Published online Jul 7, 2013. doi: 10.3748/wjg.v19.i25.4094
Revised: May 5, 2013
Accepted: May 16, 2013
Published online: July 7, 2013
Primary hepatic leiomyoma is a neoplasm of mesenchymal origin and occurs only rarely. Secondary to benign smooth muscle proliferation, it is usually found in adult women and is associated with Epstein-Barr virus (EBV) infection. Here, we report the 29th case of primary hepatic leiomyoma with its unique features related to diagnosis, treatment and developmental biology. A 48-year-old man, with an immunocompromised status, complained of pain in the upper quadrant of the abdomen. Serological analysis indicated no presence of hepatitis virus, no human immunodeficiency virus, and no EBV infection. The levels of α-fetoprotein and carcinoembryonic antigen were normal. A mass was detected in segment III of the hepatic lobe by ultrasonography and an abdominal computed tomography scan. Endoscopy had negative findings. Exploratory laparotomy found no existing extrahepatic tumor and left lateral lobectomy was performed. Pathological examination showed the mass to be a typical leiomyoma. The cells were positive for α-smooth muscle actin and desmin, and negative for the makers of gastrointestinal stromal tumor (GIST), including CD117, CD34 and DOG1 (discovered on GIST1). In situ hybridization revealed negative status for EBV-encoded small RNA. After left lateral lobectomy, the patient was not given chemotherapy or radiotherapy. During a 2-year follow-up, no sign of local recurrence or distant metastasis was observed. In conclusion, we report a rare case of primary hepatic leiomyoma in a male patient without EBV infection. Hepatic resection was curative. This case presents data to expand our knowledge concerning the complex and heterogeneous nature of primary liver leiomyoma, indicating that EBV infection is important but neither necessary nor sufficient for the development of primary liver leiomyoma.
Core tip: Primary hepatic leiomyoma is usually found in adult women and is associated with Epstein-Barr virus (EBV) infection. We report the 29th case worldwide in a 48-year-old kidney allograft recipient without EBV infection and extrahepatic tumor. He achieved clinical cure by mass resection. The leiomyoma was positive for α-smooth muscle actin and desmin, and negative for gastrointestinal stromal tumor markers, including CD117, CD34 and DOG1 (discovered on gastrointestinal stromal tumor 1). The tumor was negative for EBV-encoded small RNA. The data indicate that EBV infection is important but neither necessary nor sufficient for development of primary liver leiomyoma.