Critical infantile hepatic hemangioma: results of a nationwide survey by the Japanese Infantile Hepatic Hemangioma Study Group

J Pediatr Surg. 2011 Dec;46(12):2239-43. doi: 10.1016/j.jpedsurg.2011.09.007.

Abstract

Background: The current survey aimed to describe the clinical features of critical infantile hepatic hemangioma (IHH) and the implications of recent treatments.

Materials and methods: A nationwide survey of critical IHH patients treated between 2005 and 2010 was performed in all 117 registered pediatric surgical hospitals in Japan. As a result, 19 patients were identified and reviewed using a statistical analysis.

Results: Abdominal distention (47.4%), high-output cardiac failure (47.4%), coagulopathy (42.1%), and respiratory distress (31.6%) were the major symptoms. Three patients died (1 of coagulopathy, 1 of cardiac failure, and 1 of both). An accompanying portovenous shunt was also highlighted. Infantile hepatic hemangioma was totally insensitive to steroid treatment in 3 (23.1%) of the 13 patients, and 9 (47.4%) of the 19 patients required other treatments. Surgical resection and β-blocker improved the hematologic data, whereas hepatic arterial ligation and embolization seemed to produce a limited effect. Among the dead patients, several hematologic parameters were significantly worse: the thrombocyte count (pretherapeutic: 73,000 vs 300,000/mm(3), dead vs survivor, respectively [P < .03]; posttherapeutic: 66,000 vs 388,700/mm(3) [P < .003]) and the prothrombin time (posttherapeutic, 35.0 vs 12.1 seconds [P < .0001], dead vs survivor, respectively).

Conclusion: For critical IHH cases with steroid-insensitive hematologic disorders, alternative treatments including β-blocker therapy, surgery, and liver transplantation should be considered.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adrenergic beta-Antagonists / therapeutic use
  • Antineoplastic Agents / therapeutic use
  • Blood Coagulation Disorders / etiology
  • Cardiac Output, High / etiology
  • Critical Illness
  • Drug Resistance, Neoplasm
  • Embolization, Therapeutic
  • Hemangioendothelioma / complications
  • Hemangioendothelioma / drug therapy
  • Hemangioendothelioma / epidemiology
  • Hemangioendothelioma / surgery
  • Hemangioma / complications
  • Hemangioma / drug therapy
  • Hemangioma / epidemiology*
  • Hemangioma / surgery
  • Hemangioma, Cavernous / complications
  • Hemangioma, Cavernous / drug therapy
  • Hemangioma, Cavernous / epidemiology
  • Hemangioma, Cavernous / surgery
  • Hepatectomy
  • Hepatic Artery
  • Humans
  • Japan / epidemiology
  • Ligation
  • Liver Failure / etiology
  • Liver Neoplasms / complications
  • Liver Neoplasms / drug therapy
  • Liver Neoplasms / epidemiology*
  • Liver Neoplasms / surgery
  • Liver Transplantation
  • Prednisolone / therapeutic use
  • Respiratory Insufficiency / etiology

Substances

  • Adrenergic beta-Antagonists
  • Antineoplastic Agents
  • Prednisolone