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Iguchi T, Yamagata M, Sonoda T, et al. Malignant transformation of hepatocellular adenoma with bone marrow metaplasia arising in glycogen storage disease type I: A case report. Mol Clin Oncol. 2016;5(5):599-603doi: 10.3892/mco.2016.1034.
Iguchi, T., Yamagata, M., Sonoda, T., Yanagita, K., Fukahori, T., Tsujita, E., Aishima, S., Oda, Y., & Maehara, Y. (2016). Malignant transformation of hepatocellular adenoma with bone marrow metaplasia arising in glycogen storage disease type I: A case report. Molecular and clinical oncology, 5(5), 599-603. https://doi.org/10.3892/mco.2016.1034
Iguchi, Tomohiro, et al. "Malignant transformation of hepatocellular adenoma with bone marrow metaplasia arising in glycogen storage disease type I: A case report." Molecular and clinical oncology vol. 5,5 (2016): 599-603. doi: https://doi.org/10.3892/mco.2016.1034
Iguchi T, Yamagata M, Sonoda T, Yanagita K, Fukahori T, Tsujita E, Aishima S, Oda Y, Maehara Y. Malignant transformation of hepatocellular adenoma with bone marrow metaplasia arising in glycogen storage disease type I: A case report. Mol Clin Oncol. 2016 Nov;5(5):599-603. doi: 10.3892/mco.2016.1034. Epub 2016 Sep 21. PMID: 27900094; PMCID: PMC5103882.
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Mol Clin Oncol. 2016 Nov;5(5):599-603. doi: 10.3892/mco.2016.1034. Epub 2016 Sep 21.

Malignant transformation of hepatocellular adenoma with bone marrow metaplasia arising in glycogen storage disease type I: A case report.

Molecular and clinical oncology

Tomohiro Iguchi, Motoyuki Yamagata, Takashi Sonoda, Kimihiko Yanagita, Tetsuhiro Fukahori, Eiji Tsujita, Shinichi Aishima, Yoshinao Oda, Yoshihiko Maehara

Affiliations

  1. Department of Gastroenterological Surgery, National Kyushu Cancer Center, Fukuoka 811-1395, Japan.
  2. Department of Surgery, Saiseikai Karatsu Hospital, Karatsu, Saga 847-0852, Japan.
  3. Department of Internal Medicine, Saiseikai Karatsu Hospital, Karatsu, Saga 847-0852, Japan.
  4. Department of Radiology, Saiseikai Karatsu Hospital, Karatsu, Saga 847-0852, Japan.
  5. Department of Pathology and Microbiology, Saga Medical School Faculty of Medicine, Saga University, Saga 849-8501, Japan.
  6. Department of Anatomic Pathology, Graduate School of Medical Sciences, Kyushu University, Fukuoka, Fukuoka 812-8582, Japan.
  7. Department of Surgery and Science, Graduate School of Medical Sciences, Kyushu University, Fukuoka, Fukuoka 812-8582, Japan.

PMID: 27900094 PMCID: PMC5103882 DOI: 10.3892/mco.2016.1034

Abstract

Malignant transformation of hepatocellular adenoma (HA) is relatively rare and has been reported to be associated with dysregulation of the β-catenin pathway. The presence of bone marrow metaplasia in HA is an uncommon histological characteristic. The current report presents the case of a 46-year-old woman with glycogen storage disease type I (von Gierke's disease) who underwent resection of hepatocellular carcinoma (HCC) arising in a HA with associated bone marrow metaplasia producing three series of hematopoietic cells. The serum level of proteins induced by des-gamma-carboxy prothrombin (DCP) gradually increased as the tumors grew; following hepatic resection, DCP levels returned to normal. Nuclear accumulation of β-catenin was shown in HCC by immunohistochemistry; however, no mutation was detected in exon 3 of β-catenin. To the best of our knowledge, this is the first report of HA with absolute bone marrow metaplasia producing three series of hematopoietic cells. This occurrence suggests that elevated DCP may be an indicator of malignant transformation of HA.

Keywords: bone marrow metaplasia; des-gamma-carboxy prothrombin; hepatocellular adenoma; hepatocellular carcinoma; β-catenin

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