Insulin Autoimmune Syndrome Induced by α–Lipoic Acid in a Caucasian Woman: Case Report

Bresciani, Enrico; Bussi, Anna Rosa; Bazzigaluppi, Elena; Balestrieri, G

doi:10.2337/dc11-0600

Cited by 27 publications

(23 citation statements)

References 5 publications

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“…The syndrome is rare in Caucasian/non-Japanese populations: 60 cases in Caucasians and 20 cases in East Asians have been reported to date [2] [3]. To the best of our knowledge, the present case is the third reported in Italy (although the subject is not of Italian origin) and the first with HLA typing [4][5].…”

Section: Introductionmentioning

confidence: 76%

Insulin Autoimmune Syndrome (Hirata Disease): Case Report in a Caucasian Patient with New-Onset Diabetes

Sudano¹,

Turchi²,

Sossai³

2012

CMD

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An unusual cause of spontaneous hypoglycemia is Insulin Autoimmune Syndro me (or Hirata Disease), characterized by high levels of insulinemia and circulat ing autoantibodies to insulin in subjects without prior insulin administration. In Western Countries less than 70 cases have been published so far. Here we report a case where IAS was the prodromical stage of new onset type 1 diabetes. HLA typing in IAS was rarely reported in Western cases, here we also discuss the peculiar HLA pattern of our patient.

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Section: Introductionmentioning

confidence: 76%

Insulin Autoimmune Syndrome (Hirata Disease): Case Report in a Caucasian Patient with New-Onset Diabetes

Sudano¹,

Turchi²,

Sossai³

2012

CMD

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“…Recently an increasing number of reports have referred to an association with IAS and α lipoic acid assumption. [2][3][4] Other secondary forms of IAS have been associated with autoimmune diseases as rheumatoid arthritis, systemic lupus erythematosus, polimiositis, vasculitis or with hematological diseases (multiple myeloma, monoclonal gammopathy of undetermined significance), liver disease and neoplasms. 1,5,6 It is very hard to estimate the number of patients affected by IAS out of Japan reported in the Literature.…”

Section: Discussionmentioning

confidence: 99%

“…1,5,6 It is very hard to estimate the number of patients affected by IAS out of Japan reported in the Literature. Uchigata 4,[8][9][10][11] This suggests that the incidence of IAS is extremely rare in Italy.…”

Section: Discussionmentioning

confidence: 99%

Primary insulin autoimmune syndrome in an Italian woman: a case report

et al. 2015

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Insulin autoimmune syndrome (IAS) is a rare syndrome characterized by fasting or postprandial hypoglycemia, high levels of anti-insulin antibodies and high concentration of total serum immunoreactive insulin. It is relatively known in Japan, rare in remaining Asia and it is extremely uncommon in Western countries, being characterized by a different race-related incidence and associated with HLADR4 alleles. Usually IAS is related to particular drugs, or to autoimmune, rheumatologic or hematological diseases, while it is very rare as a primary form. Here we described a case of an Italian woman affected by a primary form of Hirata syndrome.

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“…In conclusion, new informative cases of IAS in Caucasians continued to appear in the world literature [27,28,69,70,75,[90][91][92][93][94][95][96][97][98][99]. The very wide range of their clinical presentations and biochemical findings re-emphasised that IAS is a highly heterogeneous syndrome.…”

Section: Comments and Conclusionmentioning

confidence: 98%

“…An additional drug namely α-lipoic acid (ALA) which also contains sulfur (cyclic disulfide) was also reported to cause IAS in Japanese [19]; this was quickly followed by numerous reports in patients all of whom were also Japanese [21][22][23][24][25][26]. The first case in a Caucasian (Italian) appeared in Sicily in 2011 [27]; however, within 22 months, six more cases were also identified by the same medical centre in Sicily [28]. Lipoic-acid is sold over-the-counter (OTC) and on the Internet as a universal antioxidant drug/free radical scavenger because of its unique solubility in both water and fat; it is also prescribed for conditions such as peripheral polyneuropathy [26].…”

Section: Introductionmentioning

confidence: 94%

The insulin autoimmune syndrome (IAS) as a cause of hypoglycaemia: an update on the pathophysiology, biochemical investigations and diagnosis

Ismail¹

2016

Clinical Chemistry and Laboratory Medicine (CCLM)

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Abstract:Insulin autoimmune syndrome (IAS) is considered to be very rare in Caucasians. Understanding its pathophysiology is paramount in (a) appreciating its potential impact on analyses of pancreatic hormones and (b) explaining its highly variable clinical manifestations in non-diabetic, non-acutely ill patients with indeterminate hypoglycaemia. The underlying aetiology of IAS is the presence of variable affinity/avidity endogenous insulin antibodies in significant amounts. The two types of insulin antibodies namely antibodies which bind insulin and/ or proinsulin(s) and receptor antibodies (insulin mimetic) will be discussed. Their biochemical and immunological roles in causing hypoglycaemia will be highlighted. Clinical manifestations of IAS can vary from mild and transient to spontaneous, severe and protracted hypoglycaemia necessitating in extreme cases plasmapheresis for glycaemic control. Antibodies of IAS can interfere in pancreatic immunoassay tests causing erroneous and potentially misleading results. Thorough testing for endogenous insulin antibodies must be considered in the investigations of non-diabetic, non-acutely ill patients with indeterminate and/or unexplained hypoglycaemia.Keywords: endogenous insulin antibodies; Hirata's disease, insulin autoimmune hypoglycaemia; hypoglycaemia in non-diabetic; insulin autoimmune syndrome (IAS); interference in immunoassays of insulin, proinsulin and C-peptide; wrong immunoassay results .

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Insulin Autoimmune Syndrome Induced by α–Lipoic Acid in a Caucasian Woman: Case Report

Cited by 27 publications

References 5 publications

Insulin Autoimmune Syndrome (Hirata Disease): Case Report in a Caucasian Patient with New-Onset Diabetes

Insulin Autoimmune Syndrome (Hirata Disease): Case Report in a Caucasian Patient with New-Onset Diabetes

Primary insulin autoimmune syndrome in an Italian woman: a case report

The insulin autoimmune syndrome (IAS) as a cause of hypoglycaemia: an update on the pathophysiology, biochemical investigations and diagnosis

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