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This study aimed to examine the clinical characteristics of young children diagnosed with maturity-onset diabetes (MODY) using urine glucose screening at schools. The study participants were 70 non-obese children who were clinically diagnosed with type 2 diabetes through urine glucose screening at schools in Tokyo between 1974 and 2020. Of these children, 55 underwent genetic testing, and 21 were finally diagnosed with MODY: MODY2 in eight, MODY3 in eight, MODY1 in four and MODY5 in one. A family history of diabetes was found in 76.2% of the patients. Fasting plasma glucose levels did not differ between the different MODY subtypes, while patients with MODY 3, 1, and 5 had significantly higher levels of glycosylated hemoglobin and 2-hour glucose in an oral glucose tolerance test than those with MODY2. In contrast, most patients exhibit mild insulin resistance and sustained ß-cell function. In the initial treatment, all patients with MODY2 were well controlled with diet and exercise, whereas the majority of those with MODY3, 1, and 5 required pharmacological treatment within one month of diagnosis. In conclusion, urine glucose screening in schools appears to be one of the best opportunities for early detection of the disease and providing appropriate treatment to patients.
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Islet-cell associated antibodies are predictive and diagnostic markers for type 1 diabetes. We studied the differences in the early clinical course of children with type 1 diabetes with a single antibody and those with multiple antibodies against pancreatic ß-cells. Sixty-seven children with type 1 diabetes aged less than 15 years diagnosed between 2010 and 2021 were included in the study and subdivided into two subgroups: children who were single positive for either glutamic acid decarboxylase (GAD) antibodies (n = 16) or insulinoma-associated antigen-2 (IA-2) antibodies (n = 13) and those positive for both antibodies (n = 38) at diagnosis. We compared the patients' clinical characteristics, pancreatic ß-cell function, and glycemic control during the 5 years after diagnosis. All clinical characteristics at diagnosis were similar between the two groups. One and two years after diagnosis, children who tested positive for both antibodies showed significantly lower postprandial serum C-peptide (CPR) levels than those who tested positive for either GAD or IA-2 antibodies (p < 0.05). In other periods, there was no significant difference in CPR levels between the two groups. There was a significant improvement in glycosylated hemoglobin (HbA1c) levels after starting insulin treatment in both groups (p < 0.05), but no significant difference in HbA1c levels between the groups. Residual endogenous insulin secretion may be predicted based on the number of positive islet-cell associated antibodies at diagnosis. Although there are differences in serum CPR levels, optimal glycemic control can be achieved by individualized appropriate insulin treatment, even in children with type 1 diabetes.
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Autoanticuerpos , Diabetes Mellitus Tipo 1 , Glutamato Descarboxilasa , Insulina , Insulinoma , Humanos , Diabetes Mellitus Tipo 1/tratamiento farmacológico , Masculino , Femenino , Niño , Adolescente , Hemoglobina Glucada , Insulinoma/tratamiento farmacológico , Péptido C/sangre , Insulina/uso terapéuticoRESUMEN
There are no study reports to clarify the association between gestational age (GA) or anthropometric values at birth, and plasma cortisol levels in the blood of preterm infants at birth and at one month of age. This hospital-based retrospective cohort study included infants born at <37 weeks' gestation between 2019 and 2021. First, the association between plasma cortisol level and GA or anthropometric values at birth (birth weight standard deviation score [SDS], birth length SDS, and birth head circumference SDS) was identified by regression and multiple regression analyses. Second, plasma cortisol levels in the umbilical cord at birth and at one month of age were compared between small-for-gestational age (SGA) and non-SGA infants. Sixty-one preterm infants were enrolled (SGA: 24 and non-SGA: 37). Plasma cortisol levels at birth were significantly associated with GA. Plasma cortisol levels at one month of age were associated with GA and birth head circumference SDS. Plasma cortisol levels at birth were significantly higher in SGA than non-SGA (p = 0.010). GA was an independent determinant of plasma cortisol levels at birth. SGA infants had a high plasma cortisol level at birth; resulting in speculation that a high plasma cortisol level at birth may predict abnormal neurological outcomes.
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Enfermedades del Recién Nacido , Recien Nacido Prematuro , Peso al Nacer , Femenino , Retardo del Crecimiento Fetal , Edad Gestacional , Humanos , Hidrocortisona , Lactante , Recién Nacido , Recién Nacido Pequeño para la Edad Gestacional , Estudios RetrospectivosRESUMEN
AIMS/INTRODUCTION: The aim of the study was to compare two continuous glucose monitoring (CGM) systems, intermittently scanned CGM (isCGM) and real-time CGM (rtCGM), to determine which system achieved better glycemic control in pediatric patients. MATERIALS AND METHODS: We carried out a retrospective cohort study of children and adolescents with type 1 diabetes, and compared the time in range (70-180 mg/dL), time below range (<70 mg/dL) and time above range (>180 mg/dL), and estimated glycated hemoglobin levels between patients on isCGM and rtCGM. RESULTS: Of the 112 participants, 76 (67.9%) used isCGM and 36 (32.1%) used rtCGM for glycemic management. Patients on rtCGM had significantly greater time in range (57.7 ± 12.3% vs 52.3 ± 12.3%, P = 0.0368), and had significantly lower time below range (4.3 ± 2.7% vs 10.2% ± 5.4%, P < 0.001) than those on isCGM, but there was no significant difference in the time above range (37.4 ± 12.9% vs 38.0% ± 12.5%, P = 0.881) or the glycosylated hemoglobin A1c levels (7.4 ± 0.9% vs 7.5 ± 0.8%, P = 0.734) between the two groups. CONCLUSIONS: Pediatric patients with type 1 diabetes on rtCGM also showed more beneficial effects for increase of time in range, with a notable reduction of time below range compared with those on isCGM. Real-time CGM might provide better glycemic control than isCGM in children with type 1 diabetes.
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Diabetes Mellitus Tipo 1 , Adolescente , Glucemia , Automonitorización de la Glucosa Sanguínea , Niño , Diabetes Mellitus Tipo 1/tratamiento farmacológico , Hemoglobina Glucada/análisis , Humanos , Hipoglucemiantes/uso terapéutico , Estudios RetrospectivosRESUMEN
We aimed to create percentile-based reference values of the umbilical cord blood insulin-like growth factor-1 (IGF-1) levels in Japanese newborns, as these values have not yet been established. A total of 259 newborns were classified into four gestational-age-at-birth (GA) groups: extremely preterm (<28 weeks); early preterm (28−33 weeks); late preterm (34−36 weeks); and term (≥37 weeks). They were further subclassified as small-for-gestational-age (SGA) or non-SGA. The 10th, 25th, 50th, 75th, and 90th percentiles of the umbilical cord blood IGF-1 levels were calculated and compared between the groups by using reference values of 9, 18, 33, 52, and 71 ng/mL, respectively. In the extremely preterm group, the IGF-1 levels were significantly lower than those in the early preterm, late preterm, and term groups (13.5, 24.0, 44.5, and 47.5 ng/mL, respectively; p < 0.001). The umbilical cord blood IGF-1 levels in the SGA newborns were significantly lower than those in the non-SGA newborns in all subgroups. In multivariate analyses, the GA and birth weight standard deviation scores were independent determinant factors for the umbilical cord blood IGF-1 levels. Thus, we established percentile-based reference values of umbilical cord blood IGF-1 in Japanese newborns; these reference values can be applied on the basis of the extent of prematurity and the SGA status.
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Beckwith-Wiedemann syndrome (BWS) is infrequently associated with adrenocortical carcinoma (ACC) or non-hormone-producing adrenal cytomegaly, but we recently, encountered a single case of adrenal cytomegaly in a patient with BWS, which was difficult to distinguish from androgen-producing adrenocortical carcinoma (ACC). Here, we describe the case of a 4-month-old female who presented with clitoromegaly, hemihypertrophy, and an adrenal mass identified during the prenatal period. The mass was located in detected at the left suprarenal region and detected at 20 weeks of gestational age. At birth, she also presented with clitoromegaly and elevated serum levels of 17α-hydroxyprogesterone, dehydroepiandrosterone, and testosterone at birth and experienced hyper-insulinemic hypoglycemia, which improved following diazoxide therapy. We initially suspected androgen-producing ACC with metastasis and the left adrenal mass was resected accordingly when the patient reached 4 months of age. However, histological examination revealed adrenal cytomegaly. Genetic analysis revealed paternal uniparental disomy, and the patient was finally diagnosed as having BWS. Resection of the left adrenal gland restored the serum androgen levels to normal physiological levels without any recurrence. While it is reasonably well known that BWS is sometimes accompanied by virilization due to androgen-producing ACC, our findings are among the first to suggest that adrenal cytomegaly can also increase androgen hormone production. Thus, we propose that adrenal cytomegaly should be considered one of the differential diagnoses when accompanied with hyperandrogenism in BWS patients.
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Neoplasias de la Corteza Suprarrenal , Enfermedades de las Glándulas Suprarrenales , Carcinoma Corticosuprarrenal , Síndrome de Beckwith-Wiedemann , Andrógenos , Femenino , Humanos , Lactante , Recién Nacido , Embarazo , Disomía UniparentalRESUMEN
AIMS/INTRODUCTION: We examined the impact of scanning frequency with flash glucose monitoring on glycemic control in children and adolescents with type 1 diabetes. MATERIALS AND METHODS: The study included 85 patients, aged 14.0 ± 0.5 years, with type 1 diabetes. The median time in the target glucose range (TIR) and glycosylated hemoglobin (HbA1c) values were 50.0 ± 1.4% and 7.5 ± 0.1%, respectively. RESULTS: The median scanning frequency using flash glucose monitoring was 12.0 ± 0.4 times/day. Scanning frequency showed a significant positive correlation with TIR and an inverse correlation with HbA1c. Scanning frequency was identified to be the determinant of TIR and HbA1c by using multivariate analysis. The participants whose scanning frequency was <12 times/day were categorized as the low-frequency group (n = 40), and those who carried out the scanning >12 times/day were categorized as the high-frequency group (n = 45). Patients in the high-frequency group were more likely to be treated with insulin pumps compared with those in the low-frequency group; however, this difference was not significant (21.3 vs 5.3%, P = 0.073). The high-frequency group showed significantly greater TIR than the low-frequency group (57 ± 1.6 vs 42 ± 1.7%, P = 0.002). Furthermore, the high-frequency group showed significantly lower HbA1c levels than the low-frequency group (6.8 ± 0.1 vs 8.0 ± 0.1%, P < 0.001). CONCLUSIONS: These findings showed that patients with a higher scanning frequency had better glycemic control, with greater TIRs and lower HbA1c levels, compared with those with a lower scanning frequency. Scanning frequency of >12 times/day might contribute to better glycemic outcomes in real-world practice in children with type 1 diabetes.
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Automonitorización de la Glucosa Sanguínea/métodos , Diabetes Mellitus Tipo 1/sangre , Control Glucémico/estadística & datos numéricos , Factores de Tiempo , Adolescente , Glucemia/análisis , Niño , Preescolar , Diabetes Mellitus Tipo 1/tratamiento farmacológico , Femenino , Hemoglobina Glucada/análisis , Control Glucémico/métodos , Humanos , Hipoglucemiantes/administración & dosificación , Insulina/administración & dosificación , Sistemas de Infusión de Insulina , Masculino , Análisis MultivarianteRESUMEN
BACKGROUND: We assessed the association between scanning frequency of flash glucose monitoring (FGM) and continuous glucose monitoring (CGM)-derived glycemic markers in children and adolescents with type 1 diabetes. METHODS: Subjects consisted of 85 children and adolescents with type 1 diabetes using FGM. We assessed the association between scanning frequencies of FGM- and CGM-derived metrics: Time in range (TIR) (70-180 mg/dL), time below range (TBR) (<70 mg/dL), time above range (>180 mg/dL), and other glycemic markers - laboratory-measured HbA1c and CGM-estimated glucose and HbA1c (eA1c) levels in the subjects. RESULTS: The mean number of scans was 11.5 ± 3.5 (5-20) times per day, and scanning was most frequently conducted during a period of 18-24 h. Scanning frequency showed significant positive correlation with TIR (r = 0.719, P < 0.0001) and inverse correlation with time above range (r = -0.743, P < 0.0001), but did not correlate with TBR. There were also significant inverse correlations between scanning frequency and glucose, HbA1c, and eA1c levels (r = -0.765, -0.815, and -0.793, respectively, P < 0.0001). CONCLUSIONS: Frequent glucose testing with FGM decreased hyperglycemia with increased TIR, but did not reduce TBR. Coping with a rapid fall of glucose and unexpected hypoglycemia with more advanced technology might contribute to a reduction in TBR.
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Diabetes Mellitus Tipo 1 , Hipoglucemia , Adolescente , Glucemia , Automonitorización de la Glucosa Sanguínea , Niño , Glucosa , HumanosRESUMEN
INTRODUCTION: We evaluated the frequencies of various glycemic markers derived from continuous glucose monitoring in Japanese children and adolescents with type 1 diabetes and assessed the significance of hypoglycemia duration. METHODS: We enrolled 85 children and adolescents (36 boys and 49 girls) with type 1 diabetes who used FreeStyle® Libre in the present study. Frequencies of blood glucose levels as time within target range (TIR; 70-180 mg/dL), time below target range (TBR; <70 mg/dL), time below extreme hypoglycemia range (TBER; <54 mg/dL), and time above range (TAR; >180 mg/dL) were assessed during a 3-month study period. Furthermore, we evaluated the intraday frequencies of TBR and TBER. RESULTS: The mean frequencies of TIR, TBR, and TAR were 52.7 ± 11.3%, 10.8 ± 5.4%, and 36.5 ± 10.8%, respectively, whereas the mean frequency of TBER was 1.1 ± 0.9% (0-3.0%); there was no clinical episode of severe hypoglycemia. The mean frequency of TBR was significantly greater in 0-6 h (16.9 ± 5.2%) than in 6-12 h (7.8 ± 2.9%) and 18-24 h (6.8 ± 4.8%; p < 0.01) time zones, respectively. DISCUSSION/CONCLUSION: We found similar TIR and comparatively higher TBR frequencies, particularly during sleep, than those that were previously reported. Possible reasons for the higher frequency of TBR include differences in the quality of insulin treatment and diabetes care between the present study and the European studies. The utilization of advanced technologies, such as a predictive low-glucose suspend-function pump or closed-loop therapy, can reduce the frequency of TBR, with a consequent increase in TIR frequency and comprehensive improvement in glycemic control.
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Glucemia/metabolismo , Diabetes Mellitus Tipo 1 , Hipoglucemia , Insulina , Adolescente , Niño , Preescolar , Diabetes Mellitus Tipo 1/sangre , Diabetes Mellitus Tipo 1/tratamiento farmacológico , Femenino , Humanos , Hipoglucemia/sangre , Hipoglucemia/tratamiento farmacológico , Insulina/administración & dosificación , Insulina/farmacocinética , Japón , MasculinoAsunto(s)
Intolerancia a la Glucosa/epidemiología , Trastornos del Crecimiento/tratamiento farmacológico , Hormona de Crecimiento Humana/efectos adversos , Recién Nacido Pequeño para la Edad Gestacional/crecimiento & desarrollo , Glucemia/análisis , Niño , Preescolar , Femenino , Intolerancia a la Glucosa/etiología , Prueba de Tolerancia a la Glucosa , Hemoglobina Glucada/análisis , Hormona de Crecimiento Humana/uso terapéutico , Humanos , Insulina/sangre , MasculinoRESUMEN
We assessed the significance of recommendations from the international consensus on continuous glucose monitoring (CGM)-derived metrics in Japanese children and adolescents with type 1 diabetes. Eighty-five patients (age, 13.5 ± 4.7 years) who wore the FreeStyle® Libre for a 28-day period were enrolled in this study. Seventy-three patients were treated with multiple daily injections of insulin and 12 with insulin pump therapy without using a sensor-augmented pump or a predictive low-glucose suspend-function pump. We evaluated the relationship between CGM-derived metrics: time in range (TIR: 70-180 mg/dL), time below range (TBR: <70 mg/dL), and time above range (TAR: >180 mg/dL), and laboratory-measured HbA1c and estimated HbA1c (eA1c) levels calculated from the mean glucose values. The TIR was 50.7 ± 12.2% (23-75%), TBR was 11.8 ± 5.8% (2-27%), and TAR was 37.5 ± 13.5% (9-69%). The TIR was highly correlated with HbA1c level, eA1c level, and TAR, but not with TBR. An HbA1c level of 7.0% corresponded to a TIR of 55.1% (95% CI: 53.7-56.5%), whereas a TIR of 70% corresponded to an HbA1c level of 6.1% (95% CI: 5.9-6.3%). The results of eA1c levels were similar to those observed for HbA1c levels. From these findings, we conclude that low rates of a recommended TIR of 70% may be due to less use of advanced technology and insufficient comprehensive diabetes care. Ethnic characteristics including lifestyle and eating customs may have contributed to the result. CGM-derived targets must be individualized based on ethnic characteristics, insulin treatment and diabetes care, and needs of individuals with diabetes.
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Automonitorización de la Glucosa Sanguínea , Glucemia/metabolismo , Diabetes Mellitus Tipo 1/tratamiento farmacológico , Hipoglucemiantes/administración & dosificación , Insulina/administración & dosificación , Monitoreo Ambulatorio , Adolescente , Niño , Consenso , Diabetes Mellitus Tipo 1/metabolismo , Femenino , Hemoglobina Glucada , Humanos , Bombas de Infusión Implantables , Sistemas de Infusión de Insulina , Japón , Masculino , Guías de Práctica Clínica como AsuntoRESUMEN
PURPOSE: Optical coherence tomography angiography (OCTA) was performed on patients with juvenile-onset type 1 diabetes (T1DM) but with no diabetic retinopathy to measure the foveal avascular zone (FAZ) area. STUDY DESIGN: Retrospective single-facility study METHODS: Twenty-nine patients (58 eyes) with juvenile-onset T1DM were studied. Images (3 mm x 3 mm cube centered on the fovea) were acquired using an OCTA device. Age at examination was 16.1 ± 8.7 years; onset age was 6.4 ± 3.5 years; duration of diabetes was 9.7 ± 8.3 years. Twenty-four age-matched healthy individuals were studied as controls. RESULTS: FAZ area was significantly larger in T1DM patients than in controls (0.29 ± 0.09 vs. 0.25 ± 0.08 mm2, P = 0.0234). Parafoveal vessel density was not significantly different between patients and controls (50.43 ± 4.24 vs. 50.07 ± 4.64, P = 0.8842). By generalized linear model analysis, annual HbA1c (P = 0.0190), number of serious hypoglycemic attacks (P = 0.0210), and onset age (P = 0.0447) were identified as variables significantly associated with FAZ area. Age, gender, duration of disease, total cholesterol, high or low-density lipoprotein, triglycerides, and body mass index were not significantly associated with FAZ area. CONCLUSION: Patients with juvenile-onset T1DM and no diabetic retinopathy had increased FAZ, but no significant difference in parafoveal vessel density compared to healthy controls. Larger FAZ area was associated with higher annual HbA1c, more episodes of severe hypoglycemic attacks, and older onset age.
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Diabetes Mellitus Tipo 1/fisiopatología , Fóvea Central/irrigación sanguínea , Isquemia/fisiopatología , Vasos Retinianos/fisiopatología , Adolescente , Adulto , Edad de Inicio , Índice de Masa Corporal , Niño , Preescolar , Colesterol/sangre , HDL-Colesterol/sangre , LDL-Colesterol/sangre , Cromatografía Líquida de Alta Presión , Diabetes Mellitus Tipo 1/sangre , Diabetes Mellitus Tipo 1/diagnóstico por imagen , Femenino , Angiografía con Fluoresceína , Estudios de Seguimiento , Fóvea Central/diagnóstico por imagen , Hemoglobina Glucada/metabolismo , Humanos , Isquemia/sangre , Isquemia/diagnóstico por imagen , Masculino , Vasos Retinianos/diagnóstico por imagen , Estudios Retrospectivos , Tomografía de Coherencia Óptica , Triglicéridos/sangreRESUMEN
PURPOSE: The aim was to investigate the characteristics of abdominal fat distribution in Japanese adolescents with type 2 diabetes mellitus. PATIENTS AND METHODS: Eighty-six Japanese adolescents with simple obesity or type 2 diabetes mellitus treated between 2002 and 2018 were included. The subjects were classified into the simple obesity group (SO group, n=38) and type 2 diabetes mellitus group (DM group, n=23) by matching average age and gender ratio. The metabolic parameters VFA, SFA, and V/S ratio were compared between the 2 groups. Multivariate logistic regression analysis was performed to identify clinical factors associated with type 2 diabetes mellitus. Linear regression analysis was performed between hemoglobin A1c (HbA1c) and visceral fat area (VFA), subcutaneous fat area (SFA), or VFA-to-SFA ratio (V/S ratio) among all enrolled subjects. Finally, correlation analyses were performed to determine the relationships between VFA, SFA, and V/S ratio and metabolic parameters of the DM group. For the metabolic parameters, serum lipids, alanine aminotransferase (ALT), and HbA1c were measured without fasting. The VFA and SFA at umbilical level were investigated using computed tomography. RESULTS: VFA and V/S ratio in DM group were higher than those in SO group (p=0.04 and p<0.01, respectively). SFA in DM group was lower than that in SO group (p<0.01). VFA and SFA, and non-high density lipoprotein (HDL) cholesterol were identified as being independently associated with type 2 diabetes mellitus (odds ratio, 1.05, 0.98, and 1.04, respectively, p<0.05). HbA1c was correlated with VFA and V/S ratio (p<0.01). In DM group, VFA and SFA were positively correlated with systolic blood pressure (p<0.01), ALT (p<0.05), total cholesterol (p<0.05), and non-HDL cholesterol (p<0.01); however, V/S ratio was not correlated. CONCLUSION: Abdominal fat distribution in Japanese adolescents with type 2 diabetes mellitus was different from those with simple obesity and might associate with glucose and lipid metabolism.
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Background Diabetic ketoacidosis (DKA) is a common complication of type 1 diabetes mellitus (T1DM). Infants and children with new-onset T1DM may present with DKA, and the risk of cerebral edema is high in infantile DKA. What is new? Neurological deterioration during an episode of DKA is usually attributed to cerebral edema and cerebrovascular accidents. However, cerebral infarction is a very rare complication in infantile DKA. Case presentation We describe a rare case of infantile cerebral infarction caused by severe DKA in a patient with new-onset T1DM. Conclusions Cerebral infarction is an important intracranial complication in infantile DKA. Careful observation and treatment for DKA during the first 24 h of therapy are necessary in infants with new-onset T1DM because the risk of cerebral infarction is highest during this timeframe.
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Infarto Cerebral/etiología , Diabetes Mellitus Tipo 1/fisiopatología , Cetoacidosis Diabética/complicaciones , Edad de Inicio , Infarto Cerebral/patología , Femenino , Humanos , Lactante , PronósticoRESUMEN
OBJECTIVE: To analyze changes in the annual incidence of school students with type 2 diabetes detected by urine glucose screening at schools in the Tokyo Metropolitan Area during 1975-2015. METHODS: Trend in temporal changes in the annual incidence rate were analyzed using a joinpoint regression model and the joinpoints. Annual percent change (APC) was calculated for each segmented line regression. Average annual percent change (AAPC) was also calculated for the whole period analyzed. RESULTS: In total, 301 students, including 64 primary school students and 237 junior high school students, were diagnosed with type 2 diabetes. The overall incidence of type 2 diabetes (per 100 000/year) during the entire study period was 2.58 in all students, 0.80 in primary school students, and 6.41 in junior high school students. AAPC during the entire study period was estimated at -1.5 (not significant), and the incidence significantly increased during 1975-1982 (APC = 17.49, P < 0.05), but tended to decrease during 1982-2015 (APC = -1.01). In primary school students, the incidence significantly increased during1975-2010 (APC = 3.30, P < 0.05), and tended to decrease during 2010-2015 (APC = -29.61). In junior high school students, the incidence did not significantly change during the entire study period (APC = 0.06). CONCLUSIONS: We found increasing trend in the overall incidence of school students with type 2 diabetes during 1975-1982, but a decreased tendency in recent years. This could be due to changes observed during the same time period in the primary school students. Lifestyle changes might contribute to improved incidence of childhood type 2 diabetes.
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Diabetes Mellitus Tipo 2/epidemiología , Instituciones Académicas , Estudiantes/estadística & datos numéricos , Población Urbana , Adolescente , Niño , Femenino , Humanos , Incidencia , Masculino , Estudios Retrospectivos , Instituciones Académicas/estadística & datos numéricos , Instituciones Académicas/tendencias , Tokio/epidemiología , Población Urbana/estadística & datos numéricos , Población Urbana/tendenciasRESUMEN
BACKGROUND: Neurological manifestations caused by hypoglycemia range from reversible focal deficits and transient encephalopathy to irreversible coma or death. Recently, high signal intensity lesions in the splenium of the corpus callosum on diffusion-weighted magnetic resonance imaging were reported in adults experiencing hypoglycemia. However, patients presenting with agraphia are rare. SUBJECT AND METHODS: We examined a 17-year-old left-handed female patient with type 1 diabetes who exhibited transient left agraphia with a reversible splenium lesion of the corpus callosum on diffusion-weighted imaging caused by hypoglycemia, which was improved with blood glucose management alone. CONCLUSION: This rare case indicates that agraphia, a sign of callosal disconnection syndrome, can result from a reversible splenial lesion of the corpus callosum caused by hypoglycemia.
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Agrafia/diagnóstico por imagen , Agrafia/etiología , Cuerpo Calloso/diagnóstico por imagen , Hipoglucemia/complicaciones , Hipoglucemia/diagnóstico por imagen , Adolescente , Agrafia/fisiopatología , Agrafia/terapia , Cuerpo Calloso/fisiopatología , Diabetes Mellitus Tipo 1/complicaciones , Diabetes Mellitus Tipo 1/diagnóstico por imagen , Diabetes Mellitus Tipo 1/fisiopatología , Diabetes Mellitus Tipo 1/terapia , Imagen de Difusión por Resonancia Magnética , Femenino , Humanos , Hipoglucemia/fisiopatología , Hipoglucemia/terapiaRESUMEN
BACKGROUND: We conducted an annual urine glucose screening program at schools, and diagnosed schoolchildren with diabetes at an early stage of the disease. We also identified some cases of renal glucosuria (RG), based on positive urine glucose with normal glucose tolerance. METHODS: During 2000-2015, 3 309 631 schoolchildren participated in the screening program. The positive rate for glucosuria in the first test was approximately 0.1%, whereas on repeat urine test it was approximately 0.05%. In total 350 schoolchildren were positive for glucosuria on detailed examination. Oral glucose tolerance test (OGTT) was also used to evaluate glucose intolerance. RESULTS: One hundred and two schoolchildren (29.7%) were diagnosed with diabetes, whereas RG was identified in 246 (70.3%) with normal glucose metabolism. In regard to the characteristics of RG, the percentage of boys was 50.3%, and the mean age at diagnosis was 11.2 ± 2.4 years. Twenty-eight children (11.4%) were overweight (body mass index standard deviation score [BMI-SDS] > +2.0 SD), whereas five (2.0%) were underweight (BMI-SDS < -2.0 SD). First-degree family history was suspected in 176 cases (71.5%). All RG subjects had normal glucose tolerance in the absence of insulin resistance and decreased insulin secretion (homeostasis model assessment for ß-cell function, 78.8 ± 59.5%) on OGTT. CONCLUSIONS: RG is not rare in Japanese schoolchildren with glucosuria. This disorder seems to have a strong genetic background, and to involve less growth retardation and weight loss than expected despite continuous excretion of glucose in urine.
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Intolerancia a la Glucosa/diagnóstico , Glucosuria Renal/diagnóstico , Niño , Femenino , Intolerancia a la Glucosa/epidemiología , Intolerancia a la Glucosa/orina , Prueba de Tolerancia a la Glucosa , Glucosuria Renal/epidemiología , Glucosuria Renal/orina , Humanos , Japón/epidemiología , Masculino , Tamizaje MasivoRESUMEN
The first-line pharmacological treatment for patients with maturity-onset diabetes of the young type 1 (MODY1) and maturity-onset diabetes of the young type 3 (MODY3) are sulfonylureas (SUs) or insulin. However, several reports have suggested the possibility of using incretin-associated drugs, including dipeptidyl-peptidase-4 (DPP-4) inhibitors, for the treatment of patients with these types of MODY. Here we report a case of a pediatric patient with MODY1 who was successfully treated with a DPP-4 inhibitor, alogliptin. A 13-yr-old Japanese girl with diabetes was initially treated with insulin for 5 mo. After diagnosis of MODY1, confirmed via a genetic analysis, treatment was changed from insulin to alogliptin. SUs were prescribed temporarily, but monotherapy with alogliptin finally resulted in good glycemic control. After changing to alogliptin, the patient maintained optimal glycemic control with glycated hemoglobin levels of 6.3-7.0% while maintaining substantial ß-cell function. No adverse events associated with alogliptin were observed. These results suggest that DPP-4 inhibitors may be a potential treatment for patients with MODY1 at the early stage of the disease when residual insulin secretion is still being sustained.
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We evaluated the efficacy and safety of insulin glulisine (GLU) used for continuous s.c. insulin infusion (CSII) in 20 children with type 1 diabetes after 1 year of GLU treatment. There were no significant differences in mean plasma glucose before breakfast and before dinner between before and after using GLU, but the levels after breakfast and after dinner significantly improved, from 192.5 ± 31.7 to 162.0 ± 27.3 mg/dL for breakfast, and from 191.1 ± 33.3 to 161.1 ± 24.5 mg/dL for dinner (P < 0.01). Mean hemoglobin A1c significantly decreased (from 8.0 ± 0.8 to 7.7 ± 0.8%, P < 0.05), and the mean frequency of hypoglycemia significantly reduced after using GLU (from 8.3 ± 4.9 to 6.0 ± 3.4/month, P < 0.05). In conclusion, the use of GLU rather than other rapid-acting analogues for CSII might be an effective treatment option in children with type 1 diabetes.