Humans ; Diabetes Mellitus, Type 1 ; Hypoglycemia/chemically induced* ; Hypoglycemic Agents/adverse effects* ; China ; Blood Glucose ; Insulin

Hypoglycemic disorders are rare in persons without diabetes, and clinical evaluation to identify its etiology can be challenging. We present a case of insulin autoimmune syndrome induced by carbimazole in a middle-aged Chinese man with underlying Graves’ disease, which was managed conservatively with a combination of dietary modification and alpha-glucosidase inhibitor.
Hypoglycemia ; Hyperinsulinism ; Insulin Antibodies

A four-year-old female who was born term via spontaneous vaginal delivery with a birth weight of 3.4 kg had an onset of persistent hypoglycaemia at the 6th hour of life. She was diagnosed with congenital hyperinsulinism based on high glucose load, negative ketone and a good response to glucagon. Genetic workup revealed the presence of ATP Binding Cassette Subfamily C Member 8 (ABCC8 genes) mutation which indicated a focal form of congenital hyperinsulinism. She was resistant to the standard dose of oral diazoxide but responded to subcutaneous somatostatin. At the age of 3 years and 6 months, multiple daily injections of somatostatin were replaced with a long-acting monthly somatostatin analogue. With the present treatment, she had better glycaemic control, normal growth and was able to stop tube feeding.
Congenital Hyperinsulinism ; Somatostatin

Objective: The objective of the study was to determine the effectiveness of myoinositol (MI) supplementation in the prevention of gestational diabetes mellitus (GDM) among high-risk patients. Materials and Methods: Comprehensive and systemic online searches were performed on PubMed, MEDLINE, Ovid, and Cochrane. Cross-referencing from related articles was also done. Only studies published in English were included in the study. We selected all randomized controlled trials on MI and singleton pregnant women with high risk for GDM. Data Collection and Analysis: Five randomized controlled trials were evaluated by two independent reviewers. For each comparison, the quality of evidence was assessed using the Preferred Reporting Items for Systematic Reviews and Meta-Analyses Cochrane Collaboration tool. Review Manager 5.3 was used to generate the risk of bias evaluation and the analysis of the results. Main Results: The present study identified five randomized controlled trials involving 871 participants. The comparison of the studies showed a statistically significant reduction in the incidence of GDM in MI supplementation versus the control group (odds ratio [OR] = 0.32, 95% confidence interval [CI] = 0.19–0.53, P = 0.0001, Z = 4.36) by 68%. Similarly, there is a greater reduction in the incidence of fetal macrosomia among patients in the MI group than the controlled group (OR = 0.24, 95% CI = 0.07–0.78; P = 0.02, Z = 2.36) by 78%. However, there was no difference in terms of incidence of gestational hypertension (OR = 0.61, 95% CI = 0.19–2.01; P = 0.42, Z = −0.81), cesarean section (OR = 0.89, 95% CI = 0.65–1.22; P = 0.47, Z = 0.72), and neonatal hypoglycemia (OR = 0.35, 95% CI = 0.01–8.80; P = 0.53, Z = 0.63) outcomes. Conclusion MI supplementation taken at 4 g daily would decrease the incidence of GDM and fetal macrosomia. There was no statistically significant reduction in the risk of gestational hypertension, cesarean section, and neonatal hypoglycemia in the supplementation of MI.
Cesarean section ; fetal macrosomia ; gestational diabetes mellitus ; gestational hypertension ; myoinositol ; neonatal hypoglycemia

Objective: To analyze the short-time efficacy of empagliflozin in the treatment of glycogen storage disease type Ⅰb (GSD Ⅰb). Methods: In this prospective open-label single-arm study, the data of 4 patients were collected from the pediatric department in Peking Union Medical College Hospital from December 2020 to December 2022. All of them were diagnosed by gene sequencing and had neutropenia. These patients received empagliflozin treatment. Their clinical symptoms such as height and weight increase, abdominal pain, diarrhea, oral ulcer, infection times, and drug applications were recorded at 2 weeks, 1 month, 2 months, 3 months, 6 months, 9 months, 12 months, and 15 months after treatment to assess the therapeutic effect. The liquid chromatography-tandem mass spectrometry method was used to monitor the changes in 1, 5-anhydroglucitol (1, 5AG) concentration in plasma. At the same time, adverse reactions such as hypoglycemia and urinary tract infection were closely followed up and monitored. Results: The 4 patients with GSD Ⅰb were 15, 14, 4 and 14 years old, respectively at the beginning of empagliflozin treatment, and were followed up for 15, 15, 12 and 6 months, respectively. Maintenance dose range of empagliflozin was 0.24-0.39 mg/(kg·d). The frequency of diarrhea and abdominal pain decreased in cases 2, 3, and 4 at 1, 2 and 3 months of treatment, respectively. Their height and weight increased at different degrees.The absolute count of neutrophils increased from 0.84×10⁹, 0.50×10⁹, 0.48×10⁹, 0.48×10⁹/L to 1.48×10⁹, 3.04×10⁹, 1.10×10⁹, 0.73×10⁹/L, respectively. Granulocyte colony-stimulating factor was gradually reduced in 1 patients and stopped in 3 patient. Plasma 1, 5 AG levels in 2 children were significantly decreased after administration of empagliflozin (from 46.3 mg/L to 9.6 mg/L in case 2, and from 56.1 mg/L to 15.0 mg/L in case 3). All 4 patients had no adverse reactions such as hypoglycemia, abnormal liver or kidney function, or urinary system infection. Conclusion: In short-term observation, empagliflozin can improve the symptoms of GSD Ⅰb oral ulcers, abdominal pain, diarrhea, and recurrent infection, also can alleviate neutropenia and decrease 1, 5AG concentration in plasma, with favorable safety.
Humans ; Child ; Child, Preschool ; Adolescent ; Prospective Studies ; Glycogen Storage Disease Type I/drug therapy* ; Neutropenia ; Abdominal Pain ; Diarrhea/drug therapy* ; Hypoglycemia

Objective: To systematically evaluate the correlation between prenatal steroid exposure and hypoglycemia in late preterm neonates. Methods: Eight databases in either Chinese or English, including PubMed, the Cochrane Library, Embase, Medline, Scopus, CNKI, Wanfang and VIP, were searched to extract the studies on the correlation between prenatal steroid exposure and hypoglycemia in late preterm neonates published from the establishment of each database to December 2022. The Meta-analysis was performed using Stata 14.0 statistical software. Results: A total of 9 studies were included in this Meta-analysis, including 6 retrospective cohort studies, 2 prospective cohort studies and 1 randomized controlled trial (RCT) study, involving 9 143 premature infants. The Meta-analysis showed that prenatal steroid exposure increased the risk of late preterm neonatal hypoglycemia (RR=1.55, 95%CI 1.25-1.91, P<0.001). The similar correlation between prenatal steroid exposure and hypoglycemia in late preterm neonates was all found in the following subgroups: North America (RR=1.57, 95%CI 1.37-1.80, P<0.001), enrolling pregnant women with gestational diabetes (RR=1.62, 95%CI 1.26-2.08, P<0.001), A-grade literature quality (RR=1.43, 95%CI 1.14-1.79, P=0.002), criteria for hypoglycemia ≤40 mg/dl (1 mg/dl=0.056 mmol/L, RR=1.49, 95%CI 1.28-1.73, P<0.001), sample size of 501-1 500 (RR=1.69, 95%CI 1.19-2.40, P=0.003) and >1 500 (RR=1.65, 95%CI 1.48-1.83, P<0.001), steroid injection dosage and frequency of 12 mg 2 times (RR=1.66, 95%CI 1.50-1.84, P<0.001), the time interval from antenatal corticosteroid administration to delivery of 24-47 h (RR=1.98, 95%CI 1.26-3.10, P=0.003), unadjusted gestational age (RR=1.78, 95%CI 1.02-3.10,P=0.043) and unadjusted birth weight (RR=1.80, 95%CI 1.22-2.66, P=0.003). Meta-regression results showed that steroid injection frequency and dose were the main sources of high heterogeneity among studies (P=0.030). Conclusion: Prenatal steroid exposure may be a risk factor for hypoglycemia in late preterm neonates.
Female ; Humans ; Infant ; Infant, Newborn ; Pregnancy ; Birth Weight ; Hypoglycemia/chemically induced* ; Infant, Premature ; Randomized Controlled Trials as Topic ; Steroids/adverse effects* ; Prenatal Exposure Delayed Effects

Humans ; Consensus ; Congenital Hyperinsulinism

Dumping syndrome after bariatric surgery is common. However, it is rarely seen during pregnancy because patients are usually advised to avoid pregnancy immediately after surgery. This case highlights the importance of avoidance of pregnancy after bariatric surgery. We report a case of unplanned pregnancy in a 35-year-old woman with a history of subfertility for 8 years who conceived spontaneously 3 months after gastric bypass surgery. This occurred because there was no contraception offered to her after the procedure. The pregnancy was complicated with recurrent episodes of hypoglycaemia secondary to dumping syndrome. Primary care providers need to be vigilant and have a high index of suspicion for dumping syndrome in pregnant obese women who have undergone bariatric surgery.
Dumping Syndrome ; Hypoglycemia

Objective: To explore the clinical features of hepatocerebral mitochondrial DNA depletion syndrome (MDS). Methods: The clinical data of 6 hepatocerebral MDS patients diagnosed in the Jinshan Hospital of Fudan University from January 2012 to December 2019 were retrospectively collected and analyzed. Related literature published before January 2020 were searched with the key words of "DGUOK""MPV17""POLG""C10orf2" in PubMed, China national knowledge infrastructure (CNKI) and Wanfang database. Results: All the 6 hepatocerebral MDS cases were male. The age of onset ranged from 3 days to 8 months. The most common initial symptoms were cholestasis and developmental retrogression. The main clinical manifestations included hepatomegaly (4 cases), hypotonia (3 cases), growth retardation (4 cases), cholestasis (5 cases), coagulopathy (5 cases), hypoalbuminemia (3 cases), hypoglycemia (4 cases), hyperlactacidemia (5 cases), and abnormal blood metabolism screening (6 cases). The isotope hepatobiliary imaging revealed no gallbladder and intestinal tract development within 24 hours in 2 patients. Regarding the cranial imaging examination, the head CT found widening of the extracranial space in 1 case, the brain magnetic resonance imaging (MRI) found ventricular enlargement in 2 cases, and the brain ultrasound found peripheral white matter injury in 1 case. Two cases were lost to follow-up, one died of liver failure, and three died of multiple organ failure due to aggravated infection. Among the 6 cases, there were 3 with MPV17 variation (c.182T>C and c.279G>C were novel), 1 with POLG variation (c.2993G>A was novel), 1 with DGUOK variation (c.679G>A homozygous mutation, parthenogenetic diploid of chromosome 2) and 1 with C10orf2 variation (c.1186C>T and c.1504C>T were novel). The literature review found that 129, 100, 51 and 12 cases of hepatocerebral MDS were caused by DGUOK, MPV17, POLG and C10orf2 gene variations, respectively. And the most common clinical manifestations were liver dysfunction presented with cholestasis and elevated transaminase, metabolic disorders including hypoglycemia and hyperlactacidemia, and diverse neurologic symptoms including developmental retardation, hypotonia, epilepsy and peripheral neuropathy. Besides, 1/3 of the patients with C10orf2 variation developed renal tubular injury. Conclusions: Hepatocerebral MDS mainly present with liver dysfunction, metabolic disorder and neuromuscular impairment. Different genotypes show specific clinical manifestations.
Cholestasis ; DNA, Mitochondrial/genetics* ; Female ; Humans ; Hypoglycemia/genetics* ; Infant ; Liver Diseases/genetics* ; Male ; Mitochondrial Diseases ; Muscle Hypotonia ; Retrospective Studies

Introduction: Hypoglycemia is an important and harmful complication that often occurs in inpatient and outpatient settings. This study aims to assess the incidence of inpatient hypoglycemia and its related factors. We also assessed mortality and length of hospital stay. Methodology: We performed a retrospective cohort study among patients with type 2 diabetes mellitus admitted to a tertiary hospital in Indonesia. Using multivariate regression, we analyzed age, sex, body mass index, comorbidities, history of hypoglycemia, hyperglycemia treatment administered, nutritional intake, and medical instruction as the related risk factors for inpatient hypoglycemia. Results: From 475 subjects, 80 (16.8%) had inpatient hypoglycemia, of which, 7.4% experienced severe hypoglycemia. We found that patients with a history of hypoglycemia (RR: 4.6; 95% CI: 2.8-7.6), insulin and/or sulfonylurea treatment (RR 6.4; 95% CI: 1.6-26.5), and inadequate nutritional intake (RR 2.6; 95% CI: 1.5-4.3) were more likely to have hypoglycemic events compared to those who did not. The length of hospital stay for patients in the hypoglycemic group is significantly longer than those in the non-hypoglycemic group (13 vs 7 days, p<0.001), but their mortality rates did not differ (16% vs 10.9%, p=0.18). Conclusion Inpatient hypoglycemia may be affected by a history of hypoglycemia and inadequate nutritional intake. Patients who had inpatient hypoglycemia tend to have a longer median length of hospital stay.
Hypoglycemia ; Diabetes Mellitus ; Insulin ; Mortality ; Length of Stay