The peculiarity of the presented case is the association of neuroglycopenic symptoms of hypoglycemia not preceded by autonomic hypoglycemic symptoms, with values of insulinemia and C peptide higher than the threshold for defining insulin-mediated hypoglycemia, but not so high compared to the severity and persistence of hypoglycemia and refractoriness of hypoglycemic episodes to carbohydrate ingestion and parenteral glucose administration. Here, INS is linked to Hypoglycemia.