Atypical diabetes with spontaneous remission associated with systemic lupus erythematosus in an adolescent girl of African ancestry, a case report

Background New-onset diabetes in youth encompasses type 1 diabetes, type 2 diabetes, monogenic diabetes, and rarer subtypes like Type B insulin resistance syndrome and ketosis-prone atypical diabetes in African populations. Some cases defy classification, posing management challenges. Here, we prese...

Full description

Saved in:
Bibliographic Details
Published in:BMC endocrine disorders 2023-10, Vol.23 (1), p.1-228, Article 228
Main Authors: Luterbacher, Fanny, Blouin, Jean-Louis, Schwitzgebel, Valerie M
Format: Article
Language:English
Subjects:
African Americans
Ambulatory care
Antibodies
Autoantibodies
Autoimmune diseases
Autoimmunity
Beta cell
Blood glucose
C-peptide
Case Report
Case reports
Corticosteroids
Development and progression
Diabetes
Diabetes mellitus (insulin dependent)
Diabetes mellitus (non-insulin dependent)
Diabetes therapy
Diabetic ketoacidosis
Disease
Flatbush diabetes
Gene expression
Genealogy
Genetic screening
Glucose
Glycosylated hemoglobin
Haplotypes
Health aspects
Health risk assessment
Hydroxychloroquine
Immunomodulation
Insulin
Insulin resistance
Ketoacidosis
Ketosis
Lupus
Medical screening
Metabolism
Metformin
Monogenic diabetes
Mutation
Mycophenolate mofetil
Mycophenolic acid
Patients
Peptides
Prednisone
Remission
Remission (Medicine)
Systemic lupus erythematosus
Teenage girls
Type 1 diabetes
Type 2 diabetes
Citations: Items that this one cites
Online Access:Get full text
Tags: Add Tag
No Tags, Be the first to tag this record!
Description
Summary:Background New-onset diabetes in youth encompasses type 1 diabetes, type 2 diabetes, monogenic diabetes, and rarer subtypes like Type B insulin resistance syndrome and ketosis-prone atypical diabetes in African populations. Some cases defy classification, posing management challenges. Here, we present a case of a unique, reversible diabetes subtype. Case presentation We describe an adolescent African girl recently diagnosed with systemic lupus erythematosus. At age 15, she presented with ketoacidosis, HbA1c of 108.7 mmol/mol (12.1%), and positive anti-insulin antibodies. Initially diagnosed with type 1 diabetes, insulin was prescribed. Due to the presence of obesity and signs of insulin resistance, we added metformin. Concurrently, she received treatment for lupus with hydroxychloroquine, mycophenolate mofetil, and prednisone. After discharge, she stopped insulin due to cultural beliefs. Five months later, her glycemia and HbA1c normalized (37 mmol/mol or 5.5%) without insulin, despite corticosteroid therapy and weight gain. Autoantibodies normalized, and lupus activity decreased. Genetic testing for monogenic diabetes was negative, and the type 1 genetic risk score was exceptionally low. Conclusions We present a complex, reversible diabetes subtype. Features suggest an autoimmune origin, possibly influenced by overlapping HLA risk haplotypes with lupus. Lupus treatment or immunomodulation may have impacted diabetes remission. Ancestry-tailored genetic risk scores are currently designed to improve diagnostic accuracy. Keywords: Case report, Flatbush diabetes, Monogenic diabetes, C-peptide, Beta cell, Diabetic ketoacidosis, Autoantibodies, Autoimmune
ISSN:1472-6823
1472-6823
DOI:10.1186/s12902-023-01478-0