Preview

Problems of Endocrinology

Advanced search

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. Kuznetsova

Endocrinology 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

Views: 4376


ISSN 0375-9660 (Print)
ISSN 2308-1430 (Online)