Successful treatment of recurrent hypoglycemia by pioglitazone in a patient with myotonic dystrophy

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  • Yamamoto Toshiyuki
    Department of Neurology, National Center Hospital of Neurology and Psychiatry Department of Neurology and Neurological Science, Graduate School, Tokyo Medical and Dental University
  • Oya Yasushi
    Department of Neurology, National Center Hospital of Neurology and Psychiatry
  • Furusawa Yoshihiko
    Department of Neurology, National Center Hospital of Neurology and Psychiatry
  • Nonaka Ikuya
    Department of Pediatric Neurology, National Center Hospital of Neurology and Psychiatry
  • Murata Miho
    Department of Neurology, National Center Hospital of Neurology and Psychiatry

Bibliographic Information

Other Title
  • 反復する無自覚性低血糖の治療にＰｉｏｇｌｉｔａｚｏｎｅが有効であった筋強直性ジストロフィーの１例
  • 症例報告 反復する無自覚性低血糖の治療にPioglitazoneが有効であった筋強直性ジストロフィーの1例
  • ショウレイ ホウコク ハンプク スル ムジカクセイ テイケットウ ノ チリョウ ニ Pioglitazone ガ ユウコウ デ アッタ キン ゴウチョクセイ ジストロフィー ノ 1レイ

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Abstract

A 20 year-old woman with myotonic dystrophy type 1 (DM1) presented with fatigue, daytime somnolence, and sudden poor responsiveness. Blood glucose was measured before and after each meal for 4 days, and hypoglycemia was confirmed twice, although neither perspiration nor palpitations occurred in the hypoglycemic state. On a 75g oral glucose tolerance test (OGTT), fasting blood glucose level was 83mg/dl, and fasting blood immunoreactive insulin (IRI) level was 5.96μIU/ml. However, IRI increased to 528μIU/ml at 60 minutes and blood glucose decreased to 57mg/dl at 120 minutes of the OGTT. The patient was diagnosed with reactive hypoglycemia due to excessive insulin secretion. Oral administration of pioglitazone improved the excessive insulin secretion as assessed by OGTT. After starting treatment, hypoglycemia was not detected either pre- or post-prandially. After 10 months of treatment, blood glucose level after glucose loading was higher than fasting blood glucose level during OGTT, and the IRI area under the curve of the OGTT decreased. We considered that hypoglycemia unawareness resulted from recurrent hypoglycemic episodes in this patient. Pioglitazone was effective in improving hyperinsulinemia and reactive hypoglycemia in nondiabetic DM1.<br>

Journal

  • Rinsho Shinkeigaku

    Rinsho Shinkeigaku 49 (10), 641-645, 2009

    Societas Neurologica Japonica

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