Recurrent Hypoglycemia from Insulin Autoimmune Syndrome

Wong, Sophia; Priestman, Anne; Holmes, Daniel T.

doi:10.1007/s11606-013-2588-9

Cited by 60 publications

(81 citation statements)

References 26 publications

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“…In cohort 2, findings of highly elevated insulin (usually > 100 µIU/mL) with relatively low C‐peptide and positive anti‐insulin antibody were consistent with definition of Hirata disease . The closest differential diagnoses of IAH are insulinoma and oral sulphonylurea ingestion …”

Section: Discussionsupporting

confidence: 52%

Management of Insulin Autoimmune Hypoglycaemia: Single‐centre experience from Western India with systematic review of world literature

Patel

Shah

Ramteke-Jadhav

et al. 2020

Clinical Endocrinology

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Background: Awareness about Insulin Autoimmune Hypoglycaemia (IAH) and its management remains limited. Methodology:We describe two cohorts: Cohort 1 (n = 7) included patients with IAH from a tertiary care centre in India and Cohort 2 (n = 294) included systematic review of published English literature from PubMed. They were compared with our insulinoma patients (n = 41). Results: Cohort 1 included seven female patients where two had drugs (carbimazole and thiocolchicoside) as triggering factors. Except for one patient requiring oral prednisolone, others had spontaneous remission. The unique features from our series are being first case series of IAH from India and reporting of second case of thiocolchicoside triggered IAH.Cohort 2 had 294 patients identified from 149 publications. Mean age was 54 ± 19 years. Thirty-five different triggers were identified from 160 cases.Antithyroid drugs were most common triggers in Japanese patients and most common HLA allele was DRB1*0406, while it was alpha-lipoic acid and HLA DRB1*0403 in non-Asians. Serum Insulin >100 µIU/mL and insulin to C-peptide molar ratio (ICMR) >0.25 had specificity of 100% and 97.5%, respectively, for IAH as compared to insulinoma. 56% patients had remission with complex carbohydrate diet and trigger removal while 43% required immunosuppressants. 70% achieved remission within 6 months. Conclusions:Middle age remains most common age group. Sulfhydryl drugs are most common triggers. Serum Insulin >100 µIU/mL and ICMR > 0.25 in critical sample are good predictors for diagnosis of IAH, which needs to be confirmed by IAA.Conservative management with dietary modification and trigger removal usually suffices in majority. Rests need immunosuppressants. K E Y W O R D Scase series of IAH, insulin auto-antibodies, insulin autoimmune hypoglycaemia, insulin autoimmune syndrome, management of IAH, systematic review of IAH

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Section: Discussionsupporting

confidence: 52%

Management of Insulin Autoimmune Hypoglycaemia: Single‐centre experience from Western India with systematic review of world literature

Patel

Shah

Ramteke-Jadhav

et al. 2020

Clinical Endocrinology

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“…Insulin/C-peptide molar ratio was found to be >1 in most of the cases of IAS (5). There are at least 2 cases (one with type 2 diabetes and had A1C of 7%) with IAS where insulin/C-peptide was <1 (6).…”

Section: Discussionmentioning

confidence: 95%

Insulin Autoimmune Syndrome: a rare cause of postprandial hypoglycemia

Sahni

Trivedi

Omer

2016

Endocrinology, Diabetes &Amp; Metabolism Case Reports

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SummaryA 65-year-old obese Caucasian woman presented with symptomatic postprandial hypoglycemic episodes, resolution of symptoms with carbohydrate intake and significantly elevated anti-insulin antibody levels. She did not have any evidence for the use of oral antidiabetic medications, insulin, herbal substances, performing strenuous exercise or history of bariatric surgery. Fingerstick blood glucose readings revealed blood sugar of 35 mg/dL and 48 mg/dL, when she had these symptoms. Her medical history was significant for morbid obesity, hypothyroidism and gastro esophageal reflux disease. Her home medications included levothyroxine, propranolol and omeprazole. A blood sample obtained during the symptoms revealed the following: fingerstick blood sugar 38 mg/dL, venous blood glucose 60 mg/dL (normal (n): 70–99 mg/dL), serum insulin 202 IU/mL (n: <21), proinsulin 31.3 pmol/L (n: <28.9), C-peptide 8 ng/mL (n: 0.9–7), beta-hydroxybutyrate 0.12 mmol/L (n: 0.02–0.27) anti-insulin antibody >45.4 U/mL (n: <0.4). The result obtained while screening for serum sulfonylurea and meglitinides was negative. The repeated episodes of postprandial hypoglycemia associated with significantly elevated anti-insulin antibodies led to a diagnosis of insulin antibody syndrome (IAS). Significant improvement of hypoglycemic symptoms and lower anti-insulin antibody levels (33 U/mL) was noted on nutritional management during the following 6 months. Based on a report of pantoprazole-related IAS cases, her omeprazole was switched to a H2 receptor blocker. She reported only two episodes of hypoglycemia, and anti-insulin antibody levels were significantly lower at 10 U/mL after the following 12-month follow-up.Learning points:Initial assessment of the Whipple criteria is critical to establish the clinical diagnosis of hypoglycemia accurately.Blood sugar monitoring with fingerstick blood glucose method can provide important information during hypoglycemia workup.Autoimmune hypoglycemia is a rare cause of hypoglycemia, which can be diagnosed on high index of clinical suspicion and systematic evaluation.

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“…When insulin autoantibodies bind to insulin, the half-life increases from 5 minutes to hours [7], while the half-life of the C-peptide usually remains shorter and una ected (30−35 minutes) giving rise to an elevated insulin level while having a nonelevated C-peptide and proinsulin and changing the insulin to C-peptide ratio to >1. It is also possible that the insulin autoantibody is capable of binding to endogenous proinsulin and C-peptide as well, delaying their half-lives, and possibly keeping the insulin to C-peptide ratio in the normal range of <1 [8,9]. In our patient, the proinsulin was below reference range indicating an inhibitory e ect on the proinsulin synthesis due to the hyperinsulinaemic period or a possible nonextended half-life of proinsulin which helped to clear it before the onset of hyperinsulinaemia.…”

Section: Discussionmentioning

confidence: 99%

Insulin Autoimmune Syndrome in a 25-Year-Old, Previously Healthy Kuwaiti Man

Alrashidi

Alessa²

2019

Case Reports in Endocrinology

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Insulin autoimmune syndrome (IAS) is a disease characterized by hyperinsulinaemic hypoglycaemia associated with autoantibodies against endogenous insulin. We have described a case of a 25-year-old, previously healthy Kuwaiti man who was admitted to the Mubarak Al-Kabeer hospital with a history of recurrent hypoglycaemia. e patient revealed that he had taken several di erent injectable anabolic steroids and growth hormone with oral amino acids and other tablets (fat burners) for bodybuilding in the last two months. He denied knowingly using insulin or insulin analogues. e patient had elevated fasting insulin level (>301 uIU/mL) and elevated insulin autoantibodies (>100.0 IU/mL). A er appropriate work-up, he was diagnosed with IAS. A er treatment with prednisolone (1 mg/kg/day), the patient had complete recovery. In patients with repeated hypoglycaemia, IAS should be considered in the di erential diagnosis. Glucocorticoid therapy can be e ective for the treatment of hypoglycaemia in patients with IAS.

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Recurrent Hypoglycemia from Insulin Autoimmune Syndrome

Cited by 60 publications

References 26 publications

Management of Insulin Autoimmune Hypoglycaemia: Single‐centre experience from Western India with systematic review of world literature

Management of Insulin Autoimmune Hypoglycaemia: Single‐centre experience from Western India with systematic review of world literature

Insulin Autoimmune Syndrome: a rare cause of postprandial hypoglycemia

Insulin Autoimmune Syndrome in a 25-Year-Old, Previously Healthy Kuwaiti Man

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