Rationale: Hepatic epithelioid hemangioendothelioma (HEH) is a rare vascular tumor of the liver with malignant potential. It can be of solitary type, multifocal type, or diffuse type. Although there are some characteristic features on radiologic imaging, the definitive diagnosis of HEH is based on histopathology. The surgical treatment of HEH includes liver resection and transplant. Patient concerns: A middle-aged woman presented with easy fatiguability and anorexia for 1 month was found to have multifocal lesions on radiological imaging. Diagnosis: HEH was diagnosed by needle biopsy. It can be seen from imaging that this case is a multifocal form. The largest lesion increased from 3 to 3.3 cm within 2 months, with an increase of 9.45%; no other relevant literatures have been reported. Interventions: The possibility of liver transplantation was suggested to the patient. However, the patient refused transplantation and was successfully treated by radical right hepatectomy and resection of the left lobe lesion. Outcomes: She remained disease-free throughout a year follow-up period. Conclusion: HEH is a rare disease with characteristic radiological and pathological features. Although liver transplantation is the preferred treatment for multifocal HEH, surgical excision represents one alternative when the lesions can be guaranteed to be completely excised.

BACKGROUND Use of liver allograft with hepatic hemangioma after in vivo resection of hemangioma in living donor liver transplantation (LDLT) has been previously reported. However, there are few reports describing ex vivo backtable resection of hemangioma from liver allografts in LDLT. CASE SUMMARY A 55-year-old male was evaluated as a donor for an 8-month-year old patient with acute hepatic failure due to biliary atresia. Pre-operative contrast enhanced computed tomography revealed a 9 cm hemangioma in segment 4 with vascular variations in the donor. During LDLT, an intra-operative intrahepatic cholangiography was performed to ensure no variation in the anatomy of the intrahepatic bile duct. After intra-operative pathological diagnosis, ex vivo backtable resection of the hemangioma was performed and the liver allograft was transplanted into the recipient. The donor’s and recipient’s post-operative course were uneventful. At the 2-year follow-up, the liver allograft showed good regeneration without any recurrence of hemangioma. CONCLUSION Liver allografts with hemangiomas are an acceptable alternative strategy for LDLT. Ex vivo backtable resection of hemangioma from the donor liver during pediatric LDLT is safe and feasible, and can effectively reduce the operative time and intra-operative bleeding for the donor.

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