양안의안구돌출을주소로내원하였다. 기저질환으로는갑상선기능항진증, 고혈압, 당뇨가있었으며, 초진시교정시력은우안 1.0, 좌안 0.9, 안압은우안 17 mm Hg, 좌안 16 mm Hg였으며, 안저검사에서특이소견은보이지않았다. Hertel 안구돌출계검사에서우안 23.5 mm, 좌안 23 mm(bar: 104) 로양안의안구돌출을보였으며 (Fig. 1), 안구운동검사에서모든안위에서양안의안구운동제한이관찰되었다. 안면부컴퓨터단층촬영에서네개외직근의비후소견및양안의심한안구돌출이보였으며, 안와자기공명영상촬영에서양안의시신경부종과시신경주위조영증강이관찰되었다 (Fig. 2 and ). 양안의갑상샘눈병증으로진단하고스테로이드 50 mg 경구투여를시작하였지만상기증상이더심해지고 5일간스테로이드 (Prednisolon) 500 mg 정맥투여후일주일간격으로 4 번더주사하였지만호전되지않아수술적치료를결정하였다. 수술전교정시력이우안 0.5, 좌안 0.5, 안압우안 21 mm Hg, 좌안 18 mm Hg, 안구돌출계는우안 25.5 mm, 좌안 26.5 mm(bar: 104) 로안구증상이더악화되었다. 내시경을이용한비내접근법으로양안의안와내벽, 내하벽감압술을, 외안각접근법으로외벽감압술을시행하였다. 수술은전신마취하에진행되었으며, 내시경을이용하여비내접근법으로사골동절제술을시행한후상악동자연공을넓혀안와내벽과하벽의뼈를노출시켰다. 뒤쪽으로는시신경관 (optic canal) 부터, 앞쪽으로는전두함요 (frontal recess) 까지, 바깥쪽으로는하안와신경 (infraorbital nerve) 의내측까지안와내하벽을제거한후, 노출된골막에 12번나이프를이용하여수평으로 3개의절개선을넣어안와지방이비강내로돌출되도록하여안와내부피를감소시켰다. 안와외벽감압술을위해외안각을수평으로절개한후하안검결막을절개하고골막을거상하여드릴로안와외벽을제거하였고, 골막에나이프를이용하여수평으로절개를넣어안와지방이돌출되도록하였다. 수술후 4일째부터양안모두교정시력 1.0으로개선되었고, 안구돌출계는우안 21.5 mm, 좌안 22.5 mm(bar: 104) 로양안에서 4 mm의안구돌출교정효과가있었으며, 복시및안구운동제한은호전되었다 (Figs. 1, 2C and D). 현재수술후 24개월동안안구돌출등의재발없이경과관찰중이다. 증례 2 57세여자환자가지속적으로심해지는좌안의안구돌출을주소로전원되었다. 환자는갑상선기능항진증으로타병원내과에서약물복용중이었으나최근 2개월간좌안의안구돌출로안과에서스테로이드및면역억제제를투여받았음에도지속적으로안구돌출이심해져수술적치료를위해본원으로전원되었다. 안구돌출계상양안차이가 2개월전 2 mm, 1개월전 4 mm, 본원이비인후과외래방문시 6 mm로점점심해지는양상이었으며시력은변화없었으나간헐적인복시를호소하였다 (Fig. 3). 안면부컴퓨터단층촬영에서좌안의안구내직근및하직근이비후되어있었으며, 우안의오래된안와내벽골절소견이보였으나과거력상안와주위 Fig. 1. Preoperative photo (case 1) shows mild conjunctival chemosis and severe exophthalmos (25.5 mm/ 26.6 mm) in both eyes (). Postoperatively, exophthalmos was improved by 4 mm in both eyes from 25.5 mm to 21.5 mm in the right eye and from 26.6 mm to 22.5 mm in the left eye, respectively (). 126
Three Wall Orbital Decompression Song J, et al. 수상및안구수술의병력은없었다 (Fig. 4 and ). 좌안의안구돌출이우측안와의내벽골절로인한가능성이있지만, 최근 2개월간 4 mm 이상심해져서갑상선안구병증으로인한안구돌출로판단하고수술적치료를계획하였다. 수술은 첫번째환자와같이내시경적비내접근법으로좌안의안구내벽및하내벽감압술을, 외부접근법으로안와외벽감압술을시행하였다. 수술직후복시등수술과관련된합병증은없으며, 술후 2개월째술전에비해안구돌출계검사상양안 C D Fig. 2. Preoperative coronal CT scan (case 1) shows severe enlargement of all rectus muscles (). xial view shows the distinct exophthalmos in both eyes (). Postoperative coronal CT scan shows the effect of bilateral medial, inferomedial wall (arrows) and lateral wall removal (arrowheads). Medial rectus muscle and orbital fat tissue were extruded to nasal cavity, which decrease orbital volume and pressure (C). xial view shows loss of medial (arrows) and lateral wall (arrowheads) of both orbit and improvement of exophthalmos in both eyes compared to preoperative one (D). Fig. 3. Preoperative photos (case 2) show severe exophthalmos of left orbit (). Postoperatively, left exophthalmos was improved by 4 mm (). www.kjorl.org 127
Korean J Otorhinolaryngol-Head Neck Surg 2019;62(2):125-30 차이 2 mm로호전되었으며술후 18개월째양안차이없이경과관찰중이다 (Figs. 3, 4C and D). 증례 3 53세남자환자로양안의복시와안구돌출을주소로내원 하였다. 우안이좌안에비해돌출이심하며우안이잘감기지않아간헐적인우안통증을호소하였다. 환자는갑상선기능항진증으로타병원에서약물복용중이었으며상기증상발생하여 2개월간스테로이드를투여하였지만증상호전없이악화되는소견으로수술적치료위해본원으로전원되었다. 초 C D Fig. 4. Preoperative xial CT scan (case 2) shows the distinct exophthalmos in left eye (). Coronal view shows severe enlargement of left rectus muscles (). Postoperative axial view shows loss of medial and lateral wall of left orbit and improvement of exophthalmos in left eye (C). Coronal CT scan shows the effect of medial, inferomedial wall and lateral wall removal of left orbit (D). Fig. 5. Preoperative photo (case 3) shows conjunctival chemosis and severe exophthalmos (25 mm/23 mm) in both eyes (). Postoperatively, exophthalmos was improved from 25 mm to 19 mm in the right eye and from 23 mm to 18 mm in the left eye (). 128
Three Wall Orbital Decompression Song J, et al. C D Fig. 6. Preoperative xial CT scan (case 3) shows the distinct exophthalmos in both eyes (). Coronal view shows severe enlargement of all rectus muscles (). Postoperative axial view shows loss of medial and lateral wall of orbit and improvement of exophthalmos in both eyes (C). Coronal view of facial bone CT scan shows the effect of medial, inferomedial wall and lateral wall removal (D). 진시교정시력은우안 0.2, 좌안 1.0, 안압은우안 20 mm Hg, 좌안 20 mm Hg였으며, Hertel 안구돌출계검사에서우안 25 mm, 좌안 23 mm로양안의안구돌출을보였으며, 안구운동검사에서모든안위에서양안의안구운동제한이관찰되었다. 또한안구돌출로인한장기간의안구노출로양안의각막염소견이관찰되었다 (Fig. 5). 안면부컴퓨터단층촬영에서네개외직근의비후소견및양안의심한안구돌출이보였다 (Fig. 6 and ). 양안의갑상샘눈병증으로진단하고수술적치료를계획하였다. 수술은이전증례와마찬가지로내시경적비내접근법으로양안의안구내벽및하내벽감압술을, 외부접근법으로안와외벽감압술을시행하였다. 수술직후복시등수술과관련된합병증은발생하지않았으며, 술후 1개월째우안의시력 0.5로호전되었고안구돌출계는우안 19 mm, 좌안 18 mm로우안 6 mm, 좌안 5 mm의안구돌출교정효과가있었고복시및안구운동제한도호전되었다. 현재수술후 3개월째안구돌출등의재발없이경과관찰중이다 (Figs. 5, 6C and D). 고찰 2008년 European Group on Graves Orbitopathy(EUGO- GO) 의갑상샘눈병증의치료에대한표준지침에따르면, 경 증갑상샘눈병증은금연, 갑상샘기능유지, 인공눈물등의보조적인대증요법만권하며중등도- 중증갑상샘눈병증은면역억제요법이나수술요법이필요하다. 8) 특히스테로이드는항염증및면역억제작용뿐아니라증상을호전시켜우선적으로사용하도록권하고있으며방사선조사요법도시도되고있으나이러한치료에반응하지않거나복시, 시력감퇴등이발생하면수술을고려해야한다. 8) 갑상샘눈병증환자의수술적치료로 1911 년 Dollinger 에의하여외벽감압술이처음으로시행되었다. 1,9) 1950년에 Hirsch 는안와하벽의제거를시도하였고, Walsh 와 Ogura는안와하벽및내벽을제거하여약 4~6 mm의안구돌출교정효과를얻을수있다고보고하였다. 1,9) 보통안와하벽과내벽의감압술이많이사용되고있으며, 새로운복시의발생이가장흔한합병증으로알려져있다. 수술후복시의발생은수술방법및문헌에따라약 30% 에서많게는 60% 까지보고되며, 안구의내측또는하방편위때문에발생하는것으로생각된다. 10-12) 이에 Graham 등 11) 은안와내벽과하벽및외벽을균형있게감압하는균형감압술 (balanced decompression) 을이용하면수술후복시의발생을 10% 정도로줄일수있다고하였으며, 본증례에서도안와외벽감압술을같이시행하였으며새로운복시등의합병증은발생하지않았다. 13) 수술시제거하는안와벽의제거개수에따라두벽감압술 (2 wall decom- www.kjorl.org 129
Korean J Otorhinolaryngol-Head Neck Surg 2019;62(2):125-30 pression) 과, 세벽감압술 (3 wall decompression) 이있으며, 대개안구돌출정도가심한경우에세벽감압술을시행한다. European Group on Graves Orbitopathy 의연구에따르면같은정도의안구돌출에서교정효과는두벽감압술에서 4.3 mm, 세벽감압술에서 6.0 mm 정도로더큰것으로보고하였다. 12) 저자의보고에서앞의두증례에서는세벽감압술을하였지만모두 4 mm 정도안구돌출이호전되었는데, 이는안와하벽및외벽의감압정도가충분치않아서라고생각되며, 감압정도를증가시킨마지막증례에서는 6 mm 정도의안구돌출호전결과가있었다. 최근안와감압술시행시합병증을최소화하면서수술효과를최대로증대시키기위한노력이진행되고있다. 국내에서 Lee 등 14) 은수술전컴퓨터단층촬영사진으로외안근및안와지방의침범정도를평가하고수술중안구돌출정도를평가하여그에따라수술방법을달리하는단계적감압술 (graded decompression) 을소개하였다. 또다른보고에서는기존신경외과영역에서사용되던정위적수술항해장치를이용하면좀더안전하고효과적인안와감압술을시행할수있다고하였다. 15) 시력호전의정도를보면 Levy 등 6) 과 Tang 등 7) 에의한보고에서압박시신경병증환자에서내시경을이용한안와감압술후모든예에서확연한시력의개선을보였다고하였으며, 본보고의첫증례에서도 1.0으로만족할만한시력회복을보였다. 갑상샘눈병증환자에서내시경을이용한비내접근법의장점으로흉터가남지않는다는점에서미용적인효과가있으며, 출혈및부종의감소등으로수술후기능적인회복또한기존수술법에비해유리할것으로보이고, 내시경을이용하면시신경및안와구조물이밀집해있는뒤쪽까지쉽게접근하여감압할수있으므로첫증례와같이갑상샘눈병증환자에서압박시신경병증을동반하는경우더욱효과적이라고보고된다. 6,7) 내시경수술시문제점으로지적되던수술시야의협소함및기구사용의어려움등은장비의발달에힘입어개선되고있으며, 내시경을활용하는수술분야가늘어나면서술자의숙련도및기구의편리성도점차증대되고있다. 아직이비인후과에서는내시경을이용한비강내접근법이, 안과에서는결막경유법등의외부접근법에익숙하다. 하지만안와내벽및하벽뿐만아니라복시등의합병증을줄여주고수술결과도극대화할수있는외벽감압술도같이시행하면더좋은결과를얻을수있을것이라고생각되며, 향후본증례와같이갑상샘눈병증환자에서뿐만아니라안와골 절, 안와종양등의분야에서내시경을이용한비내접근법은 더욱유용하게활용될수있을것으로보인다. 저자들은세명 의갑상샘눈병증환자에서비내시경을이용한안와내벽및 내하벽안와감압술과, 외안각접근법을이용한외벽감압술을 시행하였으며, 특별한합병증없이안구돌출및시력이호전 되었기에문헌고찰과함께보고하는바이다. REFERENCES 1) Siracuse-Lee DE, Kazim M. Orbital decompression: current concepts. Curr Opin Ophthalmol 2002;13(5):310-6. 2) Goh MSY, McNab. Orbital decompression in Graves orbitopathy: efficacy and safety. Intern Med J 2005;35(10):586-91. 3) oboridis KG, unce C. Surgical orbital decompression for thyroid eye disease. Cochrane Database Syst Rev 2011;(12):CD007630. 4) Michel O, Oberländer N, Neugebauer P, Neugebauer, Rüssmann W. Follow-up of transnasal orbital decompression in severe Graves ophthalmopathy. Ophthalmology 2001;108(2):400-4. 5) Paridaens D, Verhoeff K, ouwens D, van Den osch W. Transconjunctival orbital decompression in Graves ophthalmopathy: lateral wall approach ab interno. r J Ophthalmol 2000;84(7):775-81. 6) Levy J, Puterman M, Lifshitz T, Marcus M, Segal, Monos T. Endoscopic orbital decompression for Graves ophthalmopathy. Isr Med ssoc J 2004;6(11):673-6. 7) Tang IP, Prepageran N, Subrayan V, Tajunisah I. Endoscopic orbital decompression for optic neuropathy in thyroid ophthalmopathy. Med J Malaysia 2008;63(4):337-8. 8) artalena L, aldeschi L, Dickinson, Eckstein, Kendall-Taylor P, Marcocci C, et al. Consensus statement of the European Group on Graves orbitopathy (EUGOGO) on management of GO. Eur J Endocrinol 2008;158(3):273-85. 9) Lund VJ, Larkin G, Fells P, dams G. Orbital decompression for thyroid eye disease: a comparison of external and endoscopic techniques. J Laryngol Otol 1997;111(11):1051-5. 10) Mainville NP, Jordan DR. Effect of orbital decompression on diplopia in thyroid-related orbitopathy. Ophthal Plast Reconstr Surg 2014;30 (2):137-40. 11) Graham SM, rown CL, Carter KD, Song, Nerad J. Medial and lateral orbital wall surgery for balanced decompression in thyroid eye disease. Laryngoscope 2003;113(7):1206-9. 12) European Group on Graves Orbitopathy (EUGOGO), Mourits MP, ijl H, ltea M, aldeschi L, oboridis K, et al. Outcome of orbital decompression for disfiguring proptosis in patients with Graves orbitopathy using various surgical procedures. r J Ophthalmol 2009; 93(11):1518-23. 13) Unal M, Leri F, Konuk O, Hasanreisoğlu. alanced orbital decompression combined with fat removal in Graves ophthalmopathy: do we really need to remove the third wall? Ophthal Plast Reconstr Surg 2003;19(2):112-8. 14) Lee KH, Jang SY, Lee SY, Yoon JS. Graded decompression of orbital fat and wall in patients with Graves orbitopathy. Korean J Ophthalmol 2014;28(1):1-11. 15) Lim KS, Lee JK. Evaluation of stereotactic navigation during orbital decompression in thyroid-associated orbitopathy patients. J Korean Ophthalmol Soc 2014;55(3):337-42. 130