AACE endocrinology and diabetes · 2025
A Case of Hirata Syndrome in Pregnancy Induced by α-Lipoic Acid: Diagnosis and Management.
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Viti Luca, Tartaglione Linda, Rizzi Alessandro, Perri Alessandro, Mazzotta Francesco, De Carolis Sara, Vento Giovanni, Lanzone Antonio, Pitocco Dario
Аннотация
BACKGROUND/OBJECTIVE: Insulin autoimmune syndrome (IAS), also known as "Hirata disease", is an uncommon cause of hypoglycemia, linked to the presence of insulin autoantibodies (IAAs) in subjects who are not taking insulin and with no evidence of histopathological abnormalities of the pancreatic islets.
CASE REPORT: We report a case of a 25-year-old pregnant woman who displayed recurrent symptomatic hypoglycemia that began after treatment with α-lipoic acid. One episode of severe hypoglycemia was documented, which resulted in syncope. Magnetic resonance imaging showed no pancreatic lesions and an IAA assay was performed, which revealed elevated levels of immunoglobulin G IAAs, confirming the diagnosis of IAS. Corticosteroid therapy and discontinuation of lipoic acid resulted in a gradual reduction of hypoglycemic episodes and the disappearance of IAAs. After the delivery, the newborn developed significant hypoglycemia, which gradually resolved. A significative titer of IAAs was detected in the first weeks of life.
DISCUSSION: Corticosteroid therapy should be considered in patients with hypoglycemia who have recently taken lipoic acid or any other medications strongly associated with IAS, after other diagnoses have been excluded. Therefore, it is appropriate to initiate neonatal glucose monitoring immediately after delivery in mothers with IAS during pregnancy to prevent and treat neonatal hypoglycemia after possible transplacental passage of maternal immunoglobulin G antibodies.
CONCLUSION: This is the first case of demonstrated placental transfer of IAAs to a newborn who showed significant hypoglycemia and high titers of IAAs.
CASE REPORT: We report a case of a 25-year-old pregnant woman who displayed recurrent symptomatic hypoglycemia that began after treatment with α-lipoic acid. One episode of severe hypoglycemia was documented, which resulted in syncope. Magnetic resonance imaging showed no pancreatic lesions and an IAA assay was performed, which revealed elevated levels of immunoglobulin G IAAs, confirming the diagnosis of IAS. Corticosteroid therapy and discontinuation of lipoic acid resulted in a gradual reduction of hypoglycemic episodes and the disappearance of IAAs. After the delivery, the newborn developed significant hypoglycemia, which gradually resolved. A significative titer of IAAs was detected in the first weeks of life.
DISCUSSION: Corticosteroid therapy should be considered in patients with hypoglycemia who have recently taken lipoic acid or any other medications strongly associated with IAS, after other diagnoses have been excluded. Therefore, it is appropriate to initiate neonatal glucose monitoring immediately after delivery in mothers with IAS during pregnancy to prevent and treat neonatal hypoglycemia after possible transplacental passage of maternal immunoglobulin G antibodies.
CONCLUSION: This is the first case of demonstrated placental transfer of IAAs to a newborn who showed significant hypoglycemia and high titers of IAAs.