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      Fulminant Type 1 Diabetes Mellitus Complicated with a Life-threatening Electrolyte Abnormality and Abnormal Electrocardiogram Findings

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          Abstract

          Fulminant type 1 diabetes mellitus (T1DM) is idiopathic T1DM with the rapid destruction of pancreatic β-cells. We herein report a 48-year-old man who developed fulminant T1DM complicated with a life-threatening electrolyte abnormality and abnormal electrocardiogram findings. He had no remarkable medical history, but one day, he developed general fatigue. His blood glucose level and HbA1c were 806 mg/dL and 6.3%, and his insulin secretion was markedly suppressed. He had ketoacidosis, hyponatremia and hyperkalemia. Furthermore, a life-threatening abnormality was noted on electrocardiogram. After fluid infusion and insulin therapy, the abnormality disappeared. In conclusion, we should bear in mind the possibility of fulminant T1DM in patients complaining of general malaise.

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          A novel subtype of type 1 diabetes mellitus characterized by a rapid onset and an absence of diabetes-related antibodies. Osaka IDDM Study Group.

          Y Matsuzawa, J Miyagawa, A Imagawa … (2000)
          Type 1 diabetes mellitus is now classified as autoimmune (type 1A) or idiopathic (type 1B), but little is known about the latter. We classified 56 consecutive Japanese adults with type 1 diabetes according to the presence or absence of glutamic acid decarboxylase antibodies (their presence is a marker of autoimmunity) and compared their clinical, serologic, and pathological characteristics. We divided the patients into three groups: 36 patients with positive tests for serum glutamic acid decarboxylase autoantibodies, 9 with negative tests for serum glutamic acid decarboxylase antibodies and glycosylated hemoglobin values higher than 11.5 percent, and 11 with negative tests for serum glutamic acid decarboxylase antibodies and glycosylated hemoglobin values lower than 8.5 percent. In comparison with the first two groups, the third group had a shorter mean duration of symptoms of hyperglycemia (4.0 days), a higher mean plasma glucose concentration (773 mg per deciliter [43 mmol per liter]) in spite of lower glycosylated hemoglobin values, diminished urinary excretion of C peptide, a more severe metabolic disorder (with ketoacidosis), higher serum pancreatic enzyme concentrations, and an absence of islet-cell, IA-2, and insulin antibodies. Immunohistologic studies of pancreatic-biopsy specimens from three patients with negative tests for glutamic acid decarboxylase autoantibodies and low glycosylated hemoglobin values revealed T-lymphocyte-predominant infiltrates in the exocrine pancreas but no insulitis and no evidence of acute or chronic pancreatitis. Some patients with idiopathic type 1 diabetes have a nonautoimmune, fulminant disorder characterized by the absence of insulitis and of diabetes-related antibodies, a remarkably abrupt onset, and high serum pancreatic enzyme concentrations.
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            Fulminant type 1 diabetes: a nationwide survey in Japan.

            Akira Shimada, Azuma Kanatsuka, Eiji Kawasaki … (2003)
            To describe the clinical and immunologic characteristics of fulminant type 1 diabetes, a novel subtype of type 1 diabetes, we conducted a nationwide survey. History and laboratory data, including islet-related autoantibodies, were examined in 222 patients with fulminant and nonfulminant type 1 diabetes in our hospitals in addition to another 118 patients with fulminant type 1 diabetes located outside our hospitals in Japan. In our hospitals, of the 222 patients studied, 43 (19.4%) were diagnosed with fulminant type 1 diabetes, 137 (61.7%) were classified as having autoimmune type 1 diabetes, and 42 were type 1 diabetic subjects who were not fulminant and did not have anti-islet antibodies. An additional 118 fulminant patients outside our hospitals were enrolled, making a total of 161 fulminant type 1 diabetic subjects (83 male and 78 female subjects; 14 children/adolescents and 147 adults) identified from all over Japan. (In 2000, the average incidence was three cases per month.) Flu-like symptoms and pregnancy were more frequently observed in the fulminant than in the autoimmune group (P < 0.001). In the fulminant patients, 4.8% were positive for anti-GAD antibodies and none were positive for anti-islet antigen 2 antibodies. Fulminant type 1 diabetes is a distinct subtype and accounts for approximately 20% of the ketosis-onset type 1 diabetes cases in Japan. Flu-like symptoms are characteristic of disease onset. Metabolic derangement is more severe in this subtype than in autoimmune type 1 diabetes.
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              Fulminant type 1 diabetes: a novel clinical entity requiring special attention by all medical practitioners.

              Akihisa Imagawa, Toshiaki Hanafusa (2006)
              Fulminant type 1 diabetes is a recently discovered subtype of type 1 diabetes. It is defined as diabetes in which the process of beta-cell destruction and the progression of hyperglycemia and ketoacidosis are extremely rapid. The pathogenesis of this disease remains to be clarified, but the involvement of both genetic background-especially human leukocyte antigen genes-and viruses has been suggested. Fulminant type 1 diabetes has the following clinical characteristics: duration of hyperglycemic symptoms is 4 days on average; there is a high prevalence of preceding common-cold-like and gastrointestinal symptoms; there is a near-normal level of glycated hemoglobin in spite of very high plasma glucose levels associated with ketoacidosis; the disease is sometimes related to pregnancy; and there are increased serum pancreatic enzyme levels, absent C-peptide levels, but virtually no detectable autoantibodies against constituents of pancreatic beta cells. The presence of the above characteristics strongly indicates the diagnosis of fulminant type 1 diabetes. Once the diagnosis of this disease is suspected, treatment of diabetic ketoacidosis must be started immediately, as in all other cases of type 1 diabetes. Otherwise, the death of the patient is likely to occur within 24 h. All medical practitioners must remember that this extremely rapidly progressing type of diabetes does exist, and they must pay special attention not to overlook it.
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                Author and article information

                Journal
                Journal ID (nlm-ta): Intern Med
                Journal ID (iso-abbrev): Intern. Med
                Title: Internal Medicine
                Publisher: The Japanese Society of Internal Medicine
                ISSN (Print): 0918-2918
                ISSN (Electronic): 1349-7235
                Publication date (Electronic): 27 April 2018
                Publication date (Print): 15 September 2018
                Volume: 57
                Issue: 18
                Pages: 2685-2688
                Affiliations
                [1 ]Department of Diabetes, Endocrinology and Metabolism, Kawasaki Medical School, Japan
                [2 ]Department of General Internal Medicine 1, Kawasaki Medical School, Japan
                Author notes

                Correspondence to Dr.　Hideaki Kaneto, kaneto@ 123456med.kawasaki-m.ac.jp

                Article
                DOI: 10.2169/internalmedicine.0680-17
                PMC ID: 6191599
                PubMed ID: 29709945
                SO-VID: 9aa6ea3c-7c64-438d-8d32-b82f92b80376
                Copyright © Copyright © 2018 by The Japanese Society of Internal Medicine
                License:

                The Internal Medicine is an Open Access article distributed under the Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License. To view the details of this license, please visit ( https://creativecommons.org/licenses/by-nc-nd/4.0/).

                History
                Date received : 19 December 2017
                Date accepted : 5 February 2018
                Categories
                Subject: Case Report

                Keywords: fulminant type 1 diabetes mellitus,diabetic ketoacidosis,hyperkalemia,a life-threatening electrocardiographic abnormality
                Data availability:
                Keywords: fulminant type 1 diabetes mellitus, diabetic ketoacidosis, hyperkalemia, a life-threatening electrocardiographic abnormality

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