Scientific article
Case report
OA Policy
English

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

Published inBMC endocrine disorders, vol. 23, no. 1, 228
Publication date2023-10-20
First online date2023-10-20
Abstract

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
  • Autoantibodies
  • Autoimmune
  • Beta cell
  • C-peptide
  • Case report
  • Diabetic ketoacidosis
  • Flatbush diabetes
  • Monogenic diabetes
  • Humans
  • Adolescent
  • Female
  • Diabetes Mellitus, Type 2 / complications
  • Insulin Resistance
  • Remission, Spontaneous
  • Glycated Hemoglobin
  • Lupus Erythematosus, Systemic / complications
  • Lupus Erythematosus, Systemic / diagnosis
  • Lupus Erythematosus, Systemic / drug therapy
  • Insulin
  • Diabetes Mellitus, Type 1 / complications
Citation (ISO format)
IAFRATE-LUTERBACHER, Fanny, BLOUIN, Jean-Louis, SCHWITZGEBEL LUSCHER, Valérie. Atypical diabetes with spontaneous remission associated with systemic lupus erythematosus in an adolescent girl of African ancestry, a case report. In: BMC endocrine disorders, 2023, vol. 23, n° 1, p. 228. doi: 10.1186/s12902-023-01478-0
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accessLevelPublic
Identifiers
Journal ISSN1472-6823
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