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Insulin autoimmune syndrome: case report.

Rodrigo Oliveira Moreira¹, Giovanna Aparecida Balarini Lima, Patrícia Carla Batista Peixoto

¹Endocrinology Service, Hospital Universitário Clementino Fraga Filho, Universidade Federal do Rio de Janeiro, Rio de Janeiro, Brazil. rom_br@yahoo.com

Sao Paulo Medical Journal = Revista Paulista De Medicina

|November 16, 2004

Summary

Insulin autoimmune syndrome (IAS), a rare cause of hypoglycemia, was identified in South America. This case highlights IAS diagnosis and management challenges, even after pancreatectomy.

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Area of Science:

Endocrinology
Internal Medicine
Pathology

Background:

Insulin autoimmune syndrome (IAS), also known as Hirata disease, is a rare condition causing hypoglycemia.
IAS is characterized by spontaneous hypoglycemia, elevated serum insulin levels, and circulating insulin autoantibodies.
This report details the first identified case of IAS in South America.

Observation:

A 56-year-old male presented with neuroglycopenic symptoms and confirmed hypoglycemia with hyperinsulinemia.
Imaging studies excluded pancreatic tumors, and insulin antibodies were elevated.
Conservative treatments with verapamil and steroids were ineffective.

Findings:

Subtotal pancreatectomy was performed due to treatment failure and contraindications to immunosuppressants.

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Histopathology revealed diffuse beta-cell hypertrophy.

The patient experienced clinical remission of hypoglycemic episodes despite persistent hyperinsulinemia.

Implications:

This case expands the geographic recognition of Insulin Autoimmune Syndrome.
It underscores the importance of considering IAS in unexplained hypoglycemia, even in non-Western populations.
The findings suggest that pancreatectomy can be effective in managing refractory IAS, despite ongoing hyperinsulinemia.