Yotsuyanagi's theory is often quoted in the discussion of the etiology of congenital dilatation of the biliary duct (CDBD), but there has been no established etiology as yet. With particular reference to the relationship between anomalous arrangement of the pancreaticobiliary ducts and CDBD, Babbit collected 19 cases of CDBD. The authors also observed the anomalous arrangement in 10 of 24 cases of CDBD (42 percent). Analysis of 570 CDBD cases collected in Japan disclosed that the anomalous arrangement was present in 183 of them (33 percent), dilation of the intraheptic bile ducts in 236 (42 percent), and narrowing and malformation of the bile duct in the hepatic hilum in 58 (10 percent). The authors have proposed a new classification, including these morphologic anomalies of the whole pancreaticobiliary ductal system. This classification provides useful indices in selecting the surgical procedures. The effect of the anomalous arrangement was pathophysiologically studied on dogs. The static internal pressure of the dog pancreatic duct was 14.6 + or - 5.5 cm H2O, and that of the dog bile duct, 8.9 + or - 3.4 cm H2O. Puppies were then operated on by shunting the pancreaticobiliary ducts. As the result, the biliary tract was devastated by pancreatic juice, giving rise to similar pathologic changes to those clinically observed.

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Histochemical and immunohistochemical studies on 47 consecutive specimens excised for choledochal cyst were performed to clarify possible metaplastic changes of the biliary duct in relation to carcinogenesis. An anomalous arrangement of the pancreaticobiliary ductal system was observed in all 39 cases examined. Among the 47 patients, 5 (10.6 per cent) had biliary carcinoma. 27.3 per cent mucous gland, 13.0 per cent goblet cell and 9.5 per cent argyrophil cell in 23 children. On the other hand, 81.8 per cent exhibited mucous gland, 41.7 per cent goblet cell and 27.3 per cent argyrophil cell in 24 adults. These metaplastic changes seemed to be an intestinal metaplasia and increased with age. Immunoreactive-gastrin or -somatostatin were evident immunohistochemically in 4 adults. These findings confirmed that intestinal metaplasia may develop in the biliary duct in cases of choledochal cyst. Although direct evidence between intestinal metaplasia and the development of biliary carcinoma was not found, reflux and stasis of pancreatic enzymes in the biliary duct may relate to the development of intestinal metaplasia and be an important factor related to the carcinogenesis of choledochal cyst.

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