Case Report
Copyright ©The Author(s) 2021. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. May 6, 2021; 9(13): 3163-3169
Published online May 6, 2021. doi: 10.12998/wjcc.v9.i13.3163
Sodium-glucose co-transporter-2 inhibitor-associated euglycemic diabetic ketoacidosis that prompted the diagnosis of fulminant type-1 diabetes: A case report
Taro Yasuma, Yuko Okano, Soichiro Tanaka, Kota Nishihama, Kazuhito Eguchi, Chisa Inoue, Kanako Maki, Akihiro Uchida, Mei Uemura, Toshinari Suzuki, Corina N D'Alessandro-Gabazza, Esteban C Gabazza, Yutaka Yano
Taro Yasuma, Yuko Okano, Department of Diabetes, Metabolism, and Endocrinology and Immunology, Mie University, Tsu 514-8507, Japan
Soichiro Tanaka, Kota Nishihama, Kazuhito Eguchi, Chisa Inoue, Kanako Maki, Akihiro Uchida, Mei Uemura, Toshinari Suzuki, Department of Diabetes, Metabolism, and Endocrinology, Mie University, Tsu 514-8507, Japan
Corina N D'Alessandro-Gabazza, Department of Immunology, Mie University School of Medicine, Mie University, Tsu 514-8507, Japan
Esteban C Gabazza, Department of Immunology, Mie University Faculty and Graduate School of Medicine, Tsu 514-8507, Japan
Yutaka Yano, Department of Diabetes, Mie University Graduate School of Medicine, Tsu 514-8507, Japan
Author contributions: Yasuma T, Tanaka S, and Nishihama K were responsible for clinical treatment and clinical follow-up; Yasuma T prepared the first draft of the manuscript; Eguchi KC, Inoue CC, Maki KC, Okano Y, and Uchida A contributed with resources and acquisition of data; Suzuki T, D'Alessandro-Gabazza CN, Yano Y, and Gabazza EC contributed to the interpretation of the data and made an intellectual contribution to the manuscript's preparation.
Informed consent statement: Written informed consent was obtained from the patient to publish clinical details and images when she was alive.
Conflict-of-interest statement: Yano Y reports receiving lecture fees from Novo Nordisk; and Gabazza EC received grants from Shionogi Corporation and Asahi Kasei. Other authors declared no conflict of interest concerning this case report.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Esteban C Gabazza, MD, PhD, Director, Full Professor, Department of Immunology, Mie University Faculty and Graduate School of Medicine, Edobashi 2-174, Tsu 514-8507, Japan. gabazza@doc.medic.mie-u.ac.jp
Received: December 8, 2020
Peer-review started: December 8, 2020
First decision: January 24, 2021
Revised: January 26, 2021
Accepted: March 11, 2021
Article in press: March 11, 2021
Published online: May 6, 2021
Processing time: 135 Days and 9.9 Hours
Abstract
BACKGROUND

Fulminant type 1 diabetes mellitus (FT1DM) is a subtype of type 1 diabetes mellitus characterized by an abrupt onset and a rapid and complete functional loss of islet β cells. It is a very rare disease generally associated with ketoacidosis and the absence of circulating pancreatic islet-related autoantibodies. Diabetic ketoacidosis with normal blood glucose levels has been reported during sodium-glucose co-transporter 2 (SGLT2) inhibitor therapy.

CASE SUMMARY

The patient was a 43-year-old woman that consulted a medical practitioner for malaise, thirst, and vomiting. Blood analysis showed high blood glucose levels (428 mg/dL), a mild increase of hemoglobin A1c (6.6%), and increased ketone bodies in urine. The patient was diagnosed with type 2 diabetes mellitus. The patient was initially treated with insulin, which was subsequently changed to an oral SGLT2 inhibitor. Antibodies to glutamic acid decarboxylase were negative. Four days after receiving oral SGLT2 inhibitor, she consulted at Mie University Hospital, complaining of fatigue and vomiting. Laboratory analysis revealed diabetic ketoacidosis with almost normal blood glucose levels. The endogenous insulin secretion was markedly low, and the serum levels of islet-related autoantibodies were undetectable. We made the diagnosis of FT1DM with concurrent SGLT2 inhibitor-associated euglycemic diabetic ketoacidosis. The patient's general condition improved after therapy with intravenous insulin and withdrawal of oral medication. She was discharged on day 14 with an indication of multiple daily insulin therapy.

CONCLUSION

This patient is a rare case of FT1DM that developed SGLT2 inhibitor-associated diabetic ketoacidosis with almost normal blood glucose levels. This case report underscores the importance of considering the diagnosis of FT1DM in patients with negative circulating autoantibodies and a history of hyperglycemia that subsequently develop euglycemic diabetic ketoacidosis following treatment with a SGLT2 inhibitor.

Keywords: Euglycemic diabetic ketoacidosis; Sodium-glucose cotransporter 2 inhibitors; Fulminant type 1 diabetes; Case report

Core Tip: Fulminant type 1 diabetes mellitus (FT1DM) is characterized by a rapid functional loss of islet β cells and ketoacidosis. Herein, we report a rare case of FT1DM wrongly diagnosed by a medical practitioner as a type 2 diabetes mellitus for the absence of circulating autoantibodies and treated with an oral sodium-glucose co-transporter 2 (SGLT2) inhibitor. This treatment was associated with diabetic symptoms and euglycemic diabetic ketoacidosis. Insulin therapy and SGLT2 inhibitor withdrawal ameliorated the patient's clinical condition. This case report underscores the importance of considering FT1DM in patients with negative circulating autoantibodies and hyperglycemia that develop euglycemic diabetic ketoacidosis during SGLT2 inhibitor treatment.