Skip to main content
Top
Published in:

Open Access 02-09-2024 | Type 1 Diabetes | Case Report

Stevens-Johnson Syndrome/Toxic epidermal necrolysis complicated with fulminant type 1 diabetes mellitus: a case report and literature review

Authors: Xiaofang Zhang, Dihua Huang, Dajun Lou, Xuwei Si, Jiangfeng Mao

Published in: BMC Endocrine Disorders | Issue 1/2024

Login to get access

Abstract

Background

Stevens-Johnson syndrome (SJS) and toxic epidermal necrolysis (TEN) are rare but life-threatening skin lesion triggered by hypersensitive drug reaction. They are characterized by extensive epidermal necrosis and skin exfoliation. Fulminant type 1 diabetes mellitus (FT1DM) is featured by a rapid-onset of hyperglycemia with ketoacidosis due to severely destroyed β-cell function. Fulminant type 1 diabetes mellitus as a sequela of SJS/TEN has rarely been reported.

Case presentation

We present a 73-year-old female patient who developed SJS/TEN skin allergic reaction after taking carbamazepine and phenytoin for 35 days. Then, hyperglycemia and diabetic ketoacidosis occurred 20 days after discontinuation of antiepileptic drugs. A very low serum C-peptide level (8.79 pmol/l) and a near-normal glycosylated hemoglobin level met the diagnostic criteria for fulminant T1DM. Intravenous immunoglobulin (IVIG) and insulin were promptly administered, and the patient recovered finally.

Conclusions

This rare case indicates that monitoring blood glucose is necessary in SJS/TEN drug reaction, and comprehensive therapy with rehydration, insulin, antibiotics, and IVIG may improve the prognosis.
Literature
1.
go back to reference Bastuji-Garin S, Rzany B, Stern RS, et al. Clinical classification of cases of toxic epidermal necrolysis, Stevens-Johnson syndrome, and Erythema Multiforme. Arch Dermatol. 1993;129:92–6.CrossRefPubMed Bastuji-Garin S, Rzany B, Stern RS, et al. Clinical classification of cases of toxic epidermal necrolysis, Stevens-Johnson syndrome, and Erythema Multiforme. Arch Dermatol. 1993;129:92–6.CrossRefPubMed
3.
go back to reference Mockenhaupt M, Viboud C, Dunant A, et al. Toxic epidermal necrolysis: Assessment of Medication risks with emphasis on recently marketed drugs. The EuroSCAR-Study. J Invest Dermatol. 2008;128:35–44.CrossRefPubMed Mockenhaupt M, Viboud C, Dunant A, et al. Toxic epidermal necrolysis: Assessment of Medication risks with emphasis on recently marketed drugs. The EuroSCAR-Study. J Invest Dermatol. 2008;128:35–44.CrossRefPubMed
4.
go back to reference Imagawa A, Hanafusa T, Miyagawa J, et al. A novel subtype of type 1 diabetes mellitus characterized by a rapid onset and an absence of diabetes-related antibodies. N Engl J Med. 2000;342:301–7.CrossRefPubMed Imagawa A, Hanafusa T, Miyagawa J, et al. A novel subtype of type 1 diabetes mellitus characterized by a rapid onset and an absence of diabetes-related antibodies. N Engl J Med. 2000;342:301–7.CrossRefPubMed
5.
go back to reference Hanafusa T, Imagawa A. Fulminant type 1 diabetes: a novel clinical entity requiring special attention by all medical practitioners. Nat Clin Pract Endocrinol Metab. 2007;3:36–45.CrossRefPubMed Hanafusa T, Imagawa A. Fulminant type 1 diabetes: a novel clinical entity requiring special attention by all medical practitioners. Nat Clin Pract Endocrinol Metab. 2007;3:36–45.CrossRefPubMed
6.
go back to reference Song SO, Yun JS, Ko SH, et al. Prevalence and clinical characteristics of fulminant type 1 diabetes mellitus in Korean adults: a multi-institutional joint research. J Diabetes Investig. 2022;13:47–53.CrossRefPubMed Song SO, Yun JS, Ko SH, et al. Prevalence and clinical characteristics of fulminant type 1 diabetes mellitus in Korean adults: a multi-institutional joint research. J Diabetes Investig. 2022;13:47–53.CrossRefPubMed
7.
go back to reference Hosokawa Y, Hanafusa T, Imagawa A. Pathogenesis of fulminant type 1 diabetes: genes, viruses and the immune mechanism, and usefulness of patient-derived induced pluripotent stem cells for future research. J Diabetes Investig. 2019;10:1158–64.CrossRefPubMedPubMedCentral Hosokawa Y, Hanafusa T, Imagawa A. Pathogenesis of fulminant type 1 diabetes: genes, viruses and the immune mechanism, and usefulness of patient-derived induced pluripotent stem cells for future research. J Diabetes Investig. 2019;10:1158–64.CrossRefPubMedPubMedCentral
8.
go back to reference Zhu B, Wu J, Chen G, et al. Fulminant type 1 diabetes mellitus caused by drug reaction with eosinophilia and systemic symptoms (DRESS): a case report and review of the literature. Front Endocrinol. 2019;10:474.CrossRef Zhu B, Wu J, Chen G, et al. Fulminant type 1 diabetes mellitus caused by drug reaction with eosinophilia and systemic symptoms (DRESS): a case report and review of the literature. Front Endocrinol. 2019;10:474.CrossRef
9.
go back to reference Ardern-Jones MR, Mockenhaupt M. Making a diagnosis in severe cutaneous drug hypersensitivity reactions. Curr Opin Allergy Clin Immunol. 2019;19:283–93.CrossRefPubMed Ardern-Jones MR, Mockenhaupt M. Making a diagnosis in severe cutaneous drug hypersensitivity reactions. Curr Opin Allergy Clin Immunol. 2019;19:283–93.CrossRefPubMed
10.
go back to reference Hsu DY, Brieva J, Silverberg NB, et al. Morbidity and mortality of Stevens-Johnson syndrome and toxic epidermal necrolysis in United States adults. J Invest Dermatol. 2016;136:1387–97.CrossRefPubMed Hsu DY, Brieva J, Silverberg NB, et al. Morbidity and mortality of Stevens-Johnson syndrome and toxic epidermal necrolysis in United States adults. J Invest Dermatol. 2016;136:1387–97.CrossRefPubMed
11.
go back to reference Guvenir H, Arikoglu T, Vezir E, et al. Clinical phenotypes of severe cutaneous drug hypersensitivity reactions. Curr Pharm Des. 2019;25:3840–54.CrossRefPubMed Guvenir H, Arikoglu T, Vezir E, et al. Clinical phenotypes of severe cutaneous drug hypersensitivity reactions. Curr Pharm Des. 2019;25:3840–54.CrossRefPubMed
12.
13.
go back to reference Lerch M, Mainetti C, Terziroli Beretta-Piccoli B, et al. Current perspectives on Stevens-Johnson syndrome and toxic epidermal necrolysis. Clin Rev Allergy Immunol. 2018;54:147–76.CrossRefPubMed Lerch M, Mainetti C, Terziroli Beretta-Piccoli B, et al. Current perspectives on Stevens-Johnson syndrome and toxic epidermal necrolysis. Clin Rev Allergy Immunol. 2018;54:147–76.CrossRefPubMed
14.
go back to reference Mittmann N, Knowles SR, Koo M, et al. Incidence of toxic epidermal necrolysis and Stevens-Johnson syndrome in an HIV cohort: an observational, retrospective case series study. Am J Clin Dermatol. 2012;13:49–54.CrossRefPubMed Mittmann N, Knowles SR, Koo M, et al. Incidence of toxic epidermal necrolysis and Stevens-Johnson syndrome in an HIV cohort: an observational, retrospective case series study. Am J Clin Dermatol. 2012;13:49–54.CrossRefPubMed
15.
go back to reference Mulvey JM, Padowitz A, Lindley-Jones M, et al. Mycoplasma pneumoniae associated with Stevens Johnson syndrome. Anaesth Intensive Care. 2007;35:414–7.CrossRefPubMed Mulvey JM, Padowitz A, Lindley-Jones M, et al. Mycoplasma pneumoniae associated with Stevens Johnson syndrome. Anaesth Intensive Care. 2007;35:414–7.CrossRefPubMed
16.
go back to reference Maloney NJ, Ravi V, Cheng K, et al. Stevens-Johnson syndrome and toxic epidermal necrolysis-like reactions to checkpoint inhibitors: a systematic review. Int J Dermatol. 2020;59:e183–8.CrossRefPubMed Maloney NJ, Ravi V, Cheng K, et al. Stevens-Johnson syndrome and toxic epidermal necrolysis-like reactions to checkpoint inhibitors: a systematic review. Int J Dermatol. 2020;59:e183–8.CrossRefPubMed
17.
go back to reference Gu Y, Sebaratnam DF. Contextualising associations of SJS/TEN with COVID-19 and the vaccine. Burns. 2023;49:1776–7.CrossRefPubMed Gu Y, Sebaratnam DF. Contextualising associations of SJS/TEN with COVID-19 and the vaccine. Burns. 2023;49:1776–7.CrossRefPubMed
18.
go back to reference Le Cleach L, Delaire S, Boumsell L, et al. Blister fluid T lymphocytes during toxic epidermal necrolysis are functional cytotoxic cells which express human natural killer (NK) inhibitory receptors. Clin Exp Immunol. 2000;119:225–30.CrossRefPubMedPubMedCentral Le Cleach L, Delaire S, Boumsell L, et al. Blister fluid T lymphocytes during toxic epidermal necrolysis are functional cytotoxic cells which express human natural killer (NK) inhibitory receptors. Clin Exp Immunol. 2000;119:225–30.CrossRefPubMedPubMedCentral
19.
go back to reference Imagawa A, Hanafusa T, Awata T, et al. Report of the Committee of the Japan Diabetes Society on the research of fulminant and acute-onset type 1 diabetes mellitus: new diagnostic criteria of fulminant type 1 diabetes mellitus. J Diabetes Investig. 2012;3:536–9.CrossRefPubMedPubMedCentral Imagawa A, Hanafusa T, Awata T, et al. Report of the Committee of the Japan Diabetes Society on the research of fulminant and acute-onset type 1 diabetes mellitus: new diagnostic criteria of fulminant type 1 diabetes mellitus. J Diabetes Investig. 2012;3:536–9.CrossRefPubMedPubMedCentral
20.
go back to reference Onuma H, Tohyama M, Imagawa A, et al. High frequency of HLA B62 in fulminant type 1 diabetes with the drug-induced hypersensitivity syndrome. J Clin Endocrinol Metab. 2012;97:E2277–81.CrossRefPubMed Onuma H, Tohyama M, Imagawa A, et al. High frequency of HLA B62 in fulminant type 1 diabetes with the drug-induced hypersensitivity syndrome. J Clin Endocrinol Metab. 2012;97:E2277–81.CrossRefPubMed
21.
go back to reference Kano Y, Tohyama M, Aihara M, et al. Sequelae in 145 patients with drug-induced hypersensitivity syndrome/drug reaction with eosinophilia and systemic symptoms: Survey conducted by the Asian Research Committee on severe cutaneous adverse reactions (ASCAR). J Dermatol. 2015;4:276–82.CrossRef Kano Y, Tohyama M, Aihara M, et al. Sequelae in 145 patients with drug-induced hypersensitivity syndrome/drug reaction with eosinophilia and systemic symptoms: Survey conducted by the Asian Research Committee on severe cutaneous adverse reactions (ASCAR). J Dermatol. 2015;4:276–82.CrossRef
23.
go back to reference Chiou CC, Chung WH, Hung SI, et al. Fulminant type 1 diabetes mellitus caused by drug hypersensitivity syndrome with human herpesvirus 6 infection. J Am Acad Dermatol. 2006;54(2 Suppl):S14–7.CrossRefPubMed Chiou CC, Chung WH, Hung SI, et al. Fulminant type 1 diabetes mellitus caused by drug hypersensitivity syndrome with human herpesvirus 6 infection. J Am Acad Dermatol. 2006;54(2 Suppl):S14–7.CrossRefPubMed
24.
go back to reference Schneider JA, Cohen PR. Stevens-Johnson syndrome and toxic epidermal necrolysis: a concise review with a Comprehensive Summary of therapeutic interventions emphasizing supportive measures. Adv Ther. 2017;34:1235–44.CrossRefPubMedPubMedCentral Schneider JA, Cohen PR. Stevens-Johnson syndrome and toxic epidermal necrolysis: a concise review with a Comprehensive Summary of therapeutic interventions emphasizing supportive measures. Adv Ther. 2017;34:1235–44.CrossRefPubMedPubMedCentral
25.
go back to reference de Prost N, Ingen-Housz-Oro S, Duong T, et al. Bacteremia in Stevens-Johnson syndrome and toxic epidermal necrolysis: Epidemiology, risk factors, and predictive value of skin cultures. Med (Baltim). 2010;89:28–36.CrossRef de Prost N, Ingen-Housz-Oro S, Duong T, et al. Bacteremia in Stevens-Johnson syndrome and toxic epidermal necrolysis: Epidemiology, risk factors, and predictive value of skin cultures. Med (Baltim). 2010;89:28–36.CrossRef
26.
go back to reference Zimmermann S, Sekula P, Venhoff M, et al. Systemic immunomodulating therapies for Stevens-Johnson syndrome and toxic epidermal necrolysis: a systematic review and Meta-analysis. JAMA Dermatol. 2017;153:514–22.CrossRefPubMedPubMedCentral Zimmermann S, Sekula P, Venhoff M, et al. Systemic immunomodulating therapies for Stevens-Johnson syndrome and toxic epidermal necrolysis: a systematic review and Meta-analysis. JAMA Dermatol. 2017;153:514–22.CrossRefPubMedPubMedCentral
Metadata
Title
Stevens-Johnson Syndrome/Toxic epidermal necrolysis complicated with fulminant type 1 diabetes mellitus: a case report and literature review
Authors
Xiaofang Zhang
Dihua Huang
Dajun Lou
Xuwei Si
Jiangfeng Mao
Publication date
02-09-2024

Other articles of this Issue 1/2024

BMC Endocrine Disorders 1/2024 Go to the issue

At a glance: The STEP trials

A round-up of the STEP phase 3 clinical trials evaluating semaglutide for weight loss in people with overweight or obesity.

Developed by: Springer Medicine
Read more