Insulin autoimmune syndrome in a pregnant female

Abstract

Rationale:Insulin autoimmune syndrome (IAS) is an uncommon disorder characterized by hyperinsulinemic hypoglycemia related to insulin-binding autoantibodies. To the best of our knowledge, we report the first case of a pregnant female with IAS.Patient concerns:The 26-year-old patient with Graves disease and 10 weeks pregnant developed IAS after approximately 6 months treatment with methimazole. The patient exhibited recurrent spontaneous hypoglycemia.Diagnoses:On evaluation, laboratory findings detected both high fasting insulin (>1000 mIU/L) and insulin autoantibodies. An oral glucose tolerance test showed elevated insulin concentrations with disproportionately elevated C-peptide levels. The imaging study showed nomasslesionsinthepancreas,and the patient was clinically diagnosed with IAS.Interventions:The patient had an abortion, discontinued methimazole and switched to oral prednisone (30 mg once daily) and propylth- iouracil (100 mg 3 times daily) for 3 months.Outcomes:At the 3-month follow-up visit, hypoglycemic episodes had disappeared and insulin antibody levels were no longer detectable.Lessons:We have described this case and reviewed the relevant literature concerning diagnosis and treatment of IAS. Importantly, this case indicates that clinicians should view pregnancy as another factor of hypoglycemia in IAS.

Comparison of Two Autoimmune Dysglycemia Syndromes: Insulin Autoimmune Syndrome (IAS) and Type B Insulin Resistance Syndrome (B-IRS)

“…The most common autoimmune disorder is Graves' disease, followed by systemic lupus erythematosus (SLE) and scleroderma [17][18][19][20][21][22]. The most common trigger in IAS is drugs, especially those containing a sulfhydryl group [7,8,15,23]. In the present study, 2/13 patients had autoimmune diseases other than Graves' disease, 7/13 took methimazole for Graves' disease, and one received captopril.…”

“…IAS is significantly influenced by the ethnic background of the subject, and the low prevalence of IAS among Caucasians could be explained, at least in part, by the low prevalence of DRB1 * 0406 [14]. In the Chinese Biomedical Literature Database, there were 142 cases of IAS reported between 1994 and May 2017 [15]. The age at onset is variable for IAS.…”

Glucose Changes in a Patient With Insulin Autoimmune Syndrome Demonstrated by Continuous Glucose Monitoring

“…Some patients were diagnosed with IAS after having taken drugs containing sulfhydryl groups, such as methimazole (19). Because our patient had had no prior exposure to these culprit medications, we believe Graves disease was the most likely contributor to his development of IAS.…”

“…Nevertheless, the 1-year follow-up data did show high plasma insulin level and high titer of anti-insulin antibodies, suggesting recurrence of IAS that may warrant avoidance of methimazole. If radioiodine therapy and partial thyroidectomy are not viable options for a patient, a course of treatment with propylthiouracil might be the appropriate alternative because of its lower potential to exacerbate IAS compared to methimazole (19).…”

Insulin autoimmune syndrome in a pregnant female

“…In the article, “Insulin autoimmune syndrome in a pregnant female: A rare case report” [ 1 ] , which appeared in Volume 96, Issue 51 of Medicine , Dr. Jixiong Xu's name appeared incorrectly as Jix Iong Xu.…”