An Unusual Cause of Hypoglycemia: Insulin Autoimmune Syndrome

Year 2021, , 30 - 34, 29.01.2021

Yasemin Aydoğan Ünsal , Özen Öz Gül , Soner Cander , Canan Ersoy , Oktay Ünsal , Ensar Aydemir , Erdinç Ertürk

https://doi.org/10.46310/tjim.786828

Abstract

Insulin autoimmune syndrome (IAS) is a rare cause of hyperinsulinemic hypoglycemia characterized by antibodies to endogenous insulin without exposure to exogenous insulin. In this report, we presented a case of insulin autoimmune syndrome with a history of fasting hypoglycemia. After work up and exlusion of other causes such as insulinoma, hyperinsulinemic hypogylcemic state of the patient was considered to have been induced by etofenamate. Although IAS is generally self limiting and dietary management and withdrawal of trigger drug are enough to maintain euglycemia, in some cases corticosteroids, plasmapheresis, rituximab can be used for treatment. In our case, despite dietary management, hypoglycemia was severe and the patient’s life quality was adversely effected. After treatment with prednisolone, hypoglycemic episodes became less and less frequent. IAS should be considered as a differential diagnosis of hyperinsulinemic hypogylcemic states to avoid unnecessary interventions.

Keywords

Insulin autoimmune syndrome, hyperinsulinemic hypoglycemic state, corticosteroids

References

• 1- Hirata Y, Ishizu H, Ouchi N, Motomura S, Abe M, Hara Y, et al. Insulin autoimmunity in a case of spontaneous hypoglycemia. J Jpn Diabetes Soc 1970; 13: 312 – 20.
• 2- Redmon JB, Nuttall FQ. Autoimmune hypoglycemia. Endocrinol Metab Clin North Am. 1999; 28 (3): 603 – 618.
• 3- Uchigata Y, Hirata Y. Insulin autoimmune syndrome (IAS, Hirata disease). Ann Med Interne (Paris) 1999; 150: 245 – 53.
• 4- J. S. Han, H. J. Moon, J. S. Kim, et al. Anti-tuberculosis Treatment Induced Insulin Autoimmune Syndrome. The Ewha Medical Journal , 2016;.4: 122 – 124.
• 5- Ismail AAA. The insulin autoimmune syndrome (IAS) as a cause of hypoglycaemia: an update on the pathophysiology, biochemical investigations and diagnosis. Clin Chem Lab Med 2016; 54: 1715 - 24.
• 6- Lupsa BC, Chong AY, Cochran EK, et al. Autoimmune Forms of Hypoglycemia. Medicine (Baltimore) 2009; 88: 141 – 53.
• 7- Takei M. Insulin auto-antibodies produced by methimazole treatment in the patients with Graves’ disease. J Tokyo Women’s Med Coll 1980; 50: 54.
• 8- Uchigata Y, Omori Y, Nieda M, et al. HLA-DR4 genotype and insulin-processing in insulin autoimmune syndrome. Lancet 1992; 340: 1467.
• 9- Yamada T, Imai J, Ishigaki Y, Hinokio Y, Oka Y, Katagiri H. Possible relevance of HLA-DRB1*0403 haplotype in insulin autoimmune syndrome induced by alpha-lipoic acid, used as a dietary supplement. Diabetes Care. 2007; 30: 131.
• 10- Ismail AAA. The double whammy of endogenous insulin antibodies in non-diabetic subjects. Clin Chem Lab Med 2008; 46: 153 - 6.
• 11- Uchigata Y, Hirata Y, Iwamoto Y. Drug-induced insulin autoimmune syndrome. Diabetes Res Clin Pract. 2009;83 (1): 19 - 20.