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1.
Diabetes treatment in two pregnant women with permanent neonatal diabetes mellitus due to a KCNJ11 mutation.
Stanik, J, Barak, L, Dankovcikova, A, Valkovicova, T, Skopkova, M, Gasperikova, D
Diabetic medicine : a journal of the British Diabetic Association. 2020;(11):1956-1958
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2.
Exogenous Insulin Injection-Induced Stiff-Person Syndrome in a Patient With Latent Autoimmune Diabetes: A Case Report and Literature Review.
Lee, YY, Lin, CW, Chen, IW
Frontiers in endocrinology. 2020;:594
Abstract
Stiff-person syndrome (SPS) is highly associated with anti-glutamic acid decarboxylase (GAD) antibody. However, GAD antibodies alone appear to be insufficient to cause SPS, and they possibly are involved in only part of its pathophysiology. It is suspected that the symptoms of SPS get precipitated by external stimuli. Here, we briefly introduce the case of a patient with latent autoimmune diabetes who developed SPS through the action of subcutaneously injected insulin. A 43-year-old man was diagnosed with diabetes and initially well-controlled with oral hypoglycemic agents but progressed to requiring insulin within 1 year of diagnosis. Two months after the initiation of basal insulin therapy, he presented with abdominal stiffness and painful muscle spasms, involving the lower limbs, which resulted in walking difficulty, and thus, he refused insulin injections thereafter. He had been treated with oral anti-diabetic agents instead of insulin for 10 years until premixed insulin twice daily was started again due to poor diabetes control. Immediately after insulin injection, abdominal muscle rigidity and spasms were noted. When insulin was not administered, frequent episodes of diabetic ketoacidosis occurred. Serum GAD antibody test was positive and there was no positivity for islet antigen-2 antibody. A glucagon stimulation test demonstrated relative insulin deficiency, indicative of latent autoimmune diabetes in adults (LADA). Tolerable muscle rigidity was achieved when the dosage of basal insulin was split into two separate daily injections with lower amounts of units per injection. This case highlights a different form of autoimmune diabetes in SPS. To our knowledge, this is the first report of SPS described shortly after the initiation of insulin therapy that required basal insulin to achieve tolerable muscle symptoms and better glucose control, without the development of diabetic ketoacidosis.
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3.
Pediatric stroke as the presenting symptom of new-onset type 1 diabetes mellitus without DKA: case report and literature review.
Bharill, SA, Hunter, JD, Walsh, ET, Crudo, DF, Constantacos, C
Journal of pediatric endocrinology & metabolism : JPEM. 2019;(9):1035-1037
Abstract
Background Stroke and other neurologic complications are rare in pediatric type 1 diabetes mellitus (T1DM) without severe diabetic ketoacidosis (DKA) or poor glycemic control. Case presentation A previously healthy, 10-year-old female presented with acute thalamic stroke, non-acidotic new T1DM diagnosis and negative hypercoagulopathy workup. She received routine insulin therapy and aspirin, and returned to neurologic baseline within a year without stroke recurrence. Conclusions The contribution of non-acidotic hyperglycemia to stroke risk is better described in adults. Even though unable to prove causality, this case should at least raise awareness of the possible association of pediatric new-onset diabetes and stroke for optimal outcomes.
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4.
Closed-loop insulin delivery in end-of-life care: a case report.
Boughton, CK, Bally, L, Hartnell, S, Wilinska, M, Coll, AP, Evans, M, Stettler, C, Hovorka, R
Diabetic medicine : a journal of the British Diabetic Association. 2019;(12):1711-1714
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Abstract
BACKGROUND Glucose management for people with diabetes approaching the end of life can be very challenging. The aim is to balance a minimally invasive approach with avoidance of symptomatic hypo- and hyperglycaemia. CASE REPORT We present a case of a hospitalized individual whose glucose was managed with closed-loop insulin delivery within a randomized controlled trial setting during a period of terminal illness. During the time in which closed-loop insulin delivery was used, glucose control was safe, with no glucose-related harm. The mean ± sd sensor glucose for this individual was 11.3 ± 4.3 mmol/l, percentage of time spent in target glucose range between 6 and 15 mmol/l was 70.5%, time spent in hypoglycaemia was 2.0% and time spent in significant hyperglycaemia >20 mmol/l was 2.6%. CONCLUSION Closed-loop systems can accommodate personalized glucose targets and highly variable insulin requirements. Factory-calibrated continuous glucose sensors and insulin pump therapy are less intrusive than finger-stick glucose measurements and insulin injections, respectively. Closed-loop systems may provide a safer and less burdensome approach to glucose management towards the end of life.
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5.
Heterotaxy syndrome with agenesis of dorsal pancreas and diabetes mellitus: case report and review of the literature.
Riguetto, CM, Pelichek, S, Moura Neto, A
Archives of endocrinology and metabolism. 2019;(4):445-448
Abstract
Heterotaxy syndrome (HS) is a rare congenital condition with multifactorial heritance, characterized by an abnormal arrangement of thoraco-abdominal organs and vessels. Patients present with multiple cardiac, gastrointestinal, hepatosplenic, pancreatic, renal, neurological and skeletal disorders without any pathognomonic alteration. Despite the described increased risk of diabetes mellitus (DM) in patients with altered pancreatic anatomy, just one case was reported in Korea regarding the association of HS and DM in a 13-year-old girl. Our report refers to a 40-year-old female Brazilian patient with a history of DM and HS with polysplenia and agenesis of dorsal pancreas without cardiac abnormalities. She presented a worsening glycemic control associated with weight gain and signs of insulin resistance. After a proper clinical management of insulin and oral medications, our patient developed an improvement in glycemic control. Although it is a rare disease, HS with polysplenia and pancreatic disorders can be associated with an increased risk of DM. This case highlights the importance of investigating DM in patients with HS, especially those with pancreatic anatomical disorders, for proper clinical management of this rare condition.
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Rapid onset type-1 diabetes and diabetic ketoacidosis secondary to nivolumab immunotherapy: a review of existing literature.
Abdullah, HMA, Elnair, R, Khan, UI, Omar, M, Morey-Vargas, OL
BMJ case reports. 2019;(8)
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Abstract
Nivolumab is a programmed cell death receptor (PD-1) inhibitor that is increasingly used for various malignancies, both as a first line agent and as salvage therapy. Being a PD-1/PD-1 ligand checkpoint inhibitor, it is known to cause autoimmune inflammation of various organs and has been associated with thyroiditis, insulitis, colitis, hepatitis and encephalitis to name a few. There are increasing reports of nivolumab leading to acute onset fulminant type 1 diabetes and diabetic ketoacidosis (DKA). We present a case of a 68-year-old man who developed DKA after 2 doses of nivolumab for metastatic melanoma. He was found to have type 1 diabetes, but no diabetes related antibodies were positive. He recovered from diabetes and continues to use insulin 1 year after his diagnosis. This case and associated review illustrates the importance of educating and monitoring patients who start nivolumab therapy regarding this potentially life threatening complication.
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Sulfonylurea vs insulin therapy in individuals with sulfonylurea-sensitive permanent neonatal diabetes mellitus, attributable to a KCNJ11 mutation, and poor glycaemic control.
Stanik, J, Dankovcikova, A, Barak, L, Skopkova, M, Palko, M, Divinec, J, Klimes, I, Gasperikova, D
Diabetic medicine : a journal of the British Diabetic Association. 2018;(3):386-391
Abstract
BACKGROUND Therapy with sulfonylurea is preferable to insulin in the majority of individuals with KCNJ11 mutations, but not all of these people achieve target levels of HbA1c in long-term follow-up. We aimed to compare sulfonylurea therapy with insulin treatment in two sulfonylurea-sensitive individuals with a KCNJ11 mutation who had poorly controlled permanent neonatal diabetes mellitus. CASE REPORT We report on two individuals with a KCNJ11 mutation (p.R201H) who are currently aged 35 (SVK1) and 21 years (SVK2). These individuals were switched from insulin to sulfonylurea in 2005. Data from the first 4 (SVK2) and 8 years (SVK1) of the follow-up showed improved diabetes control and increased quality of life for both individuals. During the following years, however, both individuals failed to retain good diabetes control (HbA1c ≤ 53 mmol/mol; 7.0%). We therefore changed the therapy to a combination of insulin and sulfonylurea in both individuals, or to insulin monotherapy in SVK1, and compared the effects on HbA1c with those of sulfonylurea monotherapy. HbA1c levels in both individuals worsened after adding insulin to sulfonylurea [67 mmol/mol (8.3%) vs 77 mmol/mol (9.2%) in SVK1 and 106 mmol/mol (11.8%) vs 110±19 mmol/mol (12.2±1.7%) in SVK2], and after switching to only insulin therapy in SVK1 [57 mmol/mol (7.4%) vs 62 mmol/mol (7.8%)] when compared with sulfonylurea monotherapy. DISCUSSION Our data show that sulfonylurea monotherapy might be preferable to insulin in people with permanent neonatal diabetes mellitus sensitive to sulfonylurea even when HbA1c is above target.
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Insulin autoimmune syndrome (Hirata's disease) in an Italian patient: a case report and review of the literature.
Censi, S, Albergoni, MP, Gallo, N, Plebani, M, Boscaro, M, Betterle, C
Clinical chemistry and laboratory medicine. 2018;(6):889-895
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Abstract
We describe the case of a 54-year-old Caucasian Italian male experiencing episodes of hypoglycemia, occurring mainly after meals. He had never been exposed to insulin and was taking ramipril, flecainide and acetylsalicylic acid. An oral glucose tolerance test (OGTT) showed high blood glucose levels diagnostic for diabetes mellitus at 120 min and hypoglycemia with inappropriately high insulin levels at 240 min. The 72-h fasting test, abdominal computed tomography (CT) and positron emission tomography-CT were normal. Insulin autoantibodies were positive at high titers, prompting a diagnosis of insulin autoimmune syndrome (IAS). The patient was advised to take frequent, small meals and thus achieved a good control of his hypoglycemic symptoms. After 18 months of this dietary management, his insulin autoantibody levels decreased considerably but remained detectable. During an OGTT, his blood glucose levels at 120 min were now indicative of an impaired glucose tolerance rather than diabetes, and there was improvement in the glucose nadir. The patient had no other clinical or latent autoimmune diseases. Here we discuss the main features of IAS (also known as Hirata's disease) and review the cases of IAS reported in Italy to date.
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Medical Nutrition Therapy Is Effective in the Management of Hypoglycemia Caused by Insulin Antibodies: A Case Report and Literature Review.
Li, R, Mao, J, Yu, K, Wang, L, Hu, M, Xu, L
Journal of the American College of Nutrition. 2016;(1):86-90
Abstract
Autoimmune antibodies, induced by exogenous insulin preparations, may result in labile glucose control and frequent hypoglycemia in some rare cases. In addition to insulin cessation, immune suppressants and/or plasmapheresis have been used as the primary remedies for these patients. Some previous studies also indicate that the condition tends to remit spontaneously after discontinuation of insulin exposure. Because of this, the clinical importance of nutritional interventions and behavioral approaches, which may play a role in ameliorating the symptoms, should also be emphasized. Herein, we report on a 64-year-old man with hypoglycemia induced by insulin antibodies (IAs), whose hypoglycemic symptoms significantly improved after the implementation of nutrition therapy. This rare case expands our knowledge of the management of hypoglycemia, and for the first time highlights the significance of nutritional and lifestyle intervention in treatment of IA-induced hypoglycemia.
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Fulminant Type 1 Diabetes Mellitus Associated with Coxsackie Virus Type A2 Infection: A Case Report and Literature Review.
Ohara, N, Kaneko, M, Nishibori, T, Sato, K, Furukawa, T, Koike, T, Sone, H, Kaneko, K, Kamoi, K
Internal medicine (Tokyo, Japan). 2016;(6):643-6
Abstract
A 65-year-old Japanese man presented to our hospital in June 2013 with a 6-day history of fever and fatigue, a 24-h history of thirst, and polyuria. His temperature was 37.8°C and he was alert. However, laboratory tests revealed severe hyperglycemia, undetectable C-peptide levels, and diabetic ketoacidosis. Serum antibody testing confirmed a Coxsackie virus A2 infection. A variety of viral infections are reported to be involved in the development of fulminant type 1 diabetes mellitus (FT1D). Our patient is the first reported case of FT1D associated with Coxsackie virus A2 infection and supports the etiological role of common viral infections in FT1D.