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Aderomyoma of the Common Bile Duct --Report of a Case

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Aderomyoma of the Common Bile Duct --Report of a Case Yamanashl Med. J. 4 (2), 83"v87, 1989 Case Report AdeRomyoma of the Common Bile Duct --Report of a Case Yoshiro MATsuMoTe, Masatoshi MoGAKi, Hidehisa AoyAMA, Takayoshi SEKmAwA, Katsnhiko SuGAHARA, Koichi SuDAi), and Masayuki FuJiNo2) DePa,rt･ment of Surge7pu. i)DePartment of Pathology, 2)DePartment of lnte7"nal Medicine, YamanasJzi Medical Coglege, Tamaho, Nakakoma, Ya?nanashi 409-38, JaPan Abstract: Adenomyomas in the extrahepatic biie duc£ are extremely rare. In a 75-year- old male with acute cholangitis due to adenomyoma £erming a protruding lesion iR the terminal bile duct, pamacreatoduodenectomy was carried out, resulting complete cure, Key words: Adenomyoma of the common bile duct, Early bile dact cancer, Obstructive jaundice formed, aRd £rom the histologic examina- INTRODUCTION tion of the surgical specimen, adenomyoma Adenomyoma in the biliary ductal system of the common bile duct was confirmed. is most freguently fouRd in the gallbladder. Although beRign neoplasma o£ the bile The gallbiadder wall is more abuRdant in duct system are uncommon, the tumors are muscle fibers than is the wall o£ the bile clinically very important because they can duct. Adenomyoma in the gallbladder is cause obstructive jaundice4) and require knowlt to be closely re}ated to the forma- differentiation £rom eary cancer of the bile tion of gallstones. In other parts of the duct. biliary ductal system, kex4xeve]-, adeno- We report here the surgical results and myoma is very rarei)･2), although a few pathologic findings in a case of adeno- cases of a tumor arising from the papil}a myoma of the terminal bile duct. of Vater3) have beelt reported. CAsE Mle encountered a patient wi£h severe acute cholangitis with a 10mm diameter On December 10, l983, a 75-year-old man shado'{4r defect on the vvTall of the terminal had a sudden onset of severe right hypo- bile duct demoRstrated by percutaneous chendralgia, high fever of 400C and chilis transhepatic cholaRgiography (PTC) and after supper. Medication was provided and eRdoscopic retrograde cholangiopancreato- tkese symptoms subsided withilt two days, graphy (ERCP). As early cax{cer of the bile and subsequently, he was admitted to Hos- duct was suspected from the radiologic pital of Yamanashi Medical College. He fiRdings, paRcreatoduodenectomy was pe" was 146 cm tall, weighed 47 kg and had no past history of abdomiital pain or jaundice. Receive(I February i6, l989 Accepted March 28, 1989 Labo?"at'o7z>) findings: Laboratory findings 84 Y. Matsumoto et al. on admission to our hospital were as fol- fewpa-. x ,ff lows: T. Bili. was O.6mg/dl, D. Bili O.2 mg/dl, Alp 228 mlU, LAP 54 mlU, r-GTP lpiii(5 5fstitifgiag" '-YT"sf.e.,.J"';iEiL :, 157mlU, GOT 29mlU, GPT 29mlU, LDH 801 mlU, s-Amylase 150 U (Somogyi), RBC 481×10. Slight increase in Alp and r-GTP levels were observed. CholangiogTaf)hy: Drip infusion cholan- giography (DIC) disclosed a slightly dilated extrahepatic bile duct and a shadow defect ,1･)giili g in the terminal region (Fig. 1). ERCP showed no evidence of tumor, ulcer or paasgww liigeBi ¥'Iil'""i:tl.-, 't./., es..ee Fig. 2. .A k' A 10mm diameter circular elevated ttL'' lesion is demonstrated in the terminal bile duct. was larger than the right lobe, but no ab- normal density area was confirmed. Slightly prolonged excretion of the radionuclide into the liver, the intrahepatic bile ducts, the common bile duct, and the duodenum was demonstrated using a 99mTc-HIDA hepatobiliary scintigram. Fig. 1, DIC. A shadow defect is shown in the OPevation: December 4,1984 laparotomy terminal bile duct. was performed. Neither ascites nor ad- hesions were shown in the abdominal bleeding in the papilla of Vater, but re- cavity. Both the left and the right lobes vealed a circular protruding lesion in the of the liver were normal in color, surface terminal bile duct. No change was ob- texture and hardness, with the left lobe served in the main pancreatic duct (Fig. 2). larger than the right lobe, as visualized by PTC showed no dilatation in the intra- CT. There were no inflammatory changes hepatic bile duct, but a moderately dilated in the gallbladder or in the wall of the extrahepatic bile duct was demonstrated. extrahepatic bile duct. The common bile The location of this shadow defect was not duct was incised, and a endoscopic exami- altered by the changes in the body position, nation of the biliary ductal system was suggesting the presence of a lesion in the performed, revealing a flat semicircular biliary duct wall (Fig. 8). In computed protruding lesion on the wall of the termi- tomography (CT), the left lobe of the liver nal common duct. The surface of the Adenomyoma of the terminal bile duct 85 g 8ag Z me.kij ,X'k.': g" '' '' i;'ts i"" ･'e 5.'.,'' "''i ,S' b ,, t. ,･1z""'i'S'i-vttw' '" ',,,,t'"t.g-;(£ii, tt ii i,.. ,ti ep' ,.fr .. s,sxiiili ss, it y,ts>i, Fig. 3. PTC. Moderately dilated extrahepatic bile duct and shadow defect in the terminal bile duct are revealed and the Fig. 4. Marked thickening (arrow) of the mu- location of the lesion did not vary with cosa of the terminal bile duct. (H.E. changes in body position. stain, ×20) Iesion was red and granular, but no bleed- liculi. In the interstitium of adenomatous mg. was apparent. lesion, hyperplastic smooth muscles were Although biopsy failed to show ma- sporadically observed around the canaliculi. lignancy of the tumor in the terminal From these findings, the lesion was diag- bile duct, pancreatoduodenectomy was per- nosed as adenomyoma or adenomyomatosis formed, as we could not exclude early (Fig. 4 and 5). Microscopic examination cancer of the bile duct. The postoperative did no/t show adenomyoma in the ampulla course was uneventful, and he was dis- of Vater or in the gallbladder. charged from the hospital in the fourth DIscussloN postoperative week and has been healthy during the past four years. In the literatures, there were 33 cases Pathologic findings of the sPecimen: The of epithelial tumors including papillomas, wall of the terminal common bile duct was polyps, adenomas and cystadenomas; four thickened to 2 mm, and a protruding lesion cases of lipoma, three cases of xanthomas, measuring 10×5×5 mm was present on the one case of myxoma, and five cases of mixed wall of the common bile duct 2mm from tumors (adenofibroma, adenomyofibroma, the orifice of the ampulla of Vater. His- adenomyoma and myoadenoma)i),2)･5),6). In tologically, the elevated lesion of biliary recent years, owing to the widespread use ductal wall had adenomatous features, pre- of ERCP, it has been possible to make a senting lobular arrangement of the cana- diagnosis before surgery in an increasing Matsumoto et al. disposed scattered epithelial glandular ele- ment, similar in appearance to those found in the solid type." ss,.,,zlkee'ee In cases of adenomyoma, the tumor may occasionally appear as a diffuse thickening of the duct wall. However, the tumor mass which grows gradually in the lumen of the bile duct usually causes abdominal pain and obstructive jaundice, and, as seen in this case, the lesion arising in the portion near the terminal bile duct may cause bac- terial infection, presenting attacks of acute cholangitis. In cases with appearances of these symp- toms, the elevated lesion on the wall of the bile duct can be easily detected by direct cholangiography such as ERCP and PTC, but it will be extremely diMcult to differ- entiate the lesion from early bile duct cancer. In particular, it is not easy to con- firm before or during surgery that there are Fig. 5. High power view of Fig. 4. The mucosal no malignant changes in the elevated lesion thickening consisting of glandular hyper- in the intrapancreatic portion of the bile plasia in combination with either muscle or fibrous connective tissue. (H,E, stain, duct. For these reasons, we believe that ×1oo) when a protruding lesion in the terminal bile duct has been detected, pancreatoduo- number of cases7). However, very few cases denectomy must be carried out for success- of adenomyoma arising in the extrahepatic ful removal of the tumor. bile duct except for the gallbladder and the papilla of Vater have been reported. REFERENC,ES According to Dowdy's tota12), only one out 1) Chu PT.: Benign neoplasma of the extrahepatic of 16 cases of adenoma, was an adeno- biliary ducts --review of the literature and re- myoma in the Ieft hepatic duct. Subse- port of a case of fibroma. Arch Pathol 1950; 50: quently classified adenomas of the biliary 8<F97. 2) Dowdy GS, Orin WG, Shelton GL, Waldron duct system into three typesi): the solid GW.: Benign tumors of the extrahepatic bile type, cystic type and mixed type. Adeno- ducts-report of three cases and review of the myomas were grouped in the mixed type, literature. Arch Surg 1962; 85: 508-513. i.e., "the mixed type includes tumors of 8) Oberhofer B, Mlinaric I, Soljan N, Knezevic- glandular elements in combination with Krivak S.: Adenomyoma papillae Vateri. Act Chir Jugo 1975; 22: 15-21. either muscle or fibrous connective tissue, 4) Steenbergen WV, Ponette E, Marchal G, Van- and appears as a variously sized, firm and neste A, Geboes K, Rijkel JP, Fevery J Groote well circumscribed tumor within the wall JD.: Cystadenoma of the common bile duct of the bile duct. Histologically, the tumor demonstrated by endiscopic retrograde changio- graphy: an uncommon cause of extrahepatic consists of abundant muscle of fibrous obstruction. Am J Gastroenterol 1984; 79F: 466- connective tissue elements in which are 470, Adenomyoma of the termiiial bile duct 87 5> Moore SW, Mc Elwee RS, Romiti C.: Benign 22-27.
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