Abdominal pain is the most frequent complaint of liver hemangioma when the tumor is larger than 10 cm in diameter [1,2,3]. The mechanism of abdominal pain has not been clarified; however, the pressure effect and distention of the liver capsule secondary to infarcts and necrosis might cause pain in cases of giant liver hemangioma. Surgery and non-surgical treatments such as TAE, radio-frequency ablation therapy, and molecular targeting therapy have been reported to be effective to diminish abdominal pain [5]. However, non-surgical treatments are sometimes not effective to relieve symptoms for patients with giant liver hemangioma such as in the case reported here.
Liver function is normal in most cases of liver hemangioma; therefore, liver resection is considered one of the options if it is resectable. However, there are some unresectable cases due to the location of the tumors or coagulopathy. Liver transplantation should be considered at that time [4]. However, because there is a donor shortage problem while the liver function of most patients is normal, other surgical options should be considered. Some patients with polycystic liver disease (PLD) have conditions similar to those of patients with giant liver hemangioma, such as massive hepatomegaly which causes pain and compression of the adjacent organs and affects the patients’ performance status and quality of life. Recently, the treatment strategy for patients with PLD has been reported. Schnelldorfer et al. classified PLD patients with normal liver function according to clinical and radiographic findings into 4 groups, and they recommended hepatic resection with cyst fenestration for PLD if at least a single sector could be preserved [6]. In our present patient, the surgical strategy of PLD was useful because there were no previous reports of debulking of giant liver hemangioma. Therefore, we performed debulking right hemi-hepatectomy because preserving the normal liver parenchyma in segment 4 was thought to be possible based on the MRI findings and 3DCT volumetry.
Most reports of debulking surgery are for advanced ovarian cancer to improve survival after surgery. Very few reports on debulking surgery for benign tumors have been published. Debulking of a colon hemangioma with Kasabach-Meritt syndrome was reported in 1998. In that report, a 39-year-old man had a history of multiple hemangiomas since birth, with involvement of his left perineum and lower extremity, liver, spleen, descending colon, and rectum. He experienced eight episodes of major lower gastrointestinal bleeding since age 18. He underwent left colectomy because a technetium 99m pertechnetate-labeled red cell scan indicated the location of massive bleeding to be from the hemangioma of the sigmoid colon. The patient’s coagulation profile normalized 6 weeks after surgery, and he had not had any further episodes of bleeding 4 years after surgery [7]. A search of the PubMed database was conducted with the keywords “debulking” and “liver hemangioma” between 1975 and 2020. However, the search for reports of debulking of giant liver hemangioma on PubMed yielded no results.
Liver hemangioma is a benign disease; therefore, surgical intervention should be carefully considered when a tumor is increasing in size, larger than 10 cm in diameter, and symptomatic and has coagulopathy. However, liver resection is considered one of the options for a resectable liver hemangioma because short- and long-term surgical outcomes are good. Even if curative resection is not possible, debulking surgery should be considered because surgery improves symptoms and coagulopathy. However, this is a case report and there are no similar reports in the PubMed database. We need more evidence on whether debulking surgery could be acceptable for patients who cannot undergo complete resection of complicated liver hemangiomas.
