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A Case of Fulminant Type 1 Diabetes With Graves’ Disease

¹ From the First Department of Internal Medicine, Nagoya University School of Medicine, Nagoya, Japan

Type 1 diabetes is now classified as classic autoimmune (type 1A) and idiopathic (type 1B) diabetes. Fulminant type 1 diabetes was recently characterized as a novel subtype of type 1B diabetes. This disease was characterized as rapid onset, having an absence of diabetes-related autoantibodies, and having the presence of lymphocyte infiltration in exocrine pancreatic tissue without insulitis (1–3). Although fulminant type 1 diabetes has been clinically recognized, its etiology still remains obscure. In this article, we report a case of fulminant type 1 diabetes developed during pregnancy with manifested Graves’ disease, which was developed 1 year after onset of diabetes.

A 28-year-old female was admitted to the hospital with diabetic ketoacidosis. She was 27 weeks pregnant and presented no symptom of preceding infection. After admission, she became comatose. Her arterial pH was 6.988 and she had marked elevation of ketone bodies. Her plasma glucose was 43.8 mmol/l; however, her HbA_1c was 4.8%. Serum C-peptide was under the detection limit (<0.03 ng/ml), and urinary C-peptide was 0.86 µg/day. There was no response to a glucagon C-peptide stimulation test. Autoantibodies to the cytoplasm of islet, GAD, insulin, and tyrosine phosphate-like protein (IA-2) were all negative. Her serum amylase was 267 IU/l. Both her serum lipase and elastase 1 levels were elevated 55 IU/l and 1,200 ng/dl, respectively. These findings were consistent with symptoms of fulminant type 1 diabetes. Her fetus died, and artificial abortion was performed. The fetus had no superficial abnormality. The subject had HLA-DRB1*0101/*0901, DQB1-*0612/*0306, A2/A24 (9), B7/B61 (40), and Cw7. She was in a euthyroid state, and autoantibodies to the thyroid were negative at that time.

After 1 year she presented overt thyrotoxic symptoms such as hyperhydrosis, palpitation, finger tremor, and poor glycemic control. Her thyroid hormones were elevated, and the thyroid-stimulating hormone receptor antibody was positive. She was diagnosed with Graves’ disease and administered propylthiouracil. Her glycemic control was fair with continuous subcutaneous insulin infusion. Diabetes-related autoantibodies were still negative, and her intrinsic insulin secretion was scant. Like a previous short report (4), our case had immunogenetic characteristics of an autoimmune disease except for endocrine pancreas. We often observed classic type 1A diabetes associated with autoimmune thyroid disease. This case was unique in that diabetes developed during pregnancy and was complicated with autoimmune disease. This case may help clarify the etiology of this disease entity.

Footnotes

Address correspondence to Yoshitaka Miura, First Department of Internal Medicine, Nagoya University School of Medicine, 65 Tsurumai-cho, Showa-ku, Nagoya 466-8550 Japan, E-mail: ymiura64{at}med.nagoya-u.ac.jp.

References

1. Imagawa A, Hanafusa T, Miyagawa J, Matsuzawa Y: A novel subtype of type 1 diabetes mellitus characterized by a rapid onset and an absence of diabetes-related antibodies: Osaka IDDM Study Group. N Engl J Med 342: 301–307, 2000[Abstract/Free Full Text]
   
2. Sekine N, Motokura T, Oki T, Umeda Y, Sasaki N, Hayashi M, Sato H, Fujita T, Kaneko T, Asano Y, Kikuchi K: Rapid loss of insulin secretion in a patient with fulminant type 1 diabetes mellitus and carbamazepine hypersensitivity syndrome. JAMA 285: 1153–1154, 2001[Free Full Text]
   
3. Tanaka S, Kobayashi T, Momotsu T: A novel subtype of type 1 diabetes mellitus. N Engl J Med 342: 1835–1837, 2000[Free Full Text]
   
4. Sakaue S, Nagata M, Wakabayashi O, Honda T, Yoshimura H, Yamaguchi E, Nishimura M: A case of fulminat type 1 diabetes with elevated rheumatoid factor and the temporal presence of thyroid-stimulating hormone receptor antibody (Letter). Diabetes Care 25: 935–936, 2002[Free Full Text]
   

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This Article Extract Full Text (PDF) Purchase Article View Shopping Cart Alert me when this article is cited Alert me if a correction is posted Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Download to citation manager Request Permissions Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Miura, Y. Articles by Oiso, Y. Search for Related Content PubMed PubMed Citation Articles by Miura, Y. Articles by Oiso, Y. Social Bookmarking                What's this?