After orthotopic liver transplantation, splenic artery syndrome (SAS), a phenomenon by which the main blood flow of the impaired hepatic artery is shifted to the splenic artery or gastroduodenal artery despite the absence of a structural lesion involving the anastomosis, has occasionally been observed. We report a 20-year-old women who developed SAS with pancytopenia and refractory ascites after living donor liver transplantation despite intraoperative ligation of the splenic artery as a prophylactic treatment for SAS. In this case SAS was diagnosed by digital subtraction angiography (DSA). A celiac trunk angiogram showed relative hypoperfusion of the hepatic artery together with augmentation of the blood flow toward the spleen with the unique collateral circulation through the left gastric artery, stomach and short gastric artery, and distal splenic artery. Embolization of one of the two left gastric arteries was performed. After embolization the hepatic artery perfusion showed significant improvement, but reduced again the next day. We ultimately conducted splenectomy. This case showed portal hyperperfusion and portal hypertension, consistent with previous reports that have described an association of SAS with portal hyperperfusion. After splenectomy, there was significant improvement in the hepatic artery perfusion, ascites disappeared promptly, and pancytopenia was significantly improved.