Renping Mao, Weiwei Ruan, Jianming Zhu, Li Li, Haiyan Jiang, Yanhong Li
Affiliated Women and Children’s Hospital of Ningbo University.
China
Frontiers in Pediatrics
Front Pediatr 2025; 13:
DOI: 10.3389/fped.2025.1511892
Abstract
Background: Congenital hepatic hemangioma with arteriovenous fistula (HHAVF) is a rare condition in newborns that may manifest as respiratory distress, pulmonary hypertension, and heart failure shortly after birth. This report describes a case of HHAVF complicated by encephalomalacia identified after transcatheter arterial embolization (TAE). However, the condition improved with multidisciplinary management and long-term follow-up.
Case presentation: A full-term female infant presented with a cardiac murmur and pulmonary hypertension at birth. Contrast-enhanced CT demonstrated multiple hepatic hemangiomas with high-flow arteriovenous shunting. Pulmonary hypertension resolved after TAE; however, the recurrence of hepatic hemangioma required oral propranolol therapy, which led to complete regression by 18 months of age. Postprocedural imaging identified encephalomalacia in the right frontotemporal and parietal lobes, as well as the basal ganglia, concurrent with left-limb motor impairment. Long-term rehabilitation improved left-limb function and the extent of encephalomalacia stabilized.
Conclusions: Current research primarily focuses on early cardiopulmonary complications in HHAVF, while multidisciplinary management strategies and long-term outcomes, particularly neurological manifestations, are rarely reported.
Category
Class I. Pulmonary Hypertension Associated with Congenital Cardiovascular Disease
Surgical and Catheter-mediated Interventions for Pulmonary Vascular Disease
Age Focus: Pediatric Pulmonary Vascular Disease
Fresh or Filed Publication: Fresh (PHresh). Less than 1-2 years since publication
Article Access
Free PDF File or Full Text Article Available Through PubMed or DOI: Yes