Insulin autoimmune syndrome: a rare cause of hypoglycemia. The case report of the syndrome in pediatric practice
https://doi.org/10.14341/probl2017633189-194
Abstract
Insulin autoimmune syndrome (IAS) is characterized by spontaneous hypoglycemia accompanied by an increased insulin level and presence of insulin autoantibodies while no exogenous insulin was used. This disorder is the third leading cause of hypoglycemia in Southeastern Asia. The disorder develops in individuals with a genetic predisposition, mostly after they had administered drugs containing a sulfhydryl moiety, such as methimazole, penicillin g, etc. The syndrome is typical of adults and extremely rare among children. We report the case of developing IAS in a 3.5-year-old Caucasian girl, possibly induced by pyritinol therapy.
About the Authors
Elena S. KuznetsovaEndocrinology Research Centre
Russian Federation
MD
Oxana V. Pilipenko
Children’s City Clinical Hospital no.1
Russian Federation
MD
Maria А. Melikyan
Endocrinology Research Centre
Russian Federation
MD, PhD
References
1. Hirata Y, Ishizu H, Ouchi N, et al. Insulin autoimmunity in a case of spontaneous hypoglycemia. J Jpn Diabetes Soc. 1970;13(13):312-320.
2. Nasu T, Suzuki R, Okamoto Y, et al. Late postprandial hypoglycemia due to bioactive insulin dissociation from autoantibody leading to unconsciousness in a patient with insulin autoimmune syndrome. Intern Med. 2011;50(4):339-343. doi: 10.2169/internalmedicine.50.4145
3. Uchigata Y, Hirata Y. Insulin autoimmune syndrome (hirata disease). In: Eisenbarth GS, editor. Immunoendocrinology. New York: Humana Press. 2011;343-367.
4. Alves C, Constanca J, de Leon DD, et al. A novel atypical presentation of insulin autoimmune syndrome (hirata’s disease) in a child. J Pediatr Endocrinol Metab. 2013;26(11-12):1163-1166. doi: 10.1515/jpem-2013-0215
5. Gomez Cruz MJ, Jabbar M, Saini N, et al. Severe hypoglycemia secondary to methimazole-induced insulin autoimmune syndrome in a 16 year old african-american male. Pediatr Diabetes. 2012;13(8):652-655. doi: 10.1111/j.1399-5448.2012.00884.x
6. Moreira RO, Lima GA, Peixoto PC, et al. Insulin autoimmune syndrome: case report. Sao Paulo Med J. 2004;122(4):178-180. doi: 10.1590/s1516-31802004000400010
7. Bae SM, Mae MN, Kim EY, et al. Recurrent insulin autoimmune syndrome caused by alpha-lipoic acid in type 2 diabetes. Endocrinol Metab (Seoul). 2013;28(4):326-330. doi: 10.3803/enm.2013.28.4.326
8. Lupsa BC, Chong AY, Cochran EK, et al. Autoimmune forms of hypoglycemia. Medicine (Baltimore). 2009;88(3):141-153. doi: 10.1097/md.0b013e3181a5b42e
9. Uchigata Y, Hirata Y. Insulin autoimmune syndrome (IAS, Hirata disease). Ann Med Interne (Paris). 1999;150(3):245-253.
10. Hirata Y, Uchigata Y. Insulin autoimmune syndrome in Japan. Diabetes Res Clin Pract. 1994;24:suppl:153-157.
11. Dozio N, Scavini M, Beretta A, et al. Imaging of the buffering effect of insulin antibodies in the autoimmune hypoglycemic syndrome. J Clin Endocrinol Metab. 1998;83(2):643-648. doi: 10.1210/jcem.83.2.4599
Supplementary files
|
1. Рис. 1. Результаты орального глюкозотолерантного теста. | |
Subject | ||
Type | Исследовательские инструменты | |
View
(18KB)
|
Indexing metadata ▾ |
|
2. Рис. 2. Динамика лабораторных показателей у пациентки в ходе наблюдения. | |
Subject | ||
Type | Исследовательские инструменты | |
View
(35KB)
|
Indexing metadata ▾ |
|
3. Рис. 3. Строение молекулы пиритинола. | |
Subject | ||
Type | Исследовательские инструменты | |
View
(13KB)
|
Indexing metadata ▾ |
Review
For citations:
Kuznetsova E.S., Pilipenko O.V., Melikyan M.А. Insulin autoimmune syndrome: a rare cause of hypoglycemia. The case report of the syndrome in pediatric practice. Problems of Endocrinology. 2017;63(3):189-194. https://doi.org/10.14341/probl2017633189-194

This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License (CC BY-NC-ND 4.0).