Severely obese 14-year-old boy with central sleep apnea several years after head trauma.

Central sleep apnea (CSA) is rare in older children. Although CSA mostly arises from neurological diseases such as Chiari malformation, the frequency of CSA is significantly higher in obese children. Herein, we describe the case of a 14-yr-old boy who presented with CSA secondary to severe obesity and a history of traumatic lateral medullary syndrome at 8 yr of age. Polysomnography revealed severe sleep apnea syndrome with apnea-hypopnea index of 41.4 per hour and central apnea index of 8.9 per hour. Magnetic resonance imaging of the head showed no new brainstem or cerebellar infarcts; however, old changes in the cerebellar infarction persisted. Obesity is primarily associated with obstructive sleep apnea. However, obesity can result in CSA through pharyngeal collapse and the reduction of oxygen reserves caused by reduced thoracic volume, which suppresses respiratory center stimulation. Because the respiratory center disorder owing to head injury sequelae improved after the acute stage, obesity was deemed the cause of CSA in this case. Hence, children with severe obesity may require CSA monitoring.

Hypertensive disorders of pregnancy increase the incidence of febrile seizures in offspring.

BACKGROUND: Intrauterine exposure to hypertensive disorders of pregnancy (HDP) may increase the risk of neuropsychiatric disorders. This investigation examined for associations between maternal HDP and febrile seizures (FS) in offspring by the age of three years. METHODS: The present cohort study analyzed data from the Japan Environment and Children's Study, a large national birth cohort. We included mother-child pairs recruited between January 2011 and March 2014. Information regarding maternal HDP, the presence of FS in offspring up to 3 years of age, and potential confounding factors were assessed using written questionnaires administered to mothers. RESULTS: A total of 77,699 mother-child dyads were analyzed. The prevalence of FS was 8.4% in children without HDP exposure, 10.6% in those exposed to mild HDP, and 10.4% in those with severe HDP exposure. Among children with full-term birth, logistic regression analysis indicated that exposure to mild or severe HDP was significantly associated with a higher incidence of FS (adjusted odds ratio [95% confidence interval]: 1.27 [1.05-1.53] and 1.27 [0.90-1.78], respectively, P for trend = 0.008), compared with children without HDP exposure. CONCLUSION: In children with full-term birth, intrauterine exposure to HDP was significantly associated with FS by the age of three years. IMPACT: This study revealed a significant association between intrauterine exposure to hypertensive disorders of pregnancy (HDP) and the subsequent development of febrile seizures (FS) in offspring by three years. This increased incidence of FS by HDP was independent of preterm birth status. This is the first large nationwide birth cohort study showing the impact of intrauterine exposure to HDP on FS in early childhood.

Impact of breastfeeding during infancy on functional constipation at 3 years of age: the Japan Environment and Children's Study.

BACKGROUND: There is a lack of large, nationwide, birth cohort studies in Japan that examine the relationships of initial feeding habits and breastfeeding period duration with offspring functional constipation at 3 years of age. This study assessed the impact of breastfeeding during infancy on early childhood functional constipation. METHODS: The fixed data of 70,078 singleton births from the ongoing Japan Environment and Children's Study cohort study that commenced in 2011 were used to identify functional constipation as estimated by Rome III at 3 years of age. The exposure variables were breastfeeding period duration until 12 months of age (never, up to 6 months, or ≥ 7 months) as well as breastfeeding status at 1 month and 6 months of age (breastfeeding exclusively, partial breastfeeding, or infant formula feeding only). Multiple logistic regression analysis was employed to search for correlations for functional constipation development with breastfeeding period duration until 12 months of age and breastfeeding status during infancy. RESULTS: We identified 8,118 toddlers (11.6%) who met the Rome III criteria at 3 years of age. After controlling for potential covariates, a breastfeeding period duration of 7 months or more was inversely related to functional constipation development (≥ 7 months: adjusted odds ratio [OR] [95% confidence interval (CI)] 0.76 [0.65, 0.88] versus never breastfed, P for trend < 0.001). Other initial feeding methods were significantly related to an increased risk of functional constipation as compared with breastfeeding exclusively at 1 month of age (partial breastfeeding: adjusted OR [95% CI] 1.17 [1.11, 1.23], formula feeding only: 1.23 [1.07, 1.40]) and 6 months of age (partial breastfeeding: adjusted OR [95% CI] 1.18 [1.12, 1.24], formula feeding only: adjusted OR [95% CI] 1.42 [1.20, 1.68]). CONCLUSION: This large nationwide survey revealed a possible protective effect of a prolonged breastfeeding period duration and early exclusive breastfeeding in infancy on functional constipation at 3 years.

Impact of dog and/or cat ownership on functional constipation at 3 years of age: the Japan Environment and Children's study.

PURPOSE: This investigation assessed the impact of dog and/or cat ownership during infancy on the presence of functional constipation (FC) at 3 years of age. METHODS: The fixed data of 73,936 singleton births from a large national birth cohort study commencing in 2011 were used to identify FC as estimated by Rome III at 3 years of age. Multiple logistic regression analysis was employed to search for correlations between FC development and dog and/or cat ownership in early childhood. RESULTS: A total of 8,459 toddlers (11.6%) met the Rome III criteria for FC at 3 years of age. Overall, 57,264 (77.5%) participants had never owned a dog or cat. We identified 7,715 (10.4%) infant-period owners, 1,295 (1.8%) current owners, and 7,762 (10.5%) long-term owners. Multivariate analysis showed that infant-period ownership remained significantly associated with the risk of developing FC at 3 years of age after adjusting for covariates (adjusted OR [95% CI] 1.09 [1.01-1.19] based on non-ownership). CONCLUSIONS: This Japanese large nationwide survey uncovered a possible adverse effect of infant-period dog and/or cat ownership prior to 6 months of age on FC status at 3 years of age.

Familial and lifestyle factors related to physical activity in elementary school students: a cross-sectional study based on a nationally representative survey in Japan.

PURPOSE: The decrease in physical activity (PA) among children has become a global concern. Since the analysis of sociodemographic factors as determinants of exercise habits has been inconclusive, this study investigated the factors related to participation in organized sports and moderate-to-vigorous PA (MVPA) levels. METHODS: Cross-sectional data from the Sports-Life Survey conducted in 2019 by the Sasagawa Sports Foundation were used. Data on the gender, age, grade, annual household income, family members, and lifestyle habits of elementary school children as well as information on participation in organized sports and MVPA were collected by written questionnaires. Multiple logistic regression models were applied to calculate the adjusted odds ratio and 95% confidence interval for the association of each variable with participation in organized sports and frequent MVPA (≥ 60 min/day for ≥ 5 days/week). RESULTS: A total of 1,197 participants were included in the analysis. Whereas 1,053 (88.2%) students expressed a like for PA, only 725 students (60.8%) actually took part in organized sports. Organized sports participation was significantly associated with gender, grade, population density, household income, daily breakfast, lower screen time, and frequent exercise with parents (all P < 0.05). We observed that 12.3% of participants met the frequent MVPA level, which was significantly related to lower screen time and exercise habits with parents (both P < 0.05). CONCLUSIONS: Social and family factors may be strong determinants of engagement in PA among Japanese elementary school-aged children. Parental involvement appears particularly important for promoting PA among youths.

Impact of maternal dyslipidemia on infant neurodevelopment: The Japan Environment and Children's Study.

BACKGROUND: Various genetic and environmental influences have been studied for developmental disorders; however, the precise cause remains unknown. This study assessed the impact of maternal serum total cholesterol (TC) level in early pregnancy on early childhood neurodevelopment. METHODS: The fixed data of 31,797 singleton births from a large national birth cohort study that commenced in 2011 were used to identify developmental disorders as estimated by Ages and Stages Questionnaire, third edition (ASQ-3) scores of less than -2 standard deviations at 12 months of age. Multiple logistic regression analysis was employed to search for correlations between possibility of developmental disorders and maternal TC levels in early pregnancy classified into 4 groups based on quartile (Q1-Q4) values. RESULTS: After controlling for potential confounding factors in 27,836 participants who ultimately underwent multivariate analysis, we observed that elevated TC levels were significantly associated with a higher risk of screen positive status for communication (Q4: adjusted odds ratio [aOR] 1.20, 95% confidence interval [CI] 1.05-1.37) and gross motor (aOR 1.13, 95% CI 1.03-1.25) ASQ-3 domain scores. CONCLUSION: This large nationwide survey revealed a possible deleterious effect of hypercholesterolemia in early pregnancy on infant neurodevelopment and age-appropriate skill acquisition at 12 months age.

Insufficient maternal gestational weight gain and infant neurodevelopment at 12 months of age: the Japan Environment and Children's Study.

Abnormal maternal gestational weight gain (GWG) increases the risk of obstetric-related complications. This investigation examined the impact of GWG on infant neurodevelopmental abnormalities at 12 months of age using the data of a nationwide Japanese cohort study. Questionnaire data were obtained from the ongoing Japan Environment and Children's Study cohort study. Maternal GWG was subdivided as below, within, or above the reference values of the Institution of Medicine pregnancy weight guidelines. The Ages and Stages Questionnaire, third edition (ASQ-3) is a parent-reported developmental screening instrument for children across five domains: communication, gross motor, fine motor, problem-solving, and personal-social. Multiple logistic regression analysis was employed to identify correlations between GWG and developmental delay defined as ASQ-3 scores of less than two standard deviations below the mean. A total of 30,694 mothers with singleton live births and partners who completed the questionnaire were analyzed. The prevalence of mothers below, within, and above the GWG guidelines was 60.4% (18,527), 32.1% (9850), and 7.5% (2317), respectively. We recorded 10,943 infants (35.7%) who were outliers in at least one ASQ-3 domain. After controlling for covariates, GWG below established guidelines was associated with a significantly higher risk of developmental delay for the communication (odds ratio [OR] 1.21, 95% confidence interval [CI] 1.09-1.34), gross motor (OR 1.14, 95% CI 1.05-1.24), fine motor (OR 1.13, 95% CI 1.04-1.24), problem-solving (OR 1.09, 95% CI 1.01-1.18), and personal-social (OR 1.15, 95% CI 1.07-1.24) domains.Conclusion: This large survey revealed a possible deleterious effect of insufficient maternal GWG on infant neurodevelopment.Trial registration: The Japan Environment and Children's Study (JECS) was registered in the UMIN Clinical Trials Registry on January 15, 2018 (number UMIN000030786). What is Known: • Inappropriate maternal gestational weight gain may cause obstetric complications and adverse birth outcomes. • Excess maternal weight gain may result in gestational diabetes, hypertension, eclampsia, caesarean delivery, and macrosomia, while insufficient maternal weight gain has been associated with pre-term birth and small for gestational age. What is New: • This study provides important information on a possible adverse effect of insufficient maternal gestational weight gain on offspring neurodevelopment at 12 months of age. • Our findings indicate a need to reconsider the optimal body mass index and gestational weight gain for women desiring pregnancy.

Association between pesticide usage during pregnancy and neonatal hyperbilirubinemia requiring treatment: the Japan Environment and Children's Study.

BACKGROUND: Maternal exposure to pesticides during pregnancy may cause oxidative hemolysis leading to neonatal hyperbilirubinemia. This investigation examined for associations between maternal use of pesticides or repellents during pregnancy and neonatal hyperbilirubinemia requiring phototherapy. METHODS: We used the dataset from the Japan Environment and Children's Study, a large national birth cohort study registered from January 31, 2011 to March 31, 2014. The fixed data of 61,751 live births were used to evaluate the presence of neonatal hyperbilirubinemia and potential confounding factors. We employed multiple logistic regression analysis to identify correlations between the frequency of maternal pesticide or repellent use during pregnancy and clinically relevant neonatal hyperbilirubinemia. RESULTS: After controlling for confounding factors, there were significant associations between neonatal hyperbilirubinemia necessitating phototherapy and the frequent use of indoor insecticide spray (OR 1.21, 95% CI 1.05-1.38). For spray- or lotion-type insect repellents, an opposite relationship was observed (more than a few times a week: OR 0.70, 95% CI 0.61-0.81, up to a few times a month: OR 0.84, 95% CI 0.78-0.91). CONCLUSION: The frequent use of indoor insecticide spray during pregnancy showed an increased risk of neonatal hyperbilirubinemia requiring phototherapy, which was absent for spray- or lotion-type insect repellents. IMPACT: The frequent use of indoor insecticide spray during pregnancy showed an increased risk of neonatal hyperbilirubinemia requiring phototherapy, which was absent for spray- or lotion-type insect repellents. This is the first study examining the effects of maternal exposure to pesticides or repellents on clinically relevant neonatal hyperbilirubinemia using a dataset from a nationwide birth cohort study. This large-scale Japanese cohort study revealed that the frequent use of indoor insecticide spray during pregnancy may increase the risk of neonatal hyperbilirubinemia requiring treatment.

Maternal Exposure to Housing Renovation During Pregnancy and Risk of Offspring with Congenital Malformation: The Japan Environment and Children's Study.

There have been no large, nationwide, birth cohort studies in Japan examining the effects of house renovation during pregnancy on congenital abnormality. This study examined the impact of (1) prenatal exposure to house renovation and (2) maternal occupational exposure to organic solvents and/or formaldehyde on the incidence of congenital abnormality. The fixed data of 67,503 singleton births from a large national birth cohort study that commenced in 2011 were used to evaluate the presence of congenital abnormalities and potential confounding factors. We employed multiple logistic regression analysis to search for correlations between maternal exposure to house renovation or organic solvents and/or formaldehyde during pregnancy and such congenital abnormalities as congenital heart disease, cleft lip and/or palate, male genital abnormality, limb defect, and gastrointestinal obstruction. After controlling for potential confounding factors, we observed that house renovation was significantly associated with male genital abnormality (OR 1.81, 95% CI 1.03-3.17, P = 0.04) when stratified by congenital abnormality, with no other remarkable relations to house renovation or occupational use of organic solvents and/or formaldehyde during pregnancy. There were also significant correlations for maternal BMI before pregnancy, history of ovulation induction through medication, maternal diabetes mellitus/gestational diabetes mellitus, and hypertensive disorder of pregnancy with an increased risk of congenital abnormality. In conclusion, this large nationwide survey provides important information on a possible association of house renovation during pregnancy with congenital male genital abnormality which needs confirmation in future studies.

Alcohol Consumption During Pregnancy and Risk of Placental Abnormality: The Japan Environment and Children's Study.

There have been no large nationwide birth cohort studies examining for the effects of maternal alcohol use during pregnancy on placental abnormality. This study searched for associations between alcohol consumption and the placental abnormalities of placenta previa, placental abruption, and placenta accreta using the fixed dataset of a large national birth cohort study commencing in 2011 that included 80,020 mothers with a singleton pregnancy. The presence of placental abnormalities and potential confounding factors were recorded, and multiple logistic regression analysis was employed to search for correlations between maternal alcohol consumption during pregnancy and placental abnormalities. The overall rate of prenatal drinking until the second/third trimester was 2.7% (2,112). The prevalence of placenta previa, placental abruption, and placenta accreta was 0.58% (467), 0.43% (342), and 0.20% (160), respectively. After controlling for potential confounding factors, maternal alcohol use during pregnancy was significantly associated with the development of placenta accreta (OR 3.10, 95%CI 1.69-5.44). In conclusion, this large nationwide survey revealed an association between maternal drinking during pregnancy and placenta accreta, which may lead to excessive bleeding during delivery.

Glucose Management during Insulinoma Resection Using Real-Time Subcutaneous Continuous Glucose Monitoring.

Insulinoma is a rare neuroendocrine tumor that causes hypoglycemia due to unregulated insulin secretion. Blood glucose management during insulinoma resection is therefore challenging. We present a case in which real-time subcutaneous continuous glucose monitoring (SCGM) in combination with intermittent blood glucose measurement was used for glycemic control during surgery for insulinoma resection. The SCGM system showed the trends and peak of interstitial glucose in response to glucose loading and the change of interstitial glucose before and after insulinoma resection. These data were helpful for adjusting the glucose infusion; therefore, we think that an SCGM system as a supportive device for glucose monitoring may be useful for glucose management during surgery.

Clinical and laboratory outcomes after umbilical cord blood transplantation in a patient with mucolipidosis II alpha/beta.

Mucolipidosis (ML) II alpha/beta is an autosomal recessive disease caused by reduced enzyme activity of N-acetylglucosamine-1-phosphotransferase. Clinical symptoms of ML II are severe psychomotor delay and dysostosis multiplex; death usually occurs by 5-8 years of age from cardiopulmonary complications. Allogeneic hematopoietic stem cell transplantation (HSCT) has been attempted for ML; however, few reports have documented the detailed outcomes of HSCT for ML. A 26-month-old girl received a human leukocyte antigen 3/6-allele-matched transplant from cord blood. The preparative regimen consisted of fludarabine, cyclophosphamide, 6-Gy total body irradiation, and rabbit antithymocyte globulin. Although comparing before and after cord blood transplantation results, we observed that lysosomal enzyme activities in the plasma decreased by approximately 20-40%. Low serum levels of immunoglobulin A, G2, and G4 were also observed before HSCT; however, these values normalized after transplantation. Despite undergoing HSCT, she was treated twice for bacterial pneumonia with acute respiratory distress syndrome at ages 37 and 38 months. Although HSCT effects on the clinical manifestations were limited, laboratory data including plasma lysosomal enzyme activities and serum levels of immunoglobulin showed improvement.