A Rare Case of a Common Hepatic Duct Stricture Secondary to an Anteriorly Crossing Right Hepatic Artery in an Infant.

A 1-year-old male child presented with progressive jaundice. Investigations were suggestive of an obstructive pathology with a suspected choledochal cyst on imaging. Intraoperative cholangiogram demonstrated runoff of contrast from the cystic duct into the common bile duct with no opacification of the biliary system proximal to this. Intraoperatively, the right hepatic artery was found anteriorly crossing the common hepatic duct (CHD) causing extrinsic compression leading to complete obstruction. Following stricture excision and anastomosis of the dilated bulbous CHD to a Roux-en-Y jejunal loop, the child recovered completely. An anteriorly crossing right hepatic artery causing obstruction to the biliary duct is a rare occurrence, more so in infancy. An excision with a hepaticojejunostomy is straight forward and curative.

CASE REPORT

A 1-year-old male child presented with history of progressive jaundice for 3 months. He had history of passing high-colored urine but did not have clay-colored stools, itching, or any bleeding tendencies. There was no history of intermittent episodes of fever, vomiting, or excessive crying. He had been evaluated elsewhere and was told to have a choledochal cyst and was hence referred to us for further management.

On admission, his bleeding parameters and liver function tests were deranged (total bilirubin – 10.3 mg/dl, direct bilirubin – 6.6 mg/dl, alanine transaminase – 227, aspartate transaminase – 131, alkaline phosphatase – 845, prothrombin time/international normalized ratio – 62.6/5.4, and activated plasma thromboplastin time – 92.7). Following parenteral Vitamin K for 3 days, his bleeding parameters were normalized and he was then taken up for surgery. His preoperative ultrasonography (USG) abdomen showed a dilated common bile duct (CBD) measuring 12 mm with an abrupt cutoff and bilobar intrahepatic biliary radical dilation; hence, we questioned the diagnosis of a choledochal cyst.

A peroperative cholangiogram demonstrated free flow of dye into the duodenum with a normal CBD beyond the cystic duct. The proximal ducts however could not be visualized [Figure 1]. On proceeding with dissection of the porta, the right hepatic artery was found to be crossing the lower end of the common hepatic duct (CHD) anteriorly [Figure 2]. On lifting the artery, a stricture was found in the CHD just proximal to the cystic duct, CBD junction. The duct proximal to this was grossly dilated, and following division, distal bile flow could not be established [Figure 3]. There were no telltale signs of any previous inflammation.

Figure 1

Intraoperative cholangiogram demonstrating a) Free flow of dye into the duodenum b) Abrupt cutoff of the duct proximal to the cystic duct (arrowhead)

Figure 2

Perioperative image showing H: Dilated common hepatic duct, A: Anteriorly crossing right hepatic artery, C: Cystic duct lying parallel and inferior to the artery, G: Gallbladder

Figure 3

Cystic duct leading to the common bile duct after division from the strictured hepatic duct without drainage of bile

The duct was excised down to its distal end along with the gallbladder, and a Roux-en-Y hepaticojejunostomy was performed anterior to the crossing vessel. Postoperatively, the child made an uneventful recovery, with complete resolution of jaundice. He is now well and on regular follow-up.

DISCUSSION

Strictures of the extrahepatic biliary system in the pediatric population are quite unusual. The most common surgical etiologies for pediatric obstructive jaundice are biliary atresia or an obstructed choledochal cyst. Other less common entities include choledocholithiasis, biliary stricture secondary to primary sclerosing cholangitis, trauma, operative procedures on or near the biliary tract, and cystic fibrosis.[12]

Until recent years, diagnosis and management of these conditions have employed USG, radionuclide imaging, and computed tomography scan followed by surgery. Hancock et al. reported a CBD stricture, just distal to the cystic duct, in a 3-week-old child operated for gastroschisis.[3] She was diagnosed by USG and percutaneous transhepatic cholangiography and later underwent successful decompression of the biliary tract with percutaneous transhepatic biliary drainage through a stent and multiple episodes of dilation.

George Holcomb in 1980 described bile duct strictures in 8 children, 6 of which involved the distal CBD due to chronic pancreatitis.[1] Two involved the CHD and both children presented with jaundice alone and no signs of pancreatitis. They both ultimately underwent a Roux-en-Y choledochojejunostomy and are well. Ju et al. described two adults with biliary compression by an anteriorly crossing right hepatic artery, successfully managed with suture fixation between the gallbladder and the causative artery.[4] Honma et al. reported a right hepatic artery traveling anterior to the CBD in a cadaver of 91-year-old male;[5] however, no such pediatric reports have been described to date.

CONCLUSION

CHD stricture secondary to an anteriorly crossing right hepatic artery is a very unusual cause of obstructive jaundice. We believe this to be the first reported case in an infant. Excision of the distal CBD and a Roux-en-Y enterostomy to the dilated CHD is curative.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.