ABSTRACT
SUMMARY Insulin autoimmune syndrome (IAS, Hirata's disease) is a rare hypoglycemic disorder characterized by spontaneous hypoglycemia associated with extremely high circulating insulin levels and positive anti-insulin antibody results. Thus far, most cases have been reported in Asian countries, notably Japan, with few cases reported in western countries. As a possible cause, it is associated with the use of drugs containing sulfhydryl radicals, such as captopril. This report refers to a 63-year-old female Brazilian patient with a history of postprandial hypoglycemia. After extensive investigation and exclusion of other causes, her hyperinsulinemic hypoglycemia was considered to have likely been induced by captopril. Most cases of IAS are self-limiting. However, dietary management, corticosteroids, plasmapheresis, and rituximab have already been used to treat patients with IAS. In our case, after discontinuation of captopril, an initial decrease in insulin autoantibody levels was observed followed by improvement in episodes of hypoglycemia. Although it is a rare disease, IAS should be considered in the differential diagnosis of endogenous hyperinsulinemic hypoglycemia. Patients with suspected IAS must be screened for autoimmunity-related drugs for insulin. Initial clinical suspicion of IAS can avoid unnecessary costs associated with imaging examinations and/or invasive surgical procedures.
Subject(s)
Humans , Female , Middle Aged , Autoimmune Diseases/chemically induced , Captopril/adverse effects , Hypoglycemia/chemically induced , Hypoglycemia/immunology , Insulin Antibodies/drug effects , Antihypertensive Agents/adverse effects , Autoimmune Diseases/ethnology , Autoimmune Diseases/immunology , Syndrome , Blood Glucose/analysis , Brazil , Hypoglycemia/ethnology , Insulin Antibodies/immunologyABSTRACT
Se presenta una mujer de 33 años con una historia l1 meses de episodios recurrentes de hipoglucemia severa, asociados a anticuerpos anti-insulina y valores variables de peptido-C. Una extracción ácido-alcohólica de suero mostró un nível basal de insulina de 1.600 uU/ml. La insulina caracterizada por HPLC demostró ser insulina humana. Los anticuerpos fueron específicos para la insulina humana con una subpoblación que reaccionaba con insulina bovina y porcina (IgG, cadena liviana k). Al declinar los síntomas, el tratamiento con plasmaféresis negativizó el título con rapidez. Un seguimiento prolongado demostró la ausencia de recidivas.