Fux-Otta Carolina, Reynoso Raúl, Chedraui Peter, Estario Paula, Estario María E, Iraci Gabriel, Ramos Noelia, Di Carlo Mariana, Gamba Victoria, Sembaj Adela
Departamento de Endocrinología y Diabetes, Hospital Universitario de Maternidad y Neonatología, Facultad de Ciencias Médicas, Universidad Nacional de Córdoba, Córdoba, Argentina.
Unidad de Conocimiento Traslacional Hospitalaria (UCTH), Facultad de Ciencias Médicas, Universidad Nacional de Córdoba, Córdoba, Argentina.
Case Rep Womens Health. 2024 Aug 10;43:e00644. doi: 10.1016/j.crwh.2024.e00644. eCollection 2024 Oct.
Insulin autoimmune syndrome or Hirata's disease is a rare condition characterized by hypoglycemia associated with endogenous autoimmune hyperinsulinism. This report concerns the case of a 28-year-old Latin American woman with Graves' disease who developed insulin autoimmune syndrome and then subsequently became pregnant. She displayed symptoms related to severe hypoglycemia due to hyperinsulinemia, elevated C-peptide, and anti-insulin antibodies. Prior to pregnancy she was treated with corticosteroids and had ablative treatment with iodine-131. During follow-up of both conditions, the patient became pregnant, and clinically and biochemically hyperthyroid, for which total thyroidectomy was performed during the second trimester of pregnancy. Anti-insulin antibodies, blood glucose, and C-peptide remained normal throughout pregnancy. At 40 weeks of gestation she gave birth to a healthy female newborn with normal blood glucose values. Molecular genetic analysis determined the following genotypes: HLA-DRB104:01 in the mother; and HLA-DRB1**08:02 in the daughter. Because some HLA-DRB104 alleles are associated with susceptibility to insulin autoimmune syndrome induced by environmental factors, the patient was advised regarding the future use of drugs with a sulfhydryl group and possible triggering factors for insulin autoimmune syndrome. At 6-month follow-up the daughter presented normal growth and development, as well as normal plasma glucose values, and this remained the case at five-year follow-up.
胰岛素自身免疫综合征或平田病是一种罕见病症,其特征为低血糖与内源性自身免疫性高胰岛素血症相关。本报告涉及一名28岁患有格雷夫斯病的拉丁美洲女性病例,该患者患上胰岛素自身免疫综合征后怀孕。她表现出因高胰岛素血症、C肽升高和抗胰岛素抗体导致的严重低血糖相关症状。怀孕前她接受了皮质类固醇治疗,并进行了碘-131消融治疗。在对这两种病症的随访期间,患者怀孕,临床和生化检查显示甲状腺功能亢进,因此在妊娠中期进行了全甲状腺切除术。整个孕期抗胰岛素抗体、血糖和C肽均保持正常。妊娠40周时,她生下一名血糖值正常的健康女婴。分子遗传学分析确定了以下基因型:母亲为HLA-DRB104:01;女儿为HLA-DRB1**08:02。由于某些HLA-DRB104等位基因与环境因素诱导的胰岛素自身免疫综合征易感性相关,因此就未来使用含巯基药物及胰岛素自身免疫综合征可能的触发因素向患者提供了建议。在6个月的随访中,女儿生长发育正常,血糖值也正常,在5年随访中情况依然如此。