• Seung Hyung Han, Kyung Duk Park, and Soon Chul Kim.
• Department of Pediatrics, Jeonbuk National University Medical School and Hospital, Research Institute of Clinical Medicine of Jeonbuk National University - Biomedical research Institute of Jeonbuk National University Hospital, Jeonju, Korea.
• Medicine (Baltimore). 2021 Dec 3; 100 (48): e28105.

RationaleHepatic nodular regenerative hyperplasia (NRH), a nonspecific change in the liver parenchyma, is very rare in children. Hepatic microvascular changes may be the cause, as these vascular changes are uncommon in children. Antiphospholipid syndrome (APS), an autoimmune disease characterized by vascular thromboembolism, is extremely unusual in children.Patient ConcernsA 13-year-old girl who presented with abdominal pain and elevated liver enzymes was transferred to our hospital. Abdominal computed tomography and magnetic resonance imaging showed a massive mesenteric venous thrombus and a malignant mass with liver metastasis.DiagnosesHer immunological profile was positive for antinuclear antibodies (ANA) at a titer of 1/160 (nucleolar pattern), anticardiolipin antibodies (aCL) immunoglobulin G, and anti-histone antibody. A liver biopsy revealed hepatic NRH.InterventionsThe patient was initially started on heparin upon hospitalization and switched to warfarin and a vitamin K antagonist and continued treatment with international normalized ratio monitoring.OutcomesHer symptoms improved after 9 months of anticoagulation therapy.LessonsIn the presence of hepatic NRH or vascular thrombosis in children, we recommend that APS be differentially diagnosed using lupus anticoagulant and aCL and appropriate management be implemented.Copyright © 2021 the Author(s). Published by Wolters Kluwer Health, Inc.

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