Abstract. Drug-induced hypersensitivity syndrome (DIHS) is a severe systemic adverse drug reaction. Previous studies showed that DIHS is associated with the onset of fulminant type 1 diabetes mellitus (FT1D). Although genetic background and abnormalities in immune response or viral infection are considered to be associated with pathogenesis of FT1D, it remains unclear whether virus infection and specific human leukocyte antigen (HLA) typing are involved in DIHS-associated FT1D. Here, we report a case of a 78-year-old female patient with FT1D after DIHS treatment. She was diagnosed as DIHS caused by carbamazepine, and treatment with predonisolone was initiated. After 46 days from the occurrence of DIHS, she was admitted to our hospital because of type 1 diabetes mellitus and diabetic ketoacidosis. Although her Hemoglobin A1c (HbA1c) was elevated by predonisolone treatment (HbA1c: 9.2%), we diagnosed her as fulminant type 1 diabetes mellitus considering the abrupt onset of the ketoacidosis. Her general condition was improved by treatment with fluid infusion and insulin administration. During her clinical course, the infection of coxsackie B4 virus was observed. In addition, the examination of HLA typing showed HLA-A24 haplotype. These findings suggest that the coxsackie B4 virus infection may be involved in the pathogenesis of DIHS-induced FT1D, and that HLA-A24 haplotype might relate to DIHS-associated FT1D.Key words: Fulminant type 1 diabetes mellitus, Drug-induced hypersensitivity syndrome, Coxsackie virus B4FULMINANT TYPE 1 DIABETES MELLITUS (FT1D) is a subtype of type 1 diabetes mellitus characterized by extremely rapid progression of hyperglycemia and diabetic ketoacidosis (DKA) due to almost complete pancreatic beta-cell destruction [1,2]. Because FT1D often causes poor prognosis, we have to pay special attention to the development of FT1D in clinical settings. Drug-induced hypersensitivity syndrome (DIHS) is a severe systemic adverse drug reaction characterized by rash, fever, leukocytosis, eosinophilia and liver injury [3,4]. Reactivation of human herpes virus 6 (HHV-6) is one Submitted Jun. 8, 2017; Accepted Sep. 4, 2017 as EJ17-0249 Released online in J-STAGE as advance publication Sep. 29, 2017 Correspondence to: Ippei Kanazawa, Internal Medicine 1, Shimane University Faculty of Medicine, 89-1, Enya-cho, Izumo, Shimane, 693-8501, Japan E-mail: ippei.k@med.shimane-u.ac.jp of specific characteristics of DIHS [3]. Recently, it is reported that FT1D sometimes occurs after DIHS treatment [4,5]. Indeed, Onuma et al. previously reported that the clinical characteristics of FT1D with DIHS were similar to those without DIHS, but the frequency was much higher than that in the general population (0.54% vs. 0.01%) [4]. Because the mechanism of DIHSassociated FT1D still remains unclear, the investigation of clinical characteristics including genetic background and serological testing for viral infection are necessary to clarify this issue.
Case ReportA 78-year-old female patient visited a clinic for her blepharo...