2015, Number 4
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Med Int Mex 2015; 31 (4)
Autoimmune hypoglycemia (Hirata’s syndrome). A case report in Mexico City
Orozco-Olguín P, Romero-Morelos RD, Quintero-Beltrán M, Ahumada-Ayala M
Language: Spanish
References: 17
Page: 465-471
PDF size: 815.62 Kb.
ABSTRACT
Autoimmune hypoglycemia (Hirata’s syndrome) is an extremely rare cause of hypoglycemia, well known in oriental populations. It’s frequently associated to certain drugs and rheumatologic diseases. This would be the first case report in Mexico. This paper reports the case of a 71-year-old Mexican female, who had 15 days before admission a lower respiratory tract infection treated and relieved with azithromycin. Patient arrived to the emergency department for loss of consciousness without loss of sphincter control or abnormal movements. A plasma glucose of 32 mg/dL was recorded, administration of 50g of glucose IV, with full recovery of state of alert. She has a history of several days before admission of spontaneous diaphoresis, disorientation, dysarthria and somnolence. Physical examination was entirely normal. Laboratory results revealed extremely high insulin levels, as well as C-peptide and proinsulin. Imaging studies were negative for the presence of pancreatic tumors. Serum anti-insulin antibodies were positive. HLA typing revealed DRB1°04 and DQB1°03°02, commonly associated with Hirata’s syndrome.
REFERENCES
Martínez-Ortega AJ, et al. Enfermedad de Hirata: a propósito de un caso. Med Clin (Barc) 2013.
Chena JA, et al. Hipoglucemia por hiperinsulinismo endógeno autoinmune. A propósito de un caso y revisión de la bibliografía. Endocrinol Nutr 2002;49:171-175.
Uchigata Y, Hirata Y. Insulin autoimmune syndrome (IAS, Hirata disease) Endocrine and Organ Specific Autoimmunity Chapter 7. 2012;133-148.
Arichika D, et al. Insulin autoimmune syndrome in a health supplement user: the effectiveness of cornstarch therapy for treating hypoglycemia. Intern Med 2013;52:369-372.
Tsuneyoshi K, Moriwaki Y. Insulin autoimmune syndrome in a patient with ANCA-associated glomerulonephritis. Diabetes Metab Syndr Obes 2008;1:1-2.
Okazaki-Sakai S, Yoshimoto S, Yagi K, Wakasugi T, et al. Insulin autoimmune syndrome caused by an adhesive skin patch containing loxoprofen-sodium. Intern Med 2013;52:2447-2451.
Matsushita S, Takahashi K. Allele specificity of structural requirement for peptides bound to HLA-DRBI*0405 and -DRBI*0406 complexes: Implication for the HLA-associated susceptibility to methimazole-induced insulin autoimmune syndrome. J Exp Med 1994;180:873-883.
Oliveira MR, Balarini LGA, Batista PPA, Fletuss FML, Valsman M. Insulin autoimmune syndrome: case report. Sau Paulo Med J 2004;122:178-180.
Takeuchi Y, Miyamoto T. Insulin autoimmune syndrome possibly caused by alpha lipoic acid. Intern Med 2007;46:237-239.
Bae SM, Bae MN, et al. Recurrent insulin autoimmune syndrome caused by α-lipoic acid in type 2 diabetes. Endocrinol Metab 2013;28:326-330.
Roh E, Kim YA, Ku EJ. Two cases of methimazole-Induced insulin autoimmune syndrome in Graves’ disease. Endocrinol Metab 2013;28:55-60.
Cavaco B, Uchigata Y, Porto T. Hypoglycaemia due to insulin autoimmune syndrome: report of two cases with characterisation of HLA alleles and insulin autoantibodies. Eur J Endocrinol 2001;145:311-316.
Gomez-Cruz MJ, et al. Severe hypoglycemia secondary to methimazole-induced insulin autoinmune syndrome in a 16-year-old African-American male. Pediatric Diabetes 2012;13:652-655.
Halsall DJ, et al. Hypoglycemia due to an insulin-binding antibody in a patient with an IgA kappa myeloma. J Clin Endocrinol Metab 2007;92:2013-2016.
Alves C, Constanza J, De León D, Snider K, Stanley C. A novel atypical presentation of insulin autoimmune syndrome (Hirata’s disease) in a child. J Pediatr Endocrinol Metab 2013;26:1163-1166.
Kang SM, et al. Type B insulin-resistance syndrome presenting as autoimmune hypoglycemia, associated with systemic lupus erytematosus and interstitial lung disease. Korean J Intern Med 2013;28:98-102.
Lupsa BC, Chong AY, Cochran EK, Soos MA, et al. Autoimmune forms of hypoglycemia. Md-Journal, Lippincott Williams & Wilkins, 2009;141-152.