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대한내과학회지 : 제 78 권제 3 호 2010 증례 09-219 폐쇄황달을유발한석회쓸개즙 1 예 고려대학교의과대학내과학교실, 소화기연구소 윤태정 이홍식 윤석배 조혜진 최혁순 김진남 김창덕 A case of obstructive jaundice due to limy bile in the cystic and common bile ducts Tae Jung Yun, M.D., Hong Sik Lee, M.D., Seok Bae Yoon, M.D., Hye Jin Cho, M.D., Hyuk Soon Choi, M.D., Jin Nam Kim, M.D., and Chang Duck Kim, M.D. Department of Internal Medicine, Korea University College of Medicine, Institute of Digestive Disease and Nutrition, Seoul, Korea Limy bile is a rare condition characterized by excessive precipitation of calcium carbonate in the gallbladder; limy bile in the common bile duct is also very rare. Limy bile was first described by Churchman in 1911. The etiology of limy bile is unclear, but complete cystic duct obstruction by gallstones is believed to be a prerequisite for the development of limy bile. Recently, we experienced a case with obstructive jaundice in which the initial simple radiograph showed limy bile in the cystic and common bile ducts, but not the gallbladder. Here, we report the case and review the literature. (Korean J Med 78:352-356, 2010) Key Words: Limy bile; Obstructive jaundice 서 론 함께보고하는바이다. 석회쓸개즙 (limy bile) 은담낭내에끈적한반죽같은방사선비투과성물질이침전되는질환이며, 드물게총담관에도발생하는비교적드문질환이다 1). 침전되는물질은대부분이과량의탄산칼슘과 2,3) 무기성칼슘염으로단순복부촬영시담낭내에비투과성물질로관찰된다 4). 국내에석회쓸개즙에대한보고는매우드물며담낭관및총담관의석회쓸개즙으로인한폐쇄황달이보고된바는없다. 이에저자등은심와부동통과황달을주소로내원한 40세여성에서담낭관및총담관의석회쓸개즙을경험하였기에문헌고찰과 증례환자 : 여자 40세주소 : 심와부동통및황달현병력 : 평소소화불량이있던중내원 2주전갑자기심와부동통이발생하였다가자연적으로호전되었으며, 4일전부터발열, 오한과함께심와부의간헐적인산통및피부의황색변화가발생하여내원하였다. 과거력 : 특이사항없음. Received: 2009. 7. 21 Accepted: 2009. 8. 11 Correspondence to Hong Sik Lee, M.D., Department of Internal Medicine, Anam Hospital, Korea University College of Medicine, 126-1 5-ga, Anam-dong, Seongbuk-gu, Seoul 135-705, Korea E-mail: hslee60@korea.ac.kr - 352 -

- Tae Jung Yun, et al. Obstructive jaundice due to limy bile - Figure 1. The initial simple radiograph shows radiopaque material in the common bile duct. 가족력 : 특이사항없음. 사회력 : 특이사항없음. 진찰소견 : 내원당시생체징후는혈압 130/80 mmhg, 체온 36.5, 맥박 76회 /min, 호흡수 20회 /min이었으며급성병색을보였으나의식은명료하였다. 두부및경부진찰에서공막의황달소견을보였다. 흉부청진에서호흡음은정상이었고, 심잡음은들리지않았으며, 심음은규칙적이었다. 복부진찰에서는심와부압통과우상복부의압통은있었으나, 반발통이나 Murphy s sign은없었고, 종물은촉지되지않았다. 검사실소견 : 내원당시말초혈액검사에서백혈구 6,080 /mm 3, 혈색소 14.0 g/dl, 혈소판 272,000/mm 3 이었다. 혈청생화학검사에서 total protein 7.1 g/dl, albumin 4.1 g/dl, AST 349 IU/L, ALT 527 IU/L, alkaline phosphatase (ALP) 292 IU/L, GGT 370 IU/L, total bilirubin 5.8 mg/dl, direct bilirubin 2.7 mg/dl, amylase 426 IU/L, lipase 323 IU/L, serum sodium 140 meq/l, serum potassium 3.8 meq/l, calcium 9.1 mg/dl, glucose 106 mg/dl, BUN 10.2 mg/dl, Cr 0.8 mg/dl였으며, 단순뇨검사는 bilirubin +, urobilinogen ++, 적혈구 1~4/HPF, 백혈구 0~1/HPF 이었다. 방사선소견 : 단순촬영검사에서담낭으로생각되는부위에는뚜렷한액체층을보이지않았으나담낭관및담관으로생각되는부위에서방사선비투과성음영이나타났다 ( 그림 1). 복부전산화단층촬영검사에서담낭관부터총담관에걸쳐고음영물질이차있으며액체층을보였으나담낭내에서는관찰되지않았다 ( 그림 2A, 2B). 내시경역행췌담관조영술후에시행한복부초음파검사에서는담낭석이관찰되었다. 내시경역행췌담관조영술소견 : 총담관은경도의확장소견과함께내부에무정형의충만결손을보였으며, 내시경적유두괄약근절개술 (endoscopic sphincterotomy) 을시행하자총담관내부로부터백황색의액상물질이흘러나왔다 ( 그림 3A, 3B). 이틀후에시행한추적검사에서는총담관의확장소견이나백색의액상물질은보이지않았다. 내시경적제거후환자의증상및황달은호전되었다. 복강경적담낭절제술을고려하였으나, 환자가개인사정으로퇴원을원하고담낭관이완전히개통되어있어외래에서경과를지켜보기로하고퇴원하였다. 퇴원 4개월인현재특별한증상없이잘지내고있다. A Figure 2. There are calcified densities in the cystic duct (A) and common bile duct (B) on precontrast-enhanced computed tomography (CT). - 353 - B

- 대한내과학회지 : 제 78 권제 3 호통권제 595 호 2010 - A Figure 3. The endoscopic retrograde cholangiopancreatogram shows filling defects in the common bile duct (A) and limy bile gushes out after endoscopic sphincterotomy (B). B 고찰석회쓸개즙 (limy bile) 은 1911년 Churchman 5) 이담낭내용물이다량의칼슘염으로분석된흰색담즙의 1예를보고하였고, 이후 1926년에 Volkmann 6) 에의해 kalkmilchgalle 또는 milk of calcium bile 로불려지다가 1933년에 Knutsson 7) 이 limy bile 이라고명명하였다. 석회쓸개즙은드문질환이며세계적으로성인에서약 300예가보고되었고 8), 국내에서는 1971년처음보고된이래현재까지 9예가보고되었다 9-12). 거의모든환자에서담석이동반되어있고 13), 대부분담낭에만나타나며총담관까지존재하는석회쓸개즙에의해폐쇄황달이발생한경우는매우드물다 8,14,15). Sava 등 8) 에따르면담낭과총담관동시에석회쓸개즙이존재하는경우는 300예중 20예에서관찰되었으며, 일본에서도 10예만이보고되었다 15). 드물지만석회쓸개즙이저절로사라지기도한다는보고도있다 8,16-20). 국내의 9예는모두담낭내의석회쓸개즙을보이고있었으며그중에서빌리루빈의상승이동반된경우는 3예에서만관찰되었다. 일반적으로석회쓸개즙은탄산칼슘을대량으로함유하고있으며, Saito 등 21) 은 77.8% 가탄산칼슘으로이루어졌다고보고하였다. 성별및연령에따른발생빈도는여성에서남성보다약 3배정도높으며 3세이상의모든연령에서발생가능하나주로 40대에호발하는것으로알려져있다. 본증 례의경우도 40대의여자였다. 석회쓸개즙의발생기전에대해서는아직까지명확하게밝혀진것은없으나일반적으로담낭관이나담낭경부의협착이나폐쇄로인한담즙정체가선행하는것으로생각된다 1,4). 생화학적변화에필요한기전이나시간은잘알려져있지않지만, 담낭내정체된정상적인담즙에탄산칼슘침전이발생하여석회쓸개즙이발생하는것으로생각된다. 또한 McWhirter 등 22) 은칼슘의침착을위해서는경도의담낭염이필요하다고하였다. 그리고 Green 14) 은담낭의염증과더불어담낭관의폐쇄가담즙의알칼리화를일으키고산도가 6.6 이상일경우탄산칼슘의결정화가일어난다고보고하였으며, 혈청및소변의증가된칼슘수치로보아칼슘대사의이상도석회쓸개즙의발생에기여할것으로보고하였다. Takatori 등 23) 의예에서와같이혈청칼슘과부갑상샘호르몬의수치가증가한칼슘대사의이상에관련한다는보고도있다. 우연히단순복부촬영으로진단된경우를제외하고, 대부분의석회쓸개즙환자의증상은담도계결석증과유사하다. 급성담낭염, 췌장염, 폐쇄황달같은합병증을유발할수있으나, 증상이나합병증은석회쓸개즙때문에발생하는것이아니라관련된담도계질병에의한것으로보고되기도한다 4,24). 단순복부촬영에서담낭의일부또는전체에방사선비투 - 354 -

- 윤태정외 6 인. 폐쇄황달을유발한석회쓸개즙 1 예 - 과성음영이보이며체위변동에따라위치가변화하고, 내용물이중력방향에따라이동하는것이특징적이다. 복부전산화단층촬영은석회쓸개즙과같이석회화병리를가지며전암성병변인자기모양의담낭 (porcelain gallbladder) 과감별하는데도움을준다. 본증례의경우단순촬영검사및복부전산화단층촬영에서흔히발견되는부위인담낭내에서는석회쓸개즙이보이지않고, 담낭관및총담관에만존재하였으며, 내시경적유두괄약근절개술후시행한복부초음파및추적내시경역행췌담관조영술에서담낭석이발견되었다. 이는담낭석이담낭관이나담낭경부의협착또는폐쇄를유발하여담낭내석회쓸개즙이발생했다가, 담낭관의완전한재소통이이뤄지면서석회쓸개즙이담낭밖으로흘러내려가담낭관과총담관에만존재하여폐쇄황달을일으킨것으로추정된다. 내시경적으로총담관의석회쓸개즙을제거후추적내시경역행췌담관조영술을통해담낭관내석회쓸개즙도장내로유출된것을확인하였다. 본증례의의의는대부분의석회쓸개즙은담낭관이폐쇄된상태에서발견되어담낭절제술이필요하지만담낭관이완전히재개통된경우에는총담관내의석회쓸개즙만해결해주면담낭절제술이반드시필요하지는않다는가능성을제시해준다. 향후폐쇄황달을유발한총담관내석회쓸개즙을치료후담낭석은있으나담낭관은소통되어있고담낭의염증이없는경우수술적치료없이경과를지켜보는것도하나의치료방법으로고려해야한다. 요 저자들은심와부동통과황달을주소로내원한 40세여자에서단순촬영및복부전산화단층촬영을통해석회쓸개즙을진단하였으며, 내시경역행췌담관조영술을통하여폐쇄황달을유발한담낭관및총담관의석회쓸개즙을치료후경과관찰중으로, 담낭관이소통된경우수술적치료없이경과관찰하는것도하나의치료방법이될수있겠다. 약 중심단어 : 석회쓸개즙 ; 폐쇄황달 REFERENCES 1) Naryshkin S, Trotman BW, Raffensperger EC. Milk of calcium bile: evidence that gallbladder stasis is a key factor. Dig Dis Sci 32:1051-1055, 1987 2) Phemister DB, Rewbridge AG, Rudisill H. Calcium carbonate gall-stones and calcification of the gall-bladder following cystic-duct obstruction. Ann Surg 94:493-516, 1931 3) Saito K, Hatafuku T, Kanno S, Hirata Y, Seta K, Nakadate K. Review of five cases of limy bile: particularly on radiological, chemical and crystallographic studies. Gastroenterol Jpn 14:135-160, 1979 4) Moreaux J, Roux JM. Limy bile: a surgical experience in 16 patients. Gastroenterol Clin Biol 18:550-555, 1994 5) Churchman JW. Acute cholecystitis with large amounts of calcium soap in the gall-bladder. Bull Johns Hopkins Hosp 22:223-224, 1911 6) Volkmann J. Uber kalkmilchgalle. Munch Med Wochenschr 73:2014-2015, 1926 7) Knutsson F. On limy bile. Acta Radiol 14:453-462, 1933 8) Sava G, Millot P, Becmeur F, Vaxman F, Grenier JF. Limy bile syndrome: study of a case with double localization in the gallbladder and common bile duct. Gastroenterol Clin Biol 12:156-159, 1988 9) Paik SJ, Park HG, Park IS, Choi HJ. A case of milk of calcium bile. Korean J Gastroenterol 4:19-26, 1972 10) Song KJ, Jeon IS, Du CJ, Kim UB, Byun JH, Choi BS, Shin HJ. A case of hourglass gallbladder associated with milk of calcium bile at the fundal compartment. Korean J Med 35:574-579, 1988 11) Hwang GY, Kim A, Yoon SJ, Jung SH, Ko BS, Yang HW, John BM. A case of limy bile associated with cholangitis and calcified gallbladder. Korean J Gastrointest Endosc 31:58-61, 2005 12) Jung JH, Kang YS, Kim YS, Kim SH, Lee BS, Lee HY, Kang DY. A case of limy bile accompanied with gallbladder stones and common bile duct stone. Korean J Med 70:701-705, 2006 13) Kim JB, Chang YJ, Baek IH, Jung SW, Lee MS. Hepatobiliary and pancreatic: limy bile. J Gastroenterol Hepatol 21:1350, 2006 14) Green NA. A case of limy bile causing obstructive jaundice. Br J Surg 47:222-225, 1959 15) Tsukamoto T, Ohta Y, Shuto T, Hirohashi K, Kubo S, Tanaka H, Takemura S, Kinoshita H. Limy bile: a case of obstructive jaundice and review of 26 cases. Osaka City Med J 49:67-70, 2003 16) Schwartz A, Feuchtwanger M. Radiographic demonstration of spontaneous disappearance of limy bile. Gastroenterology 40: 809-812, 1961 17) Marquis JR, Densler J. The disappearing limy bile syndrome. Radiology 94:311-312, 1970 18) Holden WS, Turner MJ. Disappearing limy bile. Clin Radiol 23:500-507, 1972 19) Nomura F, Suzuki Y, Suzuki K, Yamamoto K, Ohshima H, Nakayama T, Yokosuka O, Ohnishi K, Saisho H, Ohhara K, Okuda K. Spontaneous disappearance of limy bile: report of a case with review of the literature. Am J Gastroenterol 79:884-888, 1984 20) Levy I, Lantsberg L, Khoda J. Spontaneous disappearance of limy bile: a cause of acute pancreatitis? J Clin Gastroenterol 18:220- - 355 -

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