Brief report
Insulin autoimmune syndrome: the second Dutch case

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Abstract

The insulin autoimmune syndrome (IAS) is the third leading cause of spontaneous hypoglycaemia in Japan. Here, we describe the second Dutch case. An 81-year-old man was analysed for collapse and attacks of disorientation, and was found to be hypoglycaemic during such an episode. The performed fasting test appeared to be positive. High levels of insulin and C-peptide postprandially and insulin antibodies were found. IAS was diagnosed.

Introduction

The insulin autoimmune syndrome (IAS) is characterised by the following diagnostic criteria: spontaneous hypoglycaemia without exogenous insulin administration, high levels of total immunoreactive insulin and high titres of insulin antibodies.

The first report of this syndrome was published in 1970 in Japan by Hirata et al. [1]. Since 1970, about 200 similar cases have been reported in Japan, where IAS is the third leading cause of spontaneous hypoglycaemia, after insulinoma and extrapancreatic neoplasms [2]. In Europe and the USA, IAS is much less common: only 21 cases have been published [3], [4], [5]. In Holland, the first patient with IAS was published in 1996 by Schlemper et al. [3]. Here, we report the second Dutch case of IAS.

Section snippets

Case report

An 81-year-old Caucasian man presented himself to the Department of Internal Medicine for evaluation of the following symptoms. For a few months he experienced episodes of confusion and sweating. During such attacks, he talked incoherently and felt dizzy; he also collapsed a couple of times. The frequency of these episodes varied from once a day to once a week; duration was unknown. While recovering he always found himself eating.

His medical history revealed nine episodes of gastric bleedings,

Discussion

At first we thought the patient had recurrent hypoglycaemia because of his renal dysfunction, although this is very rare. It was not until the C-peptide and the 125I tracer binding with serum were known that IAS was diagnosed. The high C-peptide in the blood postprandially, and the fact that we did not find injection sites made the possibility of exogenous insulin administration very unlikely.

IAS patients are relatively hyperglycaemic immediately after eating, because of the binding of the

Acknowledgements

We thank Alice J. Horsten for her help in the language correction of the manuscript.

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