untitled

CASE REPORT Intest Res 2007;5(1):87-91 복부종괴로발현된다발성침윤장자궁내막증 1 예 한양대학교의과대학구리병원내과학교실 조현석 한동수 박세우 변태준 김태엽 은창수 전용철 손주현 A Case of Multiple Deep Infiltrating Intestinal Endometriosis Presenting as Abdominal Mass Hyun Seok Cho, M.D., Dong Soo Han, M.D., Se Woo Park, M.D., Tae Jun Byun, M.D., Tae Yeob Kim, M.D., Chang Soo Eun, M.D., Yong Cheol Jeon, M.D., Joo Hyun Sohn, M.D. Department of Internal Medicine, Hanyang University College of Medicine, Guri Hospital, Guri, Korea Endometriosis is one of the most common benign gynecologic conditions in women of childbearing age. Endometriosis involving the gastrointestinal tract occurs in 5.3-12% of women and the most commonly affected site is the rectum and the sigmoid colon. The reports of intestinal endometriosis in Korea are too few to obtain a rate of occurence, and in these cases, the depth of invasion was limited to the serosa, resulting in no significant symptoms or signs. Intestinal endometriosis can show up as a mass suggestive of malignant neoplasm on imaging studies, which can result in serious errors in diagnosis and treatment. In Korean studies, the masses were solitary, and there was no reported case of multiple endometriosis in the sigmoid colon, ascending and descending colon. We report a case of multiple deep infiltrating intestinal endometriosis presenting as an abdominal mass. (Intest Res 2007; 5:87-91) Key words: Endometriosis; Intestines; Multiple abdominal mass 서 자궁내막증은가임기여성에서생기는부인과질환중가장흔한양성질환으로자궁내막조직이비정상적으로자궁이외의장소에발생하여증식하는질환이다. 1 장관이나골반을침범하는침윤자궁내막증은복막이나난소보다그빈도가적으며, 이중장관에서발생하는빈도는 5.3-12% 이다. 2,3 장침범의호발부위는직장, S 상결장, 충수돌기, 회장그리고드물게맹장의순이며, 이중에서도직장및 S 상결장을침범하는경우가가장흔하다. 4,5 그러나국내에서는간헐적으로보고되어직장및 S 상결장에대한침범빈도는알수없고, 6 침범정도는대부분장의장막에국한되어서임상적으로중요한증세를나타내지않는다. 직장점막에발생하는자궁내막증은직장수지검사에서단단한종괴로촉지될뿐만아니라영상검사에서는악성종양을추정 연락처 : 한동수, 경기도구리시교문동 249-1 (471-701) 한양대학교구리병원소화기내과 Tel: 031) 560-2226, Fax: 031) 555-2998 E-mail: hands@hanyang.ac.kr 론 케하는음영이나타나직장암으로오인되기도하여진단과치료에중대한오류를범할수있다. 7 국내에서는이러한종괴들이모두단일성이었고 S 상결장, 상행결장과하행결장그리고장간막에다발성으로침범하는예는아직보고된바없다. 저자들은복부종괴를주소로내원하여대장내시경과진단개복술후장점막을침범한다발성침윤장자궁내막증으로진단한 1 예를경험하여서이를보고한다. 증 49 세여자환자가내원 1 달전부터시작된하복부복통과종괴를주소로내원하였다. 복통은간헐적이었으며양상은쥐어짜는듯하였고자세와는관련이없었으며음식물섭취와도무관하였다. 개인력에서음주력이나흡연력은없었으며 2 년전자궁샘근육증 (adenomyosis) 으로자궁절제술을시행하였으며 1 달뒤우측유방의샘증 (adenosis) 으로종괴절제술을시행한과거력이있었다. 례 87

Intest Res : Vol. 5, No. 1, 2007 Fig. 1. Abdominal CT findings (A, B). A 2.4 cm sized well-enhancing mass is noted in the lateral side of proximal ascending colon (white arrow) and a 4.3 cm sized lobulated mass is also noted in the mid-descending colon (black arrow). Fig. 2. (A) Colonoscopic finding. The protruding lesion has a chicken skin-like abnormal mucosal appearance at the margin that measured as 0.8 cm at the sigmoid colon. (B) EUS finding. The inhomogeneous 3 cm sized mass like lesion is separated by the serosal layer and no association with the serosal layer. It destructs all layers of bowel. EUS, endoscopic ultrasonography. 내원당시활력징후는혈압 130/80 mmhg, 맥박 76회 / 분, 호흡수 18회 / 분, 체온 36.4 o C였다. 환자는비교적안정되어보였으며의식은명료하였고두경부검진에서공막에황달이나결막의창백등은관찰되지않았으며심음이나호흡음은정상이었다. 복부검사에서좌하복부에 3 cm 가량의종괴가촉지되었으며압통이나반발통은없었다. 장음은감소되었으며늑골척추각압통은없었다. 말초혈액검사에서백혈구 6,000/mm 3, 혈색소 15.3 g/dl, 헤마토크리트 42.3%, 혈소판 224,000/mm 3 였다. 생화학검사와소변검사에서이상소견은없었고, 대변잠혈검사는음성이었으며종양표지자검사에서 CEA와 CA-125는정상범위이내였다. 복부전산화단층촬영에서상행결장의외측으로 2.4 cm, 하행결장의외측으로 4.3 cm, 장간막하부에 1.3 cm, 골반강에 0.8 cm 크기의조영증강되는종괴가관찰되었다 (Fig. 1A, B). 대장내시경에서 S 상결장에주변점막의오톨도톨한변화를동반한 0.8 cm 크기의용종양돌출병변이있었고주위로점막의변형이관찰되었다 (Fig. 2A). 내시경초음파에서동일부위에장벽과분리되는내부가불균질한 3 cm 직경의종괴가관찰되었고종괴로인하여장벽전층이파괴되었으나, 장벽층과의연관성은없었다 (Fig. 2B). 이후시행한대장조영술에서상행결장근위부와하행결장원위부에충만결손이관찰되었다 (Fig. 3). 내시경조직검사에서만성염증외엔특이소견을보이지않아악성질환과의감별을위해진단개복술을 88

Hyun Seok Cho, et al:a Case of Multiple Deep Infiltrating Intestinal Endometriosis Presenting as Abdominal Mass 시행하였고, 각각의종괴를절제하였다. 수술후종괴의현미경소견은장벽으로부터시작되어주로근육층에침윤되어있는자궁내막샘과자궁내막버팀질이관찰되었으며, 이러한소견은점막하층까지확장되어있었다 (Fig. 4A, B). 환자는수술이후다발성침윤장자궁내막증으로진단되었고복통은호전되었으며, 이후 medroxyprogesterone acetate 를복용하면서특별한증상 Fig. 3. Colon study finding. A 3.2 cm sized filling defect by ext r in sic in den t a tio n is n o ted in th e lef t side of dist al descending colon (black arrow). And another smaller sized filling defect by extrinsic indentation is noted in the lateral side of the proximal ascending colon (white arrow). 없이외래추적관찰중이다. 고 자궁내막증은자궁강이외의장소에서자궁내막조직이성장하는만성적인질환이다. 8 호발부위는난소, 더글라스와, 자궁인대등의골반강이며비뇨기계, 장관, 기타폐나기관지등에서도소수발생한다. 8,9 장관을침범하는자궁내막증의빈도는약 5.3-12% 이다. 2 장침범의호발부위는직장, S상결장, 소장, 충수돌기, 맹장의순이고, 특히직장, S상결장의침범빈도는약 93% 에이른다. 4,5 국내에서는간헐적으로직장이나 S상결장에서발생한자궁내막증의예가보고되었다. 6 침윤성장자궁내막증은병변이장막표면에국한되지않고근육층, 점막하층, 점막층으로침윤되어있는상태를의미하며, 근육층에침윤한경우가 40%, 점막하층에침윤한경우는 37% 그리고점막층까지침윤한경우가 23% 이다. 10 이번증례는병변이전층을파괴하고주로근육층에침윤한경우였다. 장자궁내막증은대부분직장이나 S상결장에단일성으로침범하지만, 이번증례와같이 S상결장, 상행결장과하행결장등에다발성으로침범한경우는국내에서는아직보고된바없다. 장관에침범한자궁내막증환자의대부분은무증상이거나복부통증만을호소하는경우가많은데, 이러한경우는병변이작거나부분적으로장막표면에만침범한경우이다. 그러나넓고깊은부위로장관을침범한경우에는폐쇄, 출혈등의증상을보일수있다. 11 장자궁내막증의증상은침범한부위에따라다양하게나타 찰 Fig. 4. Pathologic findings. (A) The tissue shows endometrial glands and st r om a a cc om p an y in g f ibr o sis, w h ich is loc at ed in t h e pericolic soft tissue and muscle layer (H&E stain, 40). (B) Endometrial gland is noted (H&E stain, 400). 89

Intest Res : Vol. 5, No. 1, 2007 난다. 직장, S상결장에발생하는경우에는변의굵기와배변습관의변화, 월경주기와는뚜렷한연관성이없는혈변등이관찰되며, 2 결장에발생하는경우에는천공이나이와동반된복막염으로인한급성복증의형태로발현되기도한다. 12 이번증례에서는환자가이러한전형적인증상보다는복통과촉지되는복부종괴를호소하여쉽게진단을내리기어려웠다. 장관을침범한자궁내막증은임상적으로급성충수돌기염, 허혈성대장염, 게실염혹은종양과구분이어려워정확한진단이늦어지는경우가많고, 6,13 특히종양과의감별에주의를기울여야한다. 장자궁내막증의감별진단을위한검사방법은복부전산화단층촬영, 대장조영술, 자기공명영상, 내시경초음파이다. 복부전산화단층촬영이나대장조영술에서장자궁내막증은종괴의압박으로인한충만결손, 협착을보이고, 자기공명영상에서는 T1 강조영상의균질성의고신호강도, T2 강조영상의국소적고신호강도가포함된저신호강도의병변을보일수있다. 14 이번증례에서는전산화단층촬영에서조영이증강되는다수의종괴가관찰되었고, 대장조영술에서도상행결장과하행결장의충만결손이관찰되었다. 혈중 CA-125가때때로증가할수있으나진단에크게유용하지는않으며, 복강경을이용한진단개복술이자궁내막증을정확히감별할수있는유일한방법이다. 15 장자궁내막증은 52% 가종괴를형성하며이중 50% 이상은장벽에국한되지만, 26% 에서는광범위한점막침윤을보이는용종양자궁내막증이관찰되고, 18% 에서는발적된변연부를가지는일차성장관종양과유사한형태의궤양성점막종괴를보이는병변이관찰된다. 16 종양과의감별에도움을주는육안소견으로는뚜렷한점막의변화가없는점, 광범위한장벽침범을보이는특징적인위치의병변, 장막염증과장막협착의동반그리고장벽내작은낭종이나출혈이있으나육안소견만으로는감별이어렵기때문에조직검사를통한확진이필요하다. 그러나내시경으로얻는조직은대부분표재성이기때문에장벽의심부를침범하는침윤성장자궁내막증의경우만성적인염증만을보여줄뿐진단적인근거가부족한경우가많다. 17 이번증례에서도내시경으로얻은조직검사에서는만성염증외에는특이소견이없었고자궁내막조직은관찰되지않았다. 그후진단개복술을시행하여얻은종괴조직에서자궁내막조직의기질과선들의심부침윤이관찰되어침윤성장자궁내막증으로진단하게되었다. 최근복강경을이용한대장절제술이환자의삶의질의향상과증상의호전을가져온다는보고가나오고있 다. 18 그러나수술이후에도자궁내막증은약 20% 의재발률을보인다. 19 따라서수술치료와약물치료를병행하게되는경우도있다. 자궁내막증의약물치료로는난소기능을억제하고, 자궁내막조직의위축을일으키는경구피임제, 황체호르몬 (progestin), 안드로겐제제, 성선자극호르몬제제등을사용할수있다. 7 그러나수술후약물치료의효과에대해서는증명이되지않아아직까지는논란의여지가있다. 20 이번증례는혈변이나배변습관변화등의장자궁내막증의일반적인증상이아닌복통과복부종괴를주소로내원한환자로복부전산화단층촬영에서 S상결장, 상행결장과하행결장그리고장간막에서다수의종괴가관찰된드문예로서, 악성종양의가능성을배제하기위해시행한진단개복술을통해침윤성장자궁내막증으로진단된경우이다. 결론적으로장관에침범하는자궁내막증은임상적으로나조직학적으로감염, 허혈성대장염, 염증성장질환그리고악성종양등과감별이어려운질환이다. 가임기여성에서간헐적인하복부통증이나복부종괴를호소하면서영상진단에서는악성종양이의심되나, 내시경으로육안과조직소견이종양에합당하지않을때는침윤장자궁내막증을감별진단에포함시켜야할것으로생각한다. 색인단어 : 침윤 ; 장자궁내막증 ; 다발성복부종괴 참고문헌 1. Koninckx PR, Meuleman C, Demeyere S, Lesaffre E, Cornille FJ. Suggestive evidence that pelvic endometriosis is a progressive disease, whereas deeply infiltrating endometriosis is associated with pelvic pain. Fertil Steril 1991;55:759-765. 2. Macafee CH, Greer HL. Intestinal endometriosis. A report of 29 cases and a survey of the literature. J Obstet Gynaecol Br Emp 1960;67:539-555. 3. Weed JC, Ray JE. Endometriosis of the bowel. Obstet Gynecol 1987;69:727-730. 4. Bailey HR, Ott MT, Hartendorp P. Aggressive surgical management for advanced colorectal endometriosis. Dis Colon Rectum 1994;37:747-753. 5. Tran KT, Kuijpers HC, Willemsen WN, Bulten H. Surgical treatment of symptomatic rectosigmoid endometriosis. Eur J Surg 1996;162:139-141. 6. Lee SI, Kang JK, Lee KS, et al. Rectal endometriosis: a case report of with a clinical analysis of other cases reported in related Korean literature. Korean J Gastrointest Endosc 1999;19:667-674. 7. Sievert W, Sellin JH, Stringer CA. Pelvic endometriosis simulating colonic malignant neoplasm. Arch Intern Med 1989;149: 935-938. 90

Hyun Seok Cho, et al:a Case of Multiple Deep Infiltrating Intestinal Endometriosis Presenting as Abdominal Mass 8. Farquhar C. Endometriosis. BMJ 2007;334:249-253. 9. Schindler AE. Pathophysiology, diagnosis and treatment of endometriosis. Minerva Ginecol 2004;56:419-435. 10. Abrao MS, Podqaec S, Dias JA, et al. Deeply infiltrating endometriosis affecting the rectum and lymph nodes. Fertil Steril 2006;86:543-547. 11. Croom RD, Donovan ML, Schwesinger WH. Intestinal endometriosis. Am J Surg 1984;148:660-667. 12. Ledley GS, Shenk IM, Heit HA. Sigmoid colon perforation due to endometriosis not associated with pregnancy. Am J Gastroenterol 1988;83:1424-1426. 13. Mittal VK, Choudhury SP, Cortez JA. Endometriosis of the appendix presenting as acute appendicitis. Am J Surg 1981;142: 519-521. 14. Brosens J, Timmerman D, Starzinski-Powitz A, Brosens I. Noninvasive diagnosis of endometriosis: the role of imaging and markers. Obstet Gynecol Clin North Am 2003;30:95-114. 15. Shah M, Tager D, Feller E. Intestinal endometriosis masquerading as common digestive disorders. Arch Intern Med 1995;155: 977-980. 16. Yantiss RK, Clement PB, Young RH. Endometriosis of the intestinal tract: a study of 44 cases of a disease that may cause diverse challenges in clinical and pathologic evaluation. Am J Surg Pathol 2001;25:445-454. 17. Langlois NE, Park KG, Keenan RA. Mucosal changes in the large bowel with endometriosis: a possible cause of misdiagnosis of colitis? Hum Pathol 1994;25:1030-1034. 18. Dubernard G, Piketty M, Rouzier R, Houry S, Bazot M, Darai E. Quality of life after laparoscopic colorectal resection for endometriosis. Hum Reprod 2006;21:1243-1247. 19. Redwine DB, Wright JT. Laparoscopic treatment of complete obliteration of the cul-de-sac associated with endometriosis: longterm follow-up of en bloc resection. Fertil Steril 2001;76:358-365. 20. Yap C, Furness S, Farquhar C. Pre and post operative medical therapy for endometriosis surgery. Cochrane Database Syst Rev 2004:CD003678. 91