Insulin autoimmune syndrome

case report

Authors

  • Rodrigo Oliveira Moreira Hospital Universitário Clementino Fraga Filho, Universidade Federal do Rio de Janeiro
  • Giovanna Aparecida Balarini Lima Hospital Universitário Clementino Fraga Filho, Universidade Federal do Rio de Janeiro
  • Patrícia Carla Batista Peixoto Hospital Universitário Clementino Fraga Filho, Universidade Federal do Rio de Janeiro
  • Maria Lucia Fleiuss Farias Hospital Universitário Clementino Fraga Filho, Universidade Federal do Rio de Janeiro
  • Mario Vaisman Hospital Universitário Clementino Fraga Filho, Universidade Federal do Rio de Janeiro

Keywords:

Hypoglycemia, Hyperinsulinism, Insulin, Pancreatectomy, Insulin antibodies

Abstract

CONTEXT: Insulin autoimmune syndrome (IAS, Hirata disease) is a rare cause of hypoglycemia in Western countries. It is characterized by hypoglycemic episodes, elevated insulin levels, and positive insulin antibodies. Our objective is to report a case of IAS identified in South America. CASE REPORT: A 56-year-old Caucasian male patient started presenting neuroglycopenic symptoms during hospitalization due to severe trauma. Biochemical evaluation confirmed hypoglycemia and abnormally high levels of insulin. Conventional imaging examinations were negative for pancreatic tumor. Insulin antibodies were above the normal range. Clinical remission of the episodes was not achieved with verapamil and steroids. Thus, a subtotal pancreatectomy was performed due to the lack of response to conservative treatment and because immunosuppressants were contraindicated due to bacteremia. Histopathological examination revealed diffuse hypertrophy of beta cells. The patient continues to have high insulin levels but is almost free of hypoglycemic episodes.

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Author Biographies

Rodrigo Oliveira Moreira, Hospital Universitário Clementino Fraga Filho, Universidade Federal do Rio de Janeiro

MD, MSc. Endocrinology Service, Hospital Universitário Clementino Fraga Filho, Universidade Federal do Rio de Janeiro, Rio de Janeiro, Brazil.

Giovanna Aparecida Balarini Lima, Hospital Universitário Clementino Fraga Filho, Universidade Federal do Rio de Janeiro

MD. Endocrinology Service, Hospital Universitário Clementino Fraga Filho, Universidade Federal do Rio de Janeiro, Rio de Janeiro, Brazil.

Patrícia Carla Batista Peixoto, Hospital Universitário Clementino Fraga Filho, Universidade Federal do Rio de Janeiro

MD. Endocrinology Service, Hospital Universitário Clementino Fraga Filho, Universidade Federal do Rio de Janeiro, Brazil.

Maria Lucia Fleiuss Farias, Hospital Universitário Clementino Fraga Filho, Universidade Federal do Rio de Janeiro

MD, PhD. Endocrinology Service, Hospital Universitário Clementino Fraga Filho, Universidade Federal do Rio de Janeiro, Rio de Janeiro, Brazil.

Mario Vaisman, Hospital Universitário Clementino Fraga Filho, Universidade Federal do Rio de Janeiro

MD, PhD. Endocrinology Service, Hospital Universitário Clementino Fraga Filho, Universidade Federal do Rio de Janeiro, Rio de Janeiro, Brazil.

References

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

Redmon JB, Nuttall FQ. Autoimmune hypoglycemia. Endocrinol Metab Clin North Am. 1999;28(3):603-18.

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

Thompson GB. Diagnosis and management of insulinomas. Endocr Practice. 2002;8:385-6.

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Published

2004-07-07

How to Cite

1.
Moreira RO, Lima GAB, Peixoto PCB, Farias MLF, Vaisman M. Insulin autoimmune syndrome: case report. Sao Paulo Med J [Internet]. 2004 Jul. 7 [cited 2025 Oct. 16];122(4):178-80. Available from: https://periodicosapm.emnuvens.com.br/spmj/article/view/2517

Issue

Section

Case Report