A case of glycogenic hepatopathy developed in a patient with new-onset fulminant type 1 diabetes: the role of image modalities in diagnosing hepatic glycogen deposition including gradient-dual-echo MRI

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  • Murata Fumi
    Department of Metabolism/Diabetes and Clinical Nutrition, Nagasaki University Hospital, Nagasaki 852-8501, Japan Department of Metabolism/Diabetes and Clinical Nutrition, Nagasaki University Hospital, Nagasaki 852-8501, Japan
  • Horie Ichiro
    Department of Internal Medicine, National Hospital Organization Saga National Hospital, Saga 849-8577, Japan Department of Molecular Medicine, Nagasaki University Graduate School of Biomedical Sciences, Nagasaki 852-8523, Japan Department of Endocrinology and Metabolism, Nagasaki University Hospital, Nagasaki 852-8501, Japan Department of Internal Medicine, National Hospital Organization Saga National Hospital, Saga 849-8577, Japan Department of Molecular Medicine, Nagasaki University Graduate School of Biomedical Sciences, Nagasaki 852-8523, Japan Department of Endocrinology and Metabolism, Nagasaki University Hospital, Nagasaki 852-8501, Japan
  • Ando Takao
    Department of Endocrinology and Metabolism, Nagasaki University Hospital, Nagasaki 852-8501, Japan Department of Endocrinology and Metabolism, Nagasaki University Hospital, Nagasaki 852-8501, Japan
  • Isomoto Eriko
    Department of Endocrinology and Metabolism, Nagasaki University Hospital, Nagasaki 852-8501, Japan Department of Endocrinology and Metabolism, Nagasaki University Hospital, Nagasaki 852-8501, Japan
  • Hayashi Hideyuki
    Department of Radiology, Nagasaki University Hospital, Nagasaki 852-8501, Japan Department of Radiology, Nagasaki University Hospital, Nagasaki 852-8501, Japan
  • Akazawa Satoru
    Department of Endocrinology and Metabolism, Nagasaki University Hospital, Nagasaki 852-8501, Japan Department of Endocrinology and Metabolism, Nagasaki University Hospital, Nagasaki 852-8501, Japan
  • Ueki Ikuko
    Department of Endocrinology and Metabolism, Nagasaki University Hospital, Nagasaki 852-8501, Japan Department of Endocrinology and Metabolism, Nagasaki University Hospital, Nagasaki 852-8501, Japan
  • Nakamura Kan
    Department of Endocrinology and Metabolism, Nagasaki University Hospital, Nagasaki 852-8501, Japan Department of Endocrinology and Metabolism, Nagasaki University Hospital, Nagasaki 852-8501, Japan
  • Kobayashi Masakazu
    Department of Endocrinology and Metabolism, Nagasaki University Hospital, Nagasaki 852-8501, Japan Department of Endocrinology and Metabolism, Nagasaki University Hospital, Nagasaki 852-8501, Japan
  • Kuwahara Hironaga
    Department of Endocrinology and Metabolism, Nagasaki University Hospital, Nagasaki 852-8501, Japan Department of Endocrinology and Metabolism, Nagasaki University Hospital, Nagasaki 852-8501, Japan
  • Abiru Norio
    Department of Endocrinology and Metabolism, Nagasaki University Hospital, Nagasaki 852-8501, Japan Department of Endocrinology and Metabolism, Nagasaki University Hospital, Nagasaki 852-8501, Japan
  • Kawasaki Eiji
    Department of Metabolism/Diabetes and Clinical Nutrition, Nagasaki University Hospital, Nagasaki 852-8501, Japan Department of Metabolism/Diabetes and Clinical Nutrition, Nagasaki University Hospital, Nagasaki 852-8501, Japan
  • Yamasaki Hironori
    Center for Health and Community Medicine, Nagasaki University, Nagasaki 852-8521, Japan Center for Health and Community Medicine, Nagasaki University, Nagasaki 852-8521, Japan
  • Kawakami Atsushi
    Department of Metabolism/Diabetes and Clinical Nutrition, Nagasaki University Hospital, Nagasaki 852-8501, Japan Department of Endocrinology and Metabolism, Nagasaki University Hospital, Nagasaki 852-8501, Japan Department of Metabolism/Diabetes and Clinical Nutrition, Nagasaki University Hospital, Nagasaki 852-8501, Japan Department of Endocrinology and Metabolism, Nagasaki University Hospital, Nagasaki 852-8501, Japan

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Abstract

Glycogenic hepatopathy (GH) has been reported as a very rare and under recognized complication in long-standing poorly controlled type 1 diabetes (T1D) patients. GH is characterized by transient elevation of liver transaminase and hepatomegaly caused by reversible and excessive glycogen accumulation in hepatocytes. It has been reported that GH is indistinguishable from non-alcoholic fatty liver disease, which is more commonly seen in diabetic patients, even after a history is taken and a physical examination or imaging studies have been performed. GH can only be diagnosed by liver biopsy. We here demonstrate a 21-year-old male patient with new-onset fulminant T1D complicated with diabetic ketoacidosis who subsequently developed GH just after the initiation of insulin treatment. The marked liver dysfunction (serum levels of aspartate aminotransferase 769 IU/L and alanine aminotransferase 1348 IU/L) and hepatomegaly improved spontaneously via glycemic control without any specific treatments thereafter. Moreover, the insulin requirement dramatically decreased from 168 to 80 units per day as GH improved, suggesting a potential role of GH in insulin resistance. GH was diagnosed based on the histological findings of the liver in our case, but we were able to predict GH before the biopsy based on the findings in the gradient-dual-echo magnetic resonance imaging sequence combined with ultrasound and/or computed tomography examinations of the liver.

Journal

  • Endocrine Journal

    Endocrine Journal 59 (8), 669-676, 2012

    The Japan Endocrine Society

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