PICTURES IN DIGESTIVE PATHOLOGY

 

Biliary peritonitis secondary to spontaneous rupture of hepatic mesenchymal hamartoma

Peritonitis biliar secundaria a la perforación espontánea de hamartoma mesenquimal hepático

 

 

Rodolfo Batista-Castillo¹, Sergio González-Martínez¹, Rosa Jorba-Martin¹, Robert Memba-Ikuga¹, Fernando Mata-Sancho¹, Verónica González-Santín¹, Esperanza Bachs-Carré² and Pedro Barrios-Sánchez¹

Departments of ¹General Surgery and Digestive Diseases and ²Pathology.
Hospital de Sant Joan Despí Moisès Broggi. Consorci Sanitari Intregral. Barcelona, Spain

 

 

Introduction

The biliary hamartoma usually courses asymptomatic; we present an unusual clinical manifestation.

 

Case report

This is a 77 year old female with no medical history who presented to the emergency room with 48 hours of right hypochondrium abdominal pain, associated to conjunctive jaundice. The blood test showed elevated bilirubin and C reactive protein test. CT scan revealed gallbladder hydrops, dilated left intrahepatic bile duct, common bile duct (CBD) of 8 mm and left hepatic lobe atrophy (Fig. 1). The patient was admitted with the diagnosis of mild cholangitis and started antibiotic therapy. Cholangio-MRI showed cholelithiasis, distal choledocholithiasis with bile duct dilatation, left hepatic lobe atrophy with a dilated segmental bile duct of chronic aspect, right hepatic hypertrophy and millimeter diffuse cystic lesions (Fig. 2). Suddenly, her condition deteriorated associated with a diffuse peritonitis. She underwent a right subcostal laparotomy on emergency basis showing a choleperitoneum, normal gallbladder and the left hepatic lobe atrophy was confirmed. A perforated fibrous-cystic complex with a bile leak was found (Fig. 3). Intraoperative cholangiography illustrated the choledocholithiasis and revealed contrast leakage in the left hepatic lobe lateral segments (Fig. 4). Cholecystectomy, atrophic liver segment resection, choledochotomy with stone extraction and choledochorrhaphy over Kehr were performed as surgical treatment. She had an uneventful postoperative course. The histologic diagnosis was: Multicystic biliary hamartoma of 1.3 cm on its larger dimension, associated with acute inflammatory process (Fig. 5).

 

Discussion

The biliary mesenchymal hamartoma is a benign tumor arising from the portal triad mesenchyme. Usually presents with multiple cystic liver lesions. It is uncommon in adults. It is usually asymptomatic and it may produce local symptoms on large cysts cases. Spontaneous bile duct perforation is extremely rare and up to date we have not found any cases reported associated with biliary hamartoma. Increased CBD pressure due to impacted stones is postulated as perforation cause.

 

Recommended references

1. Ron Aguirre AC, Palpacelli MR, Serviansky TH, Silver EJ, Garciadiego Fossas P, García Merodio R. Peritonitis biliar por perforación espontánea de la vía biliar. Med Int Mex 2009;25:533-6.         [ Links ]

2. Yesim G, Gupse T, Zafer U, Ahmet A. Mesenchymal hamartoma of the liver in adulthood: Immunohistochemical profiles, clinical and histopathological features en two patients. Hepatobiliary Pancreat Surg 2005;12:502-7.         [ Links ]

3. Giunippero A, Maya AM, Gallo A, Bazzana MS, Cosentino V, Aulet F. Hamartoma mesenquimatoso hepático en un anciano. Medicina (B. Aires) 2009;69:554-6.         [ Links ]