Adult diffuse hepatic hemangiomatosis
DOI:
https://doi.org/10.4322/acr.2021.401Keywords:
Liver, Vascular Neoplasms, Hemangioma, Cavernous, PathologyAbstract
Diffuse hepatic hemangiomatosis (DHH) is an uncommon vascular lesion, though hemangiomas are the commonest benign tumors of the liver. The etiology is largely unknown to date; however, its association with giant cavernous hemangiomas (GCH) has been reported in the literature. We present herein, the case of a 37-year-old hypothyroid woman with abdominal fullness for 2 months. The contrast-enhanced computed tomography revealed multiple wellencapsulated lesions involving the liver lobes and was diagnosed as giant cavernous hemangiomas. Most of them, except the deep-seated ones, were enucleated. Histopathological examination highlighted the presence of GCH with irregular margin, replacement of hepatic parenchyma, and presence of multiple micro-hemangiomas suggesting the possibility of DHH further substantiated by retrospective radiological assessment. No extrahepatic vascular lesion was noted, and the post-operative recovery and follow-up were uneventful. Adult DHH is an uncommon entity. The diagnosis of DHH and its distinction from GCH are important from the management and prognostic point of view as recurrence, extrahepatic manifestations, features of consumption coagulopathy, and death from the complications are not uncommon.
Downloads
References
Ishak KG, Rabin L. Benign tumors of the liver. Med Clin North Am. 1975;59(4):995-1013. http://dx.doi.org/10.1016/S0025-7125(16)31998-8. PMid:167242.
Jhaveri KS, Vlachou PA, Guindi M, et al. Association of hepatic hemangiomatosis with giant cavernous hemangioma in the adult population: prevalence, imaging appearance, and relevance. AJR Am J Roentgenol. 2011;196(4):809-15. http://dx.doi.org/10.2214/AJR.09.4143. PMid:21427329.
Lehmann FS, Beglinger C, Schnabel K, Terracciano L. Progressive development of diffuse liver hemangiomatosis. J Hepatol. 1999;30(5):951-4. http://dx.doi.org/10.1016/S0168-8278(99)80152-4. PMid:10365825.
González-Nieto MI, Escobar Hoyos LA. A case of diffuse hepatic hemangiomatosis coexistent with giant hemangioma: case report and literature review. Radiol Case Rep. 2021;16(6):1518-23. http://dx.doi.org/10.1016/j.radcr.2021.03.058. PMid:33903807.
Vilgrain V, Boulos L, Vullierme MP, Denys A, Terris B, Menu Y. Imaging of atypical hemangiomas of the liver with pathologic correlation. Radiographics. 2000;20(2):379-97. http://dx.doi.org/10.1148/radiographics.20.2.g00mc01379. PMid:10715338.
He S, Chen W, Yang Y, et al. Adult diffuse hepatic hemangiomatosis: a case report and review of the literature. Clin Res Hepatol Gastroenterol. 2022;46(2):101789. http://dx.doi.org/10.1016/j.clinre.2021.101789. PMid:34384928.
Kane RC, Newman AB. Case reports: diffuse skeletal and hepatic hemangiomatosis. Calif Med. 1973;118(3):41-4. PMid:4689536.
Maeda H, Matsuo T, Nagaishi T, Ikeda T, Tomonaua Y, Mori H. Diffuse hemangiomatosis, coagulopathy and microangiopathic hemolytic anemia. Acta Pathol Jpn. 1981;31(1):135-42. http://dx.doi.org/10.1111/j.1440-1827.1981.tb00992.x. PMid:7234417.
Feurle GE. Arteriovenous shunting and cholestasis in hepatic hemangiomatosis associated with metoclopramide. Gastroenterology. 1990;99(1):258-62. http://dx.doi.org/10.1016/0016-5085(90)91256-6. PMid:2344931.
Moon WS, Yu HC, Lee JM, Kang MJ. Diffuse hepatic hemangiomatosis in an adult. J Korean Med Sci. 2000;15(4):471-4. http://dx.doi.org/10.3346/jkms.2000.15.4.471. PMid:10983701.
Kim EH, Park SY, Ihn YK, Hwang SS. Diffuse hepatic hemangiomatosis without extrahepatic involvement in an adult patient. Korean J Radiol. 2008;9(6):559-62.
Kim JD, Chang UI, Yang JM. Clinical challenges and images in GI: diffuse hepatic hemangiomatosis involving the entire liver. Gastroenterology. 2008;134(7):1830-2197. http://dx.doi.org/10.1053/j.gastro.2008.05.006. PMid:18486621.
Yoo BR, Han HY, Choi SY, Kim JH. Giant cavernous hemangioma coexistent with diffuse hepatic hemangiomatosis presenting as portal vein thrombosis and hepatic lobar atrophy. Ultrasonography. 2014;33(1):65-70. http://dx.doi.org/10.14366/usg.13003. PMid:24936497.
Ohkura Y, Hashimoto M, Lee S, Sasaki K, Matsuda M, Watanabe G. Right hepatectomy for giant cavernous hemangioma with diffuse hemangiomatosis around Glisson’s capsule. World J Gastroenterol. 2014;20(25):8312-6. http://dx.doi.org/10.3748/wjg.v20.i25.8312. PMid:25009410.
Batista A, Matos AP, Neta JO, Ramalho M. Diffuse hepatic hemangiomatosis in the adult without extrahepatic involvement: an extremely rare occurrence. J Clin Imaging Sci. 2014;4:43. http://dx.doi.org/10.4103/2156-7514.139733. PMid:25250192.
Militz M, Carlotto JR, Schmitz LD, Dal Vesco JA. Diffuse hepatic hemangiomatosis of rapid growth after bariatric surgery. Dig Liver Dis. 2016;48(12):1513. http://dx.doi.org/10.1016/j.dld.2016.06.012. PMid:27707538.
Lee JH, Yoon CJ, Kim YH, et al. Living-donor liver transplantation for giant hepatic hemangioma with diffuse hemangiomatosis in an adult: a case report. Clin Mol Hepatol. 2018;24(2):163-8. http://dx.doi.org/10.3350/cmh.2017.0002. PMid:28719965.
Shimizu Y, Komura T, Seike T, et al. A case of an elderly female with diffuse hepatic hemangiomatosis complicated with multiple organic dysfunction and Kasabach-Merritt syndrome. Clin J Gastroenterol. 2018;11(5):411-6. http://dx.doi.org/10.1007/s12328-018-0871-3. PMid:29845554.
Rao R, Naidu J, Muhammad Nawawi KN, et al. Diffuse hepatic haemangiomatosis: a case report and review of literature. Med J Malaysia. 2018;73(6):436-8. PMid:30647226.
Ota T, Kamiyama T, Kato T, et al. A rare case of cavernous hemangioma accompanied with diffuse hepatic hemangiomatosis. Surg Case Rep. 2020;6(1):251. http://dx.doi.org/10.1186/s40792-020-01023-4. PMid:33001265.
Shankar S, Rammohan A, Kulaseharan VH, Kanagavelu R, Reddy MS, Rela M. Liver transplantation for rapidly progressive giant hepatic hemangioma with diffuse hemangiomatosis. Exp Clin Transplant. 2021;19(10):1106. http://dx.doi.org/10.6002/ect.2020.0330. PMid:33535936.
Chalh O, Billah NM, Nassar I. Diffuse hepatic hemangiomatosis with giant cavernous hemangioma in an adult. J Belg Soc Radiol. 2022;106(1):17. http://dx.doi.org/10.5334/jbsr.2806. PMid:35581987.
Conter RL, Longmire WP Jr. Recurrent hepatic hemangiomas: possible association with estrogen therapy. Ann Surg. 1988;207(2):115-9. http://dx.doi.org/10.1097/00000658-198802000-00001. PMid:2829759.
Campbell V, Beckett R, Abid N, Hoey S. Resolution of consumptive hypothyroidism secondary to infantile hepatic hemangiomatosis with a combination of propranolol and levothyroxine. J Clin Res Pediatr Endocrinol. 2018;10(3):294-8. http://dx.doi.org/10.4274/jcrpe.4865. PMid:29537380.
Aslan A, Meyer Zu Vilsendorf A, Kleine M, Bredt M, Bektas H. Adult Kasabach-Merritt syndrome due to hepatic giant hemangioma. Case Rep Gastroenterol. 2009;3(3):306-12. http://dx.doi.org/10.1159/000242420. PMid:21103246.
Weimann A, Ringe B, Klempnauer J, et al. Benign liver tumors: differential diagnosis and indications for surgery. World J Surg. 1997;21(9):983-90. http://dx.doi.org/10.1007/s002689900337. PMid:9361515.
Downloads
Published
Issue
Section
License
Copyright (c) 2022 Autopsy and Case Reports

This work is licensed under a Creative Commons Attribution 4.0 International License.
Copyright
Authors of articles published by Autopsy and Case Report retain the copyright of their work without restrictions, licensing it under the Creative Commons Attribution License - CC-BY, which allows articles to be re-used and re-distributed without restriction, as long as the original work is correctly cited.