DOI: http://dx.doi.org/10.18203/2349-3933.ijam20212871

Hypoglycemia due to insulin autoimmune syndrome (Hirata’s disease): a rare cause of hypoglycaemia

Het V. Patel, Sunil Kumar, Kalpesh Moradiya, Vidhi Shah

Abstract


Although the most common cause of recurrent hypoglycaemia is diabetes mellitus as patient is on antidiabetic medications which can be prevented by modification of antidiabetic doses, nutrition therapy and lifestyle modifications. Some endogenous hyperinsulinemic conditions like insulinoma, functional beta cell disorders and insulin autoimmune syndromes, hormonal deficiencies can cause serious and sometimes life threatening hypoglycaemia. So further laboratory evaluation like plasma/serum glucose levels, c-peptide levels, insulin levels, insulin antibodies and imaging studies are needed to evaluate unexplained hypoglycaemia. Here we report a case of insulin autoimmune syndrome in a 67 year old Indian male who had presented to us with multiple episodes of spontaneous hypoglycaemia. On further workup, the patient was found to have endogenous hyperinsulinemic hypoglycemia. As the patient’s abdominal imaging revealed no apparent cause of EHH, on further evaluation he came positive for insulin antibodies. Patient was diagnosed as IAS and he was given frequent small meals and complex carbohydrate diet and he had improved symptomatically. The incidence of IAS is most common in Japan and very few cases have been reported from India, so it should be kept in differential diagnosis of recurrent hypoglycaemia.


Keywords


Hypoglycemia, Insulin autoimmune syndrome, Antidiabetic, Hirata’s disease

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References


Uchigata Y, Hirata Y, Iwamoto Y. Drug-induced insulin autoimmune syndrome. Diabetes Res Clin Pract. 2009;83(1):e19-20.

Hirata Y, Censi S, Mian C, Betterle C. Insulin autoimmune syndrome: from diagnosis to clinical management. Ann Transl Med. 2018;6(17):335.

Uchigata Y, Hirata Y. Insulin autoimmune syndrome (IAS, Hirata disease). Ann Med Interne (Paris). 1999;150(3):245-53.

Lupsa BC, Chong AY, Cochran EK, Soos MA, Semple RK, Gorden P. Autoimmune forms of hypoglycemia. Medicine. 2009;88(3):141-153.

Gopal K, Priya G, Gupta N, Praveen EP, Khadgawat R. A case of autoimmune hypoglycemia outside Japan: Rare, but in the era of expanding drug-list, important to suspect. Indian J Endocrinol Metab. 2013;17(6):1117-9.

Raizada N, Rahaman SH, Kandasamy D, Jyotsna VP. Rare association of insulin autoimmune syndrome with ankylosing spondylitis. Endocrinol Diabetes Metab Case Rep. 2015;2015:150090.

Patwari P, Narasimhan A, Srinivasan R. A case of severe hypoglycaemia in a patient with hyperthyroidism. Endocrine Abstr. 2013;32:996-9.

Burch HB, Clement S, Sokol MS, Landry F. Reactive hypoglycemic coma due to insulin autoimmune syndrome: case report and literature review. Am J Med. 1992;92(6):681-5.

Lanas A, Paredes A, Espinosa C, Caamaño E, Pérez-Bravo F, Pinto R, et al. Insulin autoimmune syndrome: Report of two cases. Rev Med Chil. 2015;143(7):938-42.

Donegan DM, Vella A. 69-year-old man with recurrent episodes of confusion and diaphoresis. Mayo Clin Proc. 2013;88(4):410-3.

Goldman J, Baldwin D, Rubenstein AH, Klink DD, Blackard WG, Fisher LK, et al. Characterization of circulating insulin and proinsulin-binding antibodies in autoimmune hypoglycemia. J Clin Invest. 1979;63(5):1050-9.

Wang YL, Yao PW, Zhang XT, Luo ZZ, Wu PQ, Xiao F. Insulin Autoimmune Syndrome: 73 Cases of Clinical Analysis. Chin Med J (Engl). 2015;128(17):2408-9.

Wong SL, Priestman A, Holmes DT. Recurrent hypoglycemia from insulin autoimmune syndrome. J Gen Intern Med. 2014;29(1):250-4.

Cavaco B, Uchigata Y, Porto T, Amparo-Santos M, Sobrinho L, Leite V. Hypoglycaemia due to insulin autoimmune syndrome: report of two cases with characterisation of HLA alleles and insulin autoantibodies. Eur J Endocrinol. 2001;145(3):311-6.