A rare case of cavernous hemangioma accompanied with diffuse hepatic hemangiomatosis

Abstract

Background Hepatic cavernous hemangioma (CH) is the most common hepatic benign tumor. Most cases are solitary, asymptomatic, and found incidentally. In symptomatic cases with rapidly growing tumors and coagulopathy, surgical treatment is considered. In rare cases, diffuse hepatic hemangiomatosis (DHH) is reported as a comorbidity. The etiology of DHH is unknown. Case presentation A 29-year-old female patient had a history of endometriosis treated with oral contraceptives. Hepatic CH was incidentally detected in the segment IVa of the liver according to the Couinaud classification. Follow-up computed tomography (CT) and ultrasound sonography showed the growth of the lesion and formation of multiple new lesions near the first. Enhanced CT and magnetic resonance imaging (MRI) revealed that the new lesions were different from CH. Although oral contraceptives were stopped, all lesions grew in size. Malignancy and possibility of rupture of these tumors were considered due to the clinical course, and we opted for surgical removal of the tumors. Left liver lobectomy and cholecystectomy were performed. Surgical findings were small red spot spreading and a mass in segment IV of the liver. Pathological examination revealed a circumscribed sponge-like tumor with diffuse irregular extension to the adjacent area. Both of the lesions consisted of blood-filled dilated vascular spaces lined by flat endothelium without atypia. The diagnosis was hepatic CH with DHH. The patient was discharged on postoperative day 12 uneventfully. Conclusion We report the successful resection of CH with DHH. The case findings suggest a relationship between oral contraceptive use and enlargement of CH and DHH. Although DHH has been poorly understood, a few previously published cases reported DHH occurrence in patients using oral contraceptives. In such cases, the decision to perform surgical resection should be made after careful examination.

Adult diffuse hepatic hemangiomatosis

“…2,3 The association of adult DHH with oral contraceptive (OC) and metoclopramide has been reported. 20,23 We could not document any history of OC or metoclopramide intake in our case. The association of DHH and hypothyroidism is known in infants, although no such association is documented in adult DHH.…”

“…17,18,21 Besides, cessation of drugs like OC/ metoclopramide may be of help. 9,20 The prognosis of DHH is uncertain due to its rarity. Liver failure is the most common complication followed by Kasabach-Merritt syndrome, disseminated intravascular coagulation (DIC), and heart or multiple organ dysfunction.…”

“…A PubMed search till May, 2022 along with the literature review by He et al 6 showed only 19 reported cases of adult DHH depicted in the English literature. 3,4,[6][7][8][9][10][11][12][13][14][15][16][17][18][19][20][21][22] Approximately half of the DHH cases can be seen in association with GCH. 2 The etiopathogenesis of DHH is uncertain, although it has been diagnosed in association with hereditary disorders, such as skeletal hemangiomatosis and Osler-Weber-Rendu disease, and drugs like oral contraceptives (OC) and metoclopramide.…”

“…2 The etiopathogenesis of DHH is uncertain, although it has been diagnosed in association with hereditary disorders, such as skeletal hemangiomatosis and Osler-Weber-Rendu disease, and drugs like oral contraceptives (OC) and metoclopramide. 9,20,23 The final diagnosis is histopathological, as imaging tests are not effective in accurately differentiating their differential diagnoses. The clinical case presented here serves as an example to corroborate this point.…”

Liver segmentation in CT imaging with enhanced mask region-based convolutional neural networks

“…Liver segmentation in medical imaging data is of great clinical significance for lesion resections and liver transplantations (1)(2)(3). In addition, accurate liver segmentation offers a substantial aid to physicians in diagnosing and treating liver diseases (4)(5)(6). However, manual liver segmentation…”