Autoimmune Hypoglycemia, though very rare in India, but can be challenging to manage. Insulin autoimmune syndrome (IAS) should be considered in any patient with hypoglycemia in the setting of unsuppressed insulin levels associated with anti-insulin or anti insulin receptor antibodies. We are reporting the clinical course of one such case of insulin autoimmune syndrome, who was initially treated with glucocorticoids. The patient relapsed and was later on treated effectively with Azathioprine for glucocorticoids failure and toxicity.

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