LETTERS TO THE EDITOR

 

Choledochocele complicated with pancreas carcinoma in an old patient. Endoscopical diagnosis and treatment

Coledococele complicado con carcinoma de páncreas en un paciente de edad avanzada. Diagnóstico y tratamiento endoscópico

 

 

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Key words: Choledochocele. Pancreatic adenocarcinoma. Endoscopic treatment.

Palabras clave: Coledocele. Adenocarcinoma de páncreas. Tratamiento endoscópico.

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Dear Editor,

The choledochal cyst (CC) is an unusual congenital pathology, which is usually diagnosed in childhood (1-4). According to the classification of Todani, there are 6 different types (5,6) and one of the less frequent one is type III CC or choledochocele (2% of CC) (7). Choledochocele is defined as dilation in the distal bile duct that protrudes in the duodenum, covered by normal biliary mucosa (8). In the present case, we report a new patient with choledochocele with some peculiarities which make it singular.

 

Clinical case

An 81 year old male patient was admitted in our hospital in February 2008, with non painful jaundice and anorexia of 2 weeks of evolution. His background history referred a progressive dementia, diabetes mellitus, high arterial tension, atrial fibrillation, chronic bronchitis, abdominal aortic aneurysm and biological mitral prosthesis.

In the physical exploration, it was remarkable a bad general state with jaundice drowsiness and disorientation. The temperature was 38.5 ^oC and a distended gallbladder was palpable. In the cell blood count a lekocytosis with neutrophilia was observed. In the general biochemistry the total bilirubin was 22.5 g/dl and the conjugated bilirrubin was 15.6 g/dl. Other liver enzymes were also raised: alkaline phosphatase 287 U/1 (normal < 129), GGT 472 U/1, AST 134 U/1 and ALT 227 U/1.

The abdominal ultrasonography (US) showed a dilated gallbladder with several stones and a dilatation of the intrahepatic and extrahepatic biliary tree. In the abdominal CT scan a mass of 3 cm of diameter in the pancreatic head was found, which obstructed the choledochal and the pancreatic ducts and invaded the gastroduodenal artery. The intrahepatic biliary tree was dilated with hydropic gallbladder. The diagnosis was pancreatic neoplasm with bile duct obstruction and vascular invasion. The choledochal cyst was not identified with this image technique.

In the endoscopic retrograde cholangiopancreatography (ERCP), we observed in the second duodenal portion a 3 cm diameter round formation, soft and loose to the contact with the sphincterothome. The papilar hole was identified on the top right edge. The contrast injection simultaneously opacified the cyst, the biliary tract and the Wirsung duct (Fig. 1). The pancreatic duct appeared amputated at the union between head and body. In the choledochus, at the same level of the pancreatic wound, a clear stricture was identified. The image was characteristic of a pancreatic neoplasm with choledochal stricture, associated to a choledochocele. An endoscopic sphyncterothomy was made and an 8 cm long uncovered self expandable metal stent was placed (Figs. 2 and 3).

 

The patient followed a favorable fast outcome with the disappearance of the fever and the jaundice. Obviously surgical treatment was rejected. Three months later the patient was asymptomatic and the total bilirubin was 1.5 g/dl with normal AST and ALT levels, alkaline phosphatase of 184 U/I and GGT 172 U/11. Nine months later he was still alive and has not needed a stent change.

 

Discussion

The case of choledochocele that we have presented unites it is short frequency to other unusual characteristics. The first showy thing is that in the eleven cases of CC which we have reported there are 2 type III CC. This is not the usual proportion. In the 2 most recent large series of CC report a total of 95 CC and they do not include any type III CC (9,10). The age of presentation is also uncommon. Our two patients with choledochocele are adults in a predominatly pediatric disease (3,4). The late diagnosis probably is because type III CC usually are less symptomatic than other type of CC. Some authors consider that choledochocele is a different disease that should be out of the CC's classification (9).

Another interesting point is the high malignization rate of those cysts. This risk is much lower in type III CC compared with the rest of types (8-10). When the cholangiocarcinoma appears, arises in the cyst mucosa in 70% of cases. In the remaining 30% it is originated in the biliary tree, gallbladder and pancreas (9), as occurs in this case. In both type III CC presented, ERCP was the main diagnostic and therapeutic tool. Imaging techniques, like abdominal US or CT scan, did not diagnose correctly the cyst.

 

R. Uribarrena-Amezaga, J. Fuentes-Olmo, N. Raventós-Irigoyen and R. Uribarrena-Echevarría
Department of Gastroenterology. Hospital Universitario Miguel Servet. Zaragoza, Spain

 

References

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3. Uribarrena Amezaga R, Raventós N, Fuentes J, Elías J, Tejedo V, Uribarrena Echebarría R. Diagnóstico y tratamiento de los quistes de colédoco: Presentación de10 nuevos casos. Rev Esp Enferm Dig 2008; 100: 71-5.        [ Links ]

4. Tsai MS, Lin VH, Hsu WM, Lai HS, Li PH, Chen WJ. Clinicopathological feature and surgical outcome of choledochal cysts in different age groups: the implication of surgical timing. J Gastrointest Surg 2008; 12: 2191-5.        [ Links ]

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6. Alonso-Lej F, Rever WB, Passagno DJ. Congenital choledochocele cyst, with a report of 2 and analysis of 94 cases. Surg Gynecol Obstet 1959; 108: 1-30.        [ Links ]

7. Todani T, Wanatabe Y, Narusue M, Tabuchi K, Okayuma K. Congenital bile duct cyst: classification, operative procedures, and review of thirty-seven cases including cancer arising from choledochal cyst. Am J Surg 1978; 134: 263-9.        [ Links ]

8. Kaytal D, Lees GM. Choledochal cysts: a retrospective review of 28 patients and a review of the literature. Can J Surg 1992; 35: 584-8.        [ Links ]

9. Berger A, Douard R, Landi B, Poupardin E, Canard JM, Cellier C, et al. Endoscopic management of a large choledochocele associated with choledocholitiasis. Gastroenterol Clin Biolog 2007; 31(2): 200-3.        [ Links ]

10. Visser BC, Suh I, Way LV, Kang SM. Congenital choledochal cyst in adults. Arch Surg 2004; 139: 855-60.        [ Links ]