A Case of Congenital Hepatoblastoma Presented with Hepatic Osteodystrophy Complicated by Multiple Bone Fractures

Hepatic osteodystrophy is frequent complication in patients with chronic liver disease, particularly with chronic cholestasis. We report a male infant with congenital hepatoblastoma, who had osteodystrophy complicated by multiple bone fractures despite adequate supplementation of fat-soluble vitamin...

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Main Authors: Yoon Heui So, Dae Sung Kim, Bo Ae Yoon, Yoo-Duk Choi, Hee Jo Baek, Hoon Kook
Format: Article
Language:English
Published: The Korean Society of Pediatric Hematology-Oncology 2019-10-01
Series:Clinical Pediatric Hematology-Oncology
Subjects:
Online Access:https://doi.org/10.15264/cpho.2019.26.2.110
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author Yoon Heui So
Dae Sung Kim
Bo Ae Yoon
Yoo-Duk Choi
Hee Jo Baek
Hoon Kook
author_facet Yoon Heui So
Dae Sung Kim
Bo Ae Yoon
Yoo-Duk Choi
Hee Jo Baek
Hoon Kook
author_sort Yoon Heui So
collection DOAJ
description Hepatic osteodystrophy is frequent complication in patients with chronic liver disease, particularly with chronic cholestasis. We report a male infant with congenital hepatoblastoma, who had osteodystrophy complicated by multiple bone fractures despite adequate supplementation of fat-soluble vitamins including vitamin D. He was born by Caesarean section because of a 7 cm–sized abdominal mass detected by prenatal ultrasonography. The pathologic diagnosis was hepatoblastoma, PRETEXT staging III or IV. Whole body bone scan at the time of diagnosis showed no abnormal uptake. Oral vitamin D3 of 2,000 IU/day was administered with other fat-soluble vitamins. Serum direct bilirubin level gradually increased up to 28.9 mg/dL at postnatal 6 days and was above 5 mg/dL until 110 days of age. Bony changes consistent with rickets became apparent in left proximal humerus since 48 days of age, and multiple bone fractures developed thereafter. With resolving cholestasis by chemotherapy, his bony lesions improved gradually after add-on treatment of bisphosphonate and parenteral administration of vitamin D with calcium. High level of suspicion and prevention of osteodystrophy is needed in patients with hepatoblastoma, especially when cholestasis persists.
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spelling doaj.art-71ad33629785411da74c5734facc20552022-12-21T23:46:53ZengThe Korean Society of Pediatric Hematology-OncologyClinical Pediatric Hematology-Oncology2233-52502019-10-0126211011410.15264/cpho.2019.26.2.110cpho.2019.26.2.110A Case of Congenital Hepatoblastoma Presented with Hepatic Osteodystrophy Complicated by Multiple Bone FracturesYoon Heui So0Dae Sung Kim1Bo Ae Yoon2Yoo-Duk Choi3Hee Jo Baek4Hoon Kook5Department of Pediatrics, Chonnam National University Hwasun Hospital, Hwasun, KoreaDepartment of Pediatrics, Chonnam National University Hwasun Hospital, Hwasun, KoreaDepartment of Pediatrics, Chonnam National University Hwasun Hospital, Hwasun, KoreaDepartments of Pathology, Chonnam National University Medical School, Gwangju, KoreaDepartment of Pediatrics, Chonnam National University Hwasun Hospital, Hwasun, KoreaDepartment of Pediatrics, Chonnam National University Hwasun Hospital, Hwasun, KoreaHepatic osteodystrophy is frequent complication in patients with chronic liver disease, particularly with chronic cholestasis. We report a male infant with congenital hepatoblastoma, who had osteodystrophy complicated by multiple bone fractures despite adequate supplementation of fat-soluble vitamins including vitamin D. He was born by Caesarean section because of a 7 cm–sized abdominal mass detected by prenatal ultrasonography. The pathologic diagnosis was hepatoblastoma, PRETEXT staging III or IV. Whole body bone scan at the time of diagnosis showed no abnormal uptake. Oral vitamin D3 of 2,000 IU/day was administered with other fat-soluble vitamins. Serum direct bilirubin level gradually increased up to 28.9 mg/dL at postnatal 6 days and was above 5 mg/dL until 110 days of age. Bony changes consistent with rickets became apparent in left proximal humerus since 48 days of age, and multiple bone fractures developed thereafter. With resolving cholestasis by chemotherapy, his bony lesions improved gradually after add-on treatment of bisphosphonate and parenteral administration of vitamin D with calcium. High level of suspicion and prevention of osteodystrophy is needed in patients with hepatoblastoma, especially when cholestasis persists.https://doi.org/10.15264/cpho.2019.26.2.110congenital hepatoblastomafracturehepatic osteodystrophy
spellingShingle Yoon Heui So
Dae Sung Kim
Bo Ae Yoon
Yoo-Duk Choi
Hee Jo Baek
Hoon Kook
A Case of Congenital Hepatoblastoma Presented with Hepatic Osteodystrophy Complicated by Multiple Bone Fractures
Clinical Pediatric Hematology-Oncology
congenital hepatoblastoma
fracture
hepatic osteodystrophy
title A Case of Congenital Hepatoblastoma Presented with Hepatic Osteodystrophy Complicated by Multiple Bone Fractures
title_full A Case of Congenital Hepatoblastoma Presented with Hepatic Osteodystrophy Complicated by Multiple Bone Fractures
title_fullStr A Case of Congenital Hepatoblastoma Presented with Hepatic Osteodystrophy Complicated by Multiple Bone Fractures
title_full_unstemmed A Case of Congenital Hepatoblastoma Presented with Hepatic Osteodystrophy Complicated by Multiple Bone Fractures
title_short A Case of Congenital Hepatoblastoma Presented with Hepatic Osteodystrophy Complicated by Multiple Bone Fractures
title_sort case of congenital hepatoblastoma presented with hepatic osteodystrophy complicated by multiple bone fractures
topic congenital hepatoblastoma
fracture
hepatic osteodystrophy
url https://doi.org/10.15264/cpho.2019.26.2.110
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