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1.
Int J Obes (Lond) ; 48(5): 717-724, 2024 May.
Article in English | MEDLINE | ID: mdl-38302592

ABSTRACT

BACKGROUND/OBJECTIVES: Delivery by cesarean section (CS) compared to vaginal delivery has been associated with increased risk of overweight in childhood. Our study examined if the presence or absence of labor events in CS delivery altered risk of overweight in early childhood (1-5 years) compared to vaginal delivery and if this association differed according to infant sex. SUBJECTS/METHODS: The study included 3073 mother-infant pairs from the CHILD Cohort Study in Canada. Data from birth records were used to categorize infants as having been vaginally delivered, or delivered by CS, with or without labor events. Age and sex adjusted weight-for-length (WFL) and body mass index (BMI) z scores were calculated from height and weight data from clinic visits at 1, 3 and 5 years and used to classify children as overweight. Associations between delivery mode and child overweight at each timepoint were assessed using regression models, adjusting for relevant confounding factors including maternal pre-pregnancy BMI. Effect modification by infant sex was tested. RESULTS: One in four infants (24.6%) were born by CS delivery; 13.0% involved labor events and 11.6% did not. Infants born by CS without labor had an increased odds of being overweight at age 1 year compared to vaginally delivered infants after adjustment for maternal pre-pregnancy BMI, maternal diabetes, smoking, infant sex and birthweight-for-gestational age (aOR 1.68 [95% CI 1.05-2.67]). These effects did not persist to 3 or 5 years of age and, after stratification by sex, were only seen in boys (aOR at 1 year 2.21 [95% CI 1.26-3.88]). CONCLUSION AND RELEVANCE: Our findings add to the body of evidence that CS, in particular CS without labor events, may be a risk factor for overweight in early life, and that this association may be sex-specific. These findings could help to identify children at higher risk for developing obesity.


Subject(s)
Cesarean Section , Pediatric Obesity , Humans , Female , Cesarean Section/statistics & numerical data , Cesarean Section/adverse effects , Canada/epidemiology , Pediatric Obesity/epidemiology , Male , Pregnancy , Infant , Longitudinal Studies , Child, Preschool , Adiposity , Body Mass Index , Risk Factors , Adult , Infant, Newborn , Delivery, Obstetric/statistics & numerical data , Delivery, Obstetric/methods
2.
Int J Mol Sci ; 24(9)2023 Apr 28.
Article in English | MEDLINE | ID: mdl-37175718

ABSTRACT

The autonomic nervous system (ANS) may play a role in the distribution of body fat and the development of obesity and its complications. Features of individuals with Prader-Willi syndrome (PWS) impacted by PWS molecular genetic classes suggest alterations in ANS function; however, these have been rarely studied and presented with conflicting results. The aim of this study was to investigate if the ANS function is altered in PWS. In this case-control study, we assessed ANS function in 20 subjects with PWS (6 males/14 females; median age 10.5 years) and 27 body mass index (BMI) z-score-matched controls (19 males/8 females; median age 12.8 years). Standardized non-invasive measures of cardiac baroreflex function, heart rate, blood pressure, heart rate variability, quantitative sudomotor axon reflex tests, and a symptom questionnaire were completed. The increase in heart rate in response to head-up tilt testing was blunted (p < 0.01) in PWS compared to controls. Besides a lower heart rate ratio with Valsalva in PWS (p < 0.01), no significant differences were observed in other measures of cardiac function or sweat production. Findings suggest possible altered sympathetic function in PWS.


Subject(s)
Pediatric Obesity , Prader-Willi Syndrome , Male , Female , Humans , Child , Prader-Willi Syndrome/complications , Pediatric Obesity/complications , Case-Control Studies , Body Mass Index , Autonomic Nervous System
3.
Paediatr Child Health ; 28(2): 107-112, 2023 May.
Article in English | MEDLINE | ID: mdl-37151929

ABSTRACT

Background: The co-presentation of severe obesity (SO) and global developmental delay (GDD) in Canadian preschool children has not been examined. However, SO and GDD may require syndromic diagnoses and unique management considerations. Objectives: To determine (1) minimum incidence; (2) age of onset and risk factors; and (3) health care utilization for co-presenting SO and GDD. Methods: Through the Canadian Paediatric Surveillance Program (CPSP), a monthly form was distributed to participants from February 2018 to January 2020 asking for reports of new cases of SO and GDD among children ≤5 years of age. We performed descriptive statistics for quantitative questions and qualitative content analysis for open-ended questions. Results: Forty-seven cases (64% male; 51% white; mean age: 3.5 ± 1.2 years) were included. Age of first weight concern was 2.5 ± 1.3 years and age of GDD diagnosis was 2.7 ± 1.4 years. Minimum incidence of SO and GDD was 3.3 cases per 100,000 for ≤5 years of age per year. Identified problems included school and/or behavioural problems (n = 17; 36%), snoring (n = 14; 30%), and asthma/recurrent wheeze (n = 10; 21%). Mothers of 32% of cases (n = 15) had obesity and 21% of cases (n = 10) received neonatal intensive care. Microarray was ordered for 57% (n = 27) of children. A variety of clinicians and services were accessed. As reported by CPSP participants, challenges faced by families and health service access were barriers to care. Conclusion: Children with SO and GDD have multiple comorbidities, and require early identification and referral to appropriate services. These cases may also benefit from additional testing to rule out known genetic obesity syndromes.

4.
Int J Obes (Lond) ; 46(9): 1712-1719, 2022 09.
Article in English | MEDLINE | ID: mdl-35840772

ABSTRACT

BACKGROUND/OBJECTIVES: Differences in gut microbiota, metabolites and immune markers have been observed between individuals with and without obesity. Our study determined the temporal association between infant fecal gut metabolites, sIgA and body mass index (BMI) z score of preschool children, independent of pre/postnatal factors. SUBJECTS/METHODS: The study includes a subset of 647 infants from the CHILD Cohort Study (recruited between January 1, 2009, and December 31, 2012). Fecal metabolites and sIgA were measured at 3-4 months of age, and age and sex adjusted BMI z scores at 1 and 3 years of age. Associations between the metabolites, IgA, and child BMI z scores at age 1 and 3 years were tested using linear regression adjusted for pre/postnatal factors (breastfeeding, birthweight-for-gestational age, birthmode and IAP, solid food introduction). RESULTS: Mean BMI z score for all infants was 0.34 (SD 1.16) at 1 year (N = 647) and 0.71 (SD 1.06) at 3 years (N = 573). High fecal formate in infancy was associated with a significantly lower BMI z score (adjusted mean difference -0.23 (95% CI -0.42, -0.04)) and high butyrate was associated with a higher BMI z score (adjusted mean difference 0.21 (95% CI 0.01, 0.41)) at age 3 years only. The influence of formate and butyrate on BMI z score at age 3 were seen only in those that were not exclusively breastfed at stool sample collection (adjusted mean difference for high formate/EBF- group: -0.33 (95%CI -0.55, -0.10) and 0.25 (95% CI 0.02, 0.47) for high butyrate/EBF- group). No associations were seen between sIgA and BMI z score at age 1 or 3 years in adjusted regression models. CONCLUSION AND RELEVANCE: Differences in fecal metabolite levels in early infancy were associated with childhood BMI. This study identifies an important area of future research in understanding the pathogenesis of obesity.


Subject(s)
Immunoglobulin A, Secretory , Pediatric Obesity , Body Mass Index , Butyrates , Child , Child, Preschool , Cohort Studies , Female , Formates , Humans , Infant , Obesity , Pediatric Obesity/epidemiology , Prospective Studies
5.
Int J Mol Sci ; 23(18)2022 Sep 13.
Article in English | MEDLINE | ID: mdl-36142494

ABSTRACT

Debate remains as to how to balance the use of recombinant human growth hormone (rhGH) as an important treatment in Prader-Willi syndrome (PWS) with its potential role in obstructive sleep apnea. This single-center, retrospective study assessed differences in overnight polysomnography results between children with and without PWS and changes in respiratory parameters before and after the initiation of rhGH treatment in those with PWS. Compared with age-, sex-, and body-mass-index-matched controls (n = 87), children with PWS (n = 29) had longer total sleep time (434 ± 72 vs. 365 ± 116 min; p < 0.01), higher sleep efficiency (86 ± 7 vs. 78 ± 15%; p < 0.05), and lower arousal events (8.1 ± 4.5 vs. 13.0 ± 8.9 events/h; p < 0.05). Mean oxygen saturation was lower in PWS children (94.3 ± 6.0 vs. 96.0 ± 2.0%; p < 0.05), with no other differences in respiratory parameters between groups. Eleven children with PWS (38%) met the criteria for further analyses of the impact of rhGH; polysomnography parameters did not change with treatment. Compared with other children undergoing polysomnography, children with PWS had more favorable markers of sleep continuity and lower oxygen saturation for the same level of respiratory disturbance. rhGH administration was not associated with changes in respiratory parameters in PWS.


Subject(s)
Human Growth Hormone , Prader-Willi Syndrome , Child , Human Growth Hormone/therapeutic use , Humans , Polysomnography , Prader-Willi Syndrome/complications , Prader-Willi Syndrome/drug therapy , Retrospective Studies , Sleep
6.
Int J Mol Sci ; 23(23)2022 Dec 01.
Article in English | MEDLINE | ID: mdl-36499438

ABSTRACT

Syndromic and non-syndromic obesity conditions in children, such as Prader-Willi syndrome (PWS) and non-alcoholic fatty liver disease (NAFLD), both lower quality of life and increase risk for chronic health complications, which further increase health service utilization and cost. In a pilot observational study, we compared body composition and muscle strength in children aged 7−18 years with either PWS (n = 9), NAFLD (n = 14), or healthy controls (n = 16). Anthropometric and body composition measures (e.g., body weight, circumferences, skinfolds, total/segmental composition, and somatotype), handgrip strength, six minute-walk-test (6MWT), physical activity, and markers of liver and cardiometabolic dysfunction (e.g., ALT, AST, blood pressure, glucose, insulin, and lipid profile) were measured using standard procedures and validated tools. Genotyping was determined for children with PWS. Children with PWS had reduced lean body mass (total/lower limb mass), lower handgrip strength, 6MWT and increased sedentary activity compared to healthy children or those with NAFLD (p < 0.05). Children with PWS, including those of normal body weight, had somatotypes consistent with relative increased adiposity (endomorphic) and reduced skeletal muscle robustness (mesomorphic) when compared to healthy children and those with NAFLD. Somatotype characterizations were independent of serum markers of cardiometabolic dysregulation but were associated with increased prevalence of abnormal systolic and diastolic blood pressure Z-scores (p < 0.05). Reduced lean body mass and endomorphic somatotypes were associated with lower muscle strength/functionality and sedentary lifestyles, particularly in children with PWS. These findings are relevant as early detection of deficits in muscle strength and functionality can ensure effective targeted treatments that optimize physical activity and prevent complications into adulthood.


Subject(s)
Non-alcoholic Fatty Liver Disease , Prader-Willi Syndrome , Child , Humans , Adult , Prader-Willi Syndrome/complications , Non-alcoholic Fatty Liver Disease/etiology , Pilot Projects , Hand Strength , Quality of Life , Body Composition , Muscle Strength , Obesity , Body Mass Index
7.
Eat Weight Disord ; 26(3): 879-886, 2021 Apr.
Article in English | MEDLINE | ID: mdl-32430885

ABSTRACT

BACKGROUND: The pediatric relative fat mass (RFM) has been recently presented and validated as an index for estimating percentage body fat (%BF) in North American children and adolescents. Similar to body mass index (BMI) and tri-ponderal mass index (TMI), RFM uses anthropometric measures (i.e., weight, height and waist circumference) to estimate body composition. The primary purpose of this study was to validate the newly developed RFM equation for %BF prediction in Southern Brazilian adolescents; as secondary objective, we compared %BF estimation from BMI- and TMI-derived equations. METHODS: A total of 631 individuals (434 boys) aged 11 to 18 were analyzed. Bland-Altman analyses were used to determine concordance between predicted equations and %BF measured by DXA; results are presented using mean difference (i.e., bias) and standard deviation. Sensitivity and specificity were calculated for %BF percentile classifications. RESULTS: RFM underestimated %BF in 65.2% of boys (- 4.3 ± 2.8%) and 84.8% of girls (- 5.3 ± 2.7%). In contrast, TMI overestimated %BF in 62.9% of boys (4.0 ± 2.9%) and 56.3% (3.5 ± 3.0%) of girls. The performance of BMI showed mixed results; %BF was overestimated in 68.4% of boys (5.0 ± 4.0%) and underestimated in 67.5% of girls (- 3.9 ± 2.6%), all p < 0.001. Although, RFM had the highest specificity for %BF percentile classifications, sensitivity was low and inferior to BMI and TMI. CONCLUSION: TMI was superior to RFM and BMI in predicting %BF in Southern Brazilian adolescents. Using RFM, BMI or TMI equations for %BF prediction without a population-specific correction factor may lead to incorrect interpretations. We suggest that correction factors should be investigated to improve the accuracy of these surrogate indices for body composition assessment. LEVEL OF EVIDENCE: Level V, cross sectional descriptive study.


Subject(s)
Adipose Tissue , Body Composition , Absorptiometry, Photon , Adiposity , Adolescent , Body Mass Index , Brazil , Child , Cross-Sectional Studies , Female , Humans , Male , Waist Circumference
8.
Clin Endocrinol (Oxf) ; 93(5): 579-589, 2020 11.
Article in English | MEDLINE | ID: mdl-32638409

ABSTRACT

BACKGROUND: The effects of dietary macronutrients on orexigenic and anorexigenic hormones in children are poorly understood. OBJECTIVE: To explore effects of varying dietary macronutrients on appetite-regulating hormones [acyl ghrelin (AG) and desacyl ghrelin (DAG), glucagon-like peptide 1 (GLP-1), peptide tyrosine tyrosine (PYY) and insulin] in children with PWS and healthy children (HC). DESIGN: Randomized, cross-over experiments compared two test diets [high protein-low carbohydrate (HP-LC) and high protein-low fat (HP-LF)] to a STANDARD meal (55% carbohydrate, 30% fat, 15% protein). Experiment 1 included ten children with PWS (median age 6.63 years; BMI z 1.05); experiment 2 had seven HC (median age 12.54 years; BMI z 0.95). Blood samples were collected at baseline and at 60-minute intervals for 4 hours. Independent linear mixed models were adjusted for age, sex and BMI z-score. RESULTS: Fasting and post-prandial AG and DAG concentrations are elevated in PWS children; the ratio of AG/DAG is normal. Food consumption reduced AG and DAG concentrations in both PWS and HC. GLP-1 levels were higher in PWS after the HP-LC and HP-LF meals than the STANDARD meal (P = .02-0.04). The fasting proinsulin to insulin ratio (0.08 vs 0.05) was higher in children with PWS (P = .05) than in HC. Average appetite scores in HC declined after all three meals (P = .02) but were lower after the HP-LC and HP-LF meals than the STANDARD meal. CONCLUSION: Altered processing of proinsulin and increased GLP-1 secretion in children with PWS after a high protein meal intake might enhance satiety and reduce energy intake.


Subject(s)
Prader-Willi Syndrome , Blood Glucose , Child , Fasting , Ghrelin , Humans , Nutrients
9.
Clin Endocrinol (Oxf) ; 90(4): 553-561, 2019 04.
Article in English | MEDLINE | ID: mdl-30614551

ABSTRACT

OBJECTIVE: Macronutrient regulation of hyperphagia and adiposity in Prader-Willi syndrome (PWS) is poorly understood. We compared fasting and postprandial concentrations of hormones and metabolites in eight PWS children (age 9-18 years) fed, in random order, low carbohydrate, high-fat (LC, 15% carb; 65% fat; 20% protein) and low-fat, high carbohydrate (LF, 65% carb, 15% fat, 20% protein) diets matched for calories and protein. METHODS: Participants were randomized to consume either the LC or LF diet during a first hospital admission and the second diet during a subsequent admission. Blood samples were obtained after overnight fasting and 1 hour after a mixed meal. RESULTS: Relative to subjects consuming the LF diet, subjects consuming the LC diet had: lower postprandial insulin concentrations (P = 0.02); higher fasting GLP-1 AND GIP concentrations and increased postprandial GLP-1 (P < 0.02); reduced ratio of fasting ghrelin to GLP-1 (P = 0.0078); increased FFA and fatty acid oxidation, as assessed by concentrations of even-chain acylcarnitines (P < 0.001); lower fasting TG and TG/HDL ratio (P < 0.01); and higher concentrations of branch chain amino acids (P < 0.01). There were no changes in glucose, PYY, or adiponectin. CRP, AST and ALT were all higher (P < 0.01) on the LC diet. CONCLUSIONS: Increases in GLP-1 with low carbohydrate feeding and reductions in the ratio of ghrelin to GLP-1 might limit food intake and improve glycaemic control in PWS. Other potential benefits of carbohydrate restriction may include fat mobilization and oxidation and reductions in the TG/HDL ratio, a marker of insulin resistance. However, increases in CRP, AST and ALT necessitate longer-term studies of low carbohydrate efficacy and safety.


Subject(s)
Prader-Willi Syndrome/metabolism , Adiposity/physiology , Adolescent , Amino Acids/blood , Amino Acids/metabolism , Blood Glucose/metabolism , Child , Fasting/blood , Female , Glucagon-Like Peptide 1 , Humans , Insulin/blood , Insulin/metabolism , Insulin Resistance , Male , Peptide YY/blood , Peptide YY/metabolism , Prader-Willi Syndrome/blood
10.
Int J Mol Sci ; 20(13)2019 Jul 04.
Article in English | MEDLINE | ID: mdl-31277383

ABSTRACT

Autism Spectrum Disorder (ASD) is a developmental disorder characterized by social and communication deficits and repetitive behaviors. Children with ASD are also at a higher risk for developing overweight or obesity than children with typical development (TD). Childhood obesity has been associated with adverse health outcomes, including insulin resistance, diabetes, heart disease, and certain cancers. Importantly some key factors that play a mediating role in these higher rates of obesity include lifestyle factors and biological influences, as well as secondary comorbidities and medications. This review summarizes current knowledge about behavioral and lifestyle factors that could contribute to unhealthy weight gain in children with ASD, as well as the current state of knowledge of emerging risk factors such as the possible influence of sleep problems, the gut microbiome, endocrine influences and maternal metabolic disorders. We also discuss some of the clinical implications of these risk factors and areas for future research.


Subject(s)
Autism Spectrum Disorder/complications , Obesity/complications , Weight Gain , Autism Spectrum Disorder/genetics , Child , Exercise , Feeding Behavior , Humans , Obesity/genetics , Risk Factors
11.
Am J Med Genet A ; 176(11): 2309-2317, 2018 11.
Article in English | MEDLINE | ID: mdl-30216657

ABSTRACT

Dietary management is important to prevent severe obesity in individuals with Prader-Willi syndrome (PWS); however, few studies have examined dietary intake and quality in youth with PWS. Our objective was to estimate intake of essential nutrients and diet quality in youth with PWS compared to those without PWS. Three-day food records were used to estimate intake of energy, nutrients, nutrient-density, foods, and adherence to healthy eating guidelines. Data were presented as medians and interquartile ranges with Mann-Whitney U and Fisher's test used to compare between groups with p < .05 considered significant. Youth with (n = 23) and without (n = 23) PWS were similar in age and sex distribution. The PWS group had a lower energy intake (p ≤ .001), higher nutrient density (p = .003), and better adherence to guidelines (p = .007) compared to the control group. The proportion with nutrient intake from food below Estimated Average Requirement or Adequate Intake were similar between groups. Fiber, vitamin D, calcium, and potassium intake were below recommendations in 50% or more in both groups. The inclusion of supplement intake lowered the proportion below recommendations, except for fiber and potassium. Youth with PWS had a similar nutrient intake as those without PWS despite a lower energy intake, which could be attributed to higher diet quality. However, more than half of youth with PWS were at risk of inadequate fiber, vitamin D, calcium, and potassium intake. A greater emphasis on nutrient-dense foods would improve nutrient intake, but supplements may be warranted in youth with PWS who do not meet recommendations.


Subject(s)
Diet , Eating , Prader-Willi Syndrome/epidemiology , Adolescent , Beverages , Case-Control Studies , Child , Diet, Healthy , Dietary Supplements , Energy Intake , Female , Humans , Male , Micronutrients , Nutrients
12.
Pediatr Transplant ; 21(6)2017 Sep.
Article in English | MEDLINE | ID: mdl-28557140

ABSTRACT

Little has been studied regarding the diets of children following LTX. The study aim was to assess and compare dietary intake and DQ of healthy children and children post-LTX. Children and adolescents (2-18 years) post-LTX (n=27) and healthy children (n=28) were studied. Anthropometric and demographic data and two 24-hour recalls (one weekend; one weekday) were collected. Intake of added sugar, HFCS, fructose, GI, and GL was calculated. DQ was measured using three validated DQ indices: the HEI-C, the DGI-CA, and the DQI-I. Although no differences in weight-for-age z-scores were observed between groups, children post-LTX had lower height-for-age z-scores than healthy children (P<.01). With the exception of vitamin B12, no significant differences in energy and macronutrient (protein, carbohydrate, and fat), added sugar, HFCS, fructose, GI, GL, and micronutrient intakes and DQ indices (HEI-C, DGI-CA, and DQI-I) between groups were observed (P>.05). The majority of children in both groups (>40%) had low DQ scores. No significant interrelationships between dietary intake, anthropometric, and demographic were found (P>.05). Both healthy and children post-LTX consume diets with poor DQ. This has implications for risk of obesity and metabolic dysregulation, particularly in transplant populations on immunosuppressive therapies.


Subject(s)
Diet , Liver Transplantation , Nutritional Status , Adolescent , Case-Control Studies , Child , Child, Preschool , Female , Humans , Male , Nutrition Assessment , Pilot Projects , Postoperative Period , Prospective Studies
13.
J Pediatr Gastroenterol Nutr ; 65(4): 462-466, 2017 10.
Article in English | MEDLINE | ID: mdl-28452835

ABSTRACT

BACKGROUND: Vitamin D (VitD) deficiency and obesity are reaching epidemic proportions in North America, particularly in those with comorbid conditions such as diabetes or liver disease. The study objective was to determine the prevalence of suboptimal vitD status and interrelationships with anthropometric, cardiometabolic, liver, mental health, and lifestyle (sleep/screen time) parameters in an ambulatory population of children with obesity. METHODS: Children (2-18 years) attending a pediatric weight management clinic (n = 217) were retrospectively reviewed. Variables studied included anthropometric (weight, height, body mass index, waist circumference), vitD (serum 25-hydroxyvitamin D), cardiometabolic (systolic blood pressure, diastolic blood pressure, glucose, insulin, homeostasis model assessment for insulin resistance, triglyceride, high-density lipoprotein, low-density lipoprotein, total cholesterol), liver enzymes (alanine aminotransferase, gamma-glutamyl transferase), and mental health (number, diagnosis) parameters. RESULTS: Suboptimal vitD status (25-hydroxyvitamin D <75 nmol/L was present in 76% of children with obesity (12.0 ±â€Š2.9 years). Blood pressure categorized as prehypertension, stage I hypertension, and stage II hypertension was present in 14%, 25%, and 7% of children, respectively. Mental health diagnoses including anxiety, attention-deficit hyperactivity disorder, mood disorders, and learning disabilities/developmental delays occurred in 18%, 17%, 10%, and 15%, of children, respectively. Waist circumferences >100 cm were associated with lower vitD levels (58 ±â€Š18 vs 65 ±â€Š17 nmol/L; P = 0.01). VitD status ≥50 nmol/L was associated with lower insulin (15.8 [11.7-23.1] mU/L vs 21.1 [14.3-34.2] mU/L; P < 0.01) and homeostasis model assessment for insulin resistance (3.5 [2.5-4.9] vs 4.8 [3.1-6.9]; P < 0.01) values and systolic blood pressure percentiles (73.0 ±â€Š25.8 vs 80.6 ±â€Š17.0; P = 0.04). CONCLUSIONS: Children with obesity had a high prevalence of vitD deficiency, particularly those at risk for hypertension, reduced insulin sensitivity, and central obesity.


Subject(s)
Hepatic Insufficiency/etiology , Hypertension/etiology , Insulin Resistance , Mental Disorders/etiology , Pediatric Obesity/complications , Vitamin D Deficiency/etiology , Adolescent , Biomarkers/metabolism , Child , Child, Preschool , Female , Hepatic Insufficiency/diagnosis , Hepatic Insufficiency/epidemiology , Humans , Hypertension/diagnosis , Hypertension/epidemiology , Male , Mental Disorders/diagnosis , Mental Disorders/epidemiology , Pediatric Obesity/metabolism , Pediatric Obesity/psychology , Pediatric Obesity/therapy , Prevalence , Retrospective Studies , Risk Factors , Vitamin D Deficiency/diagnosis , Vitamin D Deficiency/epidemiology , Waist Circumference , Weight Reduction Programs
14.
Int J Mol Sci ; 18(3)2017 Feb 28.
Article in English | MEDLINE | ID: mdl-28264487

ABSTRACT

Prader-Willi syndrome (PWS), a rare genetic disorder caused by the lack of expression of paternal genes from chromosome 15q11-13, has been investigated for autism spectrum disorder (ASD) symptomatology in various studies. However, previous findings have been variable, and no studies investigating ASD symptomatology in PWS have exclusively studied children. We aimed to characterize social communication functioning and other ASD-related symptoms in children with PWS, and assessed agreement across measures and rates of ASD diagnosis. Measures included the Autism Diagnostic Observation Schedule-2 (ADOS-2), the Social Communication Questionnaire (SCQ), Social Responsiveness Scale-2 (SRS-2), Social Skills Improvement System-Rating Scales (SSIS-RS), and the Vineland Adaptive Behavioral Scales-II (VABS-II). General adaptive and intellectual skills were also assessed. Clinical best estimate (CBE) diagnosis was determined by an experienced developmental pediatrician, based on history and review of all available study measures, and taking into account overall developmental level. Participants included 10 children with PWS, aged 3 to 12 years. Three of the 10 children were male and genetic subtypes were two deletion (DEL) and eight uniparental disomy (UPD) (with a total of 6 female UPD cases). Although 8 of the 10 children exceeded cut-offs on at least one of the ASD assessments, agreement between parent questionnaires (SCQ, SRS-2, SSIS-RS) and observational assessment (ADOS-2) was very poor. None of the children were assigned a CBE diagnosis of ASD, with the caveat that the risk may have been lower because of the predominance of girls in the sample. The lack of agreement between the assessments emphasizes the complexity of interpreting ASD symptom measures in children with PWS.


Subject(s)
Autism Spectrum Disorder/diagnosis , Phenotype , Prader-Willi Syndrome/diagnosis , Prader-Willi Syndrome/psychology , Child , Child, Preschool , Female , Humans , Male , Psychological Tests , Reproducibility of Results
15.
Appetite ; 99: 17-24, 2016 Apr 01.
Article in English | MEDLINE | ID: mdl-26713776

ABSTRACT

Hyperphagia and obsessive preoccupation with food are hallmark characteristics of Prader-Willi Syndrome (PWS). Although hyperphagia in PWS is linked to hypothalamic dysfunction, the underlying mechanisms behind this problem are poorly understood. Moreover, our understanding of how chemosensory perceptions and food choice/preferences relate to hyperphagia in individuals with PWS is very limited. This narrative review synthesizes studies that assessed chemosensory perceptions, food choices and food-related behaviours in PWS individuals and highlights knowledge gaps in research for further exploration. Twenty seven publications from relevant databases met inclusion criteria and were organized thematically by study technique in the review. Results suggested that PWS individuals have consistent preferences for sweet tastes and in most studies have exhibited a preference for calorie-dense foods over lower calorie foods. No firm conclusions were drawn concerning the chemosensory perceptions of PWS individuals and their influence on food preferences or choices; chemosensation among PWS individuals is an understudied topic. Current evidence suggests that eating behaviour in PWS is a complex phenomenon that involves a dysfunctional satiation and not excessive hunger. Food preferences, choices, and related behaviours and the impact of these on obesity management in those with PWS remain poorly understood and require further study using validated tools and methodologies.


Subject(s)
Feeding Behavior , Food Preferences , Prader-Willi Syndrome/physiopathology , Taste Perception , Choice Behavior , Databases, Factual , Humans , Hunger , Hyperphagia/physiopathology , Prader-Willi Syndrome/psychology , Satiation
16.
Clin Endocrinol (Oxf) ; 83(6): 797-805, 2015 Dec.
Article in English | MEDLINE | ID: mdl-25736874

ABSTRACT

OBJECTIVES: To identify metabolic factors controlling appetite and insulin sensitivity in PWS and assess effects of GH treatment. METHODS: We compared amino acids, fatty acids and acylcarnitines in GH-treated and untreated PWS children and obese and lean controls to identify biomarkers associated with ghrelin, peptide YY and markers of insulin sensitivity (adiponectin and HOMA-IR). RESULTS: Compared with obese controls (OC), children with PWS had fasting hyperghrelinaemia, hyperadiponectinaemia, hypoinsulinaemia and increased ghrelin/PYY. Hyperghrelinaemia, hyperadiponectinaemia and hypoinsulinaemia were more striking in PWS females than males, and decreases in BCAA were detected only in PWS females. GH-treated PWS subjects had lower leptin and higher IGF-1 and adiponectin than untreated subjects; fasting ghrelin, PYY and insulin levels were comparable. Ghrelin correlated inversely with BCAA in PWS but not OC. Adiponectin correlated negatively with BMIz and HOMA-IR in PWS; in contrast, adiponectin correlated more strongly with BCAA than BMIz or HOMA-IR in OC. CONCLUSIONS: BCAA levels were lower in PWS females than OC females and correlated inversely with ghrelin. Low BCAA in PWS females may promote hyperghrelinaemia and hyperphagia, while hyperadiponectinaemia may maintain insulin sensitivity despite excess weight gain. GH treatment may reduce leptin and increase adiponectin, but does not affect fasting ghrelin or PYY.


Subject(s)
Growth Hormone/therapeutic use , Obesity/blood , Obesity/complications , Prader-Willi Syndrome/blood , Prader-Willi Syndrome/drug therapy , Adiponectin/blood , Adolescent , Child , Fasting/blood , Female , Ghrelin/blood , Humans , Insulin/blood , Leptin/blood , Male , Peptide YY/blood , Prader-Willi Syndrome/etiology , Prader-Willi Syndrome/metabolism
17.
Am J Med Genet A ; 167A(12): 2936-44, 2015 Dec.
Article in English | MEDLINE | ID: mdl-26331980

ABSTRACT

Prader-Willi syndrome (PWS) is a rare genetic disorder that results from lack of expression of paternally-derived genes on chromosome 15q11-13; caused by a deletion (DEL), uniparental disomy (UPD), or a rare imprinting center defect. PWS is associated with a distinct behavioral phenotype that in some respects overlaps with autism spectrum disorder (ASD), a neurodevelopmental disorder characterized by restricted or repetitive behaviors (RRBs) and social-communication impairment. The goal of this review was to (i) review published literature investigating core ASD symptoms in PWS and (ii) provide a prevalence estimate of ASD in PWS. Two independent reviewers searched Medline, CINAHL, PsychINFO, Embase, and Web of Science to find studies that answered the research questions. Individuals with PWS demonstrate significant levels of RRBs and social-communication impairment, in some reports reaching similar levels to those of non-PWS ASD comparison groups. Individuals with UPD had more social-communication impairment than those with DEL. Of 786 PWS participants, 210 (26.7%) were reported as meeting criteria for ASD, either based on clinical diagnosis or by exceeding clinical cut-points on relevant ASD symptom measures. In studies that distinguished genetic subtypes, rates of ASD were higher in individuals with PWS with UPD (67 of 190; 35.3%) than those with DEL (47 of 254; 18.5%). Published data on the association of PWS and ASD to date are limited to sample means of 8 years of age and older. Further research is needed to identify early markers of ASD in PWS children, to support earlier diagnosis and intervention for this important comorbidity.


Subject(s)
Autism Spectrum Disorder/etiology , Prader-Willi Syndrome/psychology , Autism Spectrum Disorder/epidemiology , Autism Spectrum Disorder/genetics , Humans , Prader-Willi Syndrome/complications , Sequence Deletion , Social Communication Disorder/genetics , Uniparental Disomy
18.
Pediatr Exerc Sci ; 27(4): 525-34, 2015 Nov.
Article in English | MEDLINE | ID: mdl-26181653

ABSTRACT

Prader-Willi Syndrome (PWS), the best characterized form of syndromic obesity, presents with abnormally high fat mass. In children, obesity presents with low-grade systemic inflammation. This study evaluated if PWS and/or nonsyndromic obesity affected cytokine responses to intermittent aerobic exercise in children. Eleven children with PWS (11 ± 2 y, 45.4 ± 9.5% body fat), 12 children with obesity (OB) (9 ± 1 y, 39.9 ± 6.8% body fat), and 12 lean (LN) children (9 ± 1 y, 17.5 ± 4.6% body fat) participated. Children completed 10 2-min cycling bouts of vigorous intensity, separated by 1-min rest. Blood samples were collected preexercise (PRE), immediately postexercise (IP), and 15, 30, and 60 min into recovery to analyze possible changes in cytokines. In all groups, IL-6 and IL-8 concentrations were greater during recovery compared with PRE. PWS and OB exhibited higher IL-6 area under the curve (AUC) than LN (p < .01 for both). PWS demonstrated higher IL-8 AUC than LN (p < .04). IL-10, TNF-α, and IFN-γ did not change with exercise (p > .05 for all). Results indicate that children with PWS respond with increased Il-6 and IL-8 concentrations to acute exercise similarly to controls. Excess adiposity and epigenetic modifications may explain the greater integrated IL-6 and IL-8 responses in PWS compared with controls.


Subject(s)
Cytokines/blood , Exercise/physiology , Prader-Willi Syndrome/blood , Thinness/blood , Adiposity , Adolescent , Area Under Curve , Child , Exercise Test , Female , Humans , Interferon-gamma/blood , Interleukin-10/blood , Interleukin-6/blood , Interleukin-8/blood , Male , Pediatric Obesity/blood , Physical Exertion/physiology , Tumor Necrosis Factor-alpha/blood
20.
Child Obes ; 20(7): 526-535, 2024 Oct.
Article in English | MEDLINE | ID: mdl-38621159

ABSTRACT

Background: The built environment can impact health outcomes. Our purpose was to examine relationships between built environment variables related to physical activity and excess weight in preschoolers. Methods: In this retrospective, population-level study of 4- to 6-year-olds, anthropometric measurements were taken between 2009 and 2017 in Calgary and Edmonton, Alberta, Canada. Based on BMI z-scores (BMIz), children were classified as normal weight (-2 ≤ BMIz <1) or excess weight (BMIz ≥1; overweight and obesity). Physical activity-related built environment variables were calculated (distances to nearest playground, major park, school; street intersection density; number of playgrounds and major parks within an 800 m buffer zone). Binomial logistic regression models estimated associations between physical activity-related built environment variables and excess weight. Results: Our analysis included 140,368 participants (females: n = 69,454; Calgary: n = 84,101). For Calgary, adjusted odds ratios (aORs) showed the odds of excess weight increased 1% for every 100-intersection increase [1.010 (1.006-1.015); p < 0.0001] and 13.6% when there were ≥4 playgrounds (vs. 0 or 1) within an 800 m buffer zone [1.136 (1.037-1.243); p = 0.0059]. For Edmonton, aORs revealed lower odds of excess weight for every 100 m increase in distances between residences to nearest major park [0.991 (0.986-0.996); p = 0.0005] and school [0.992 (0.990-0.995); p < 0.0001]. The odds of excess weight decreased as the number of major parks within the 800 m buffer zone increased from 0 to 1 [0.943 (0.896-0.992); p = 0.023] and from 0 to ≥3 [0.879 (0.773-0.999); p = 0.048]. Conclusion: The physical activity-related built environment was associated with excess weight in preschoolers, although relationships varied between cities that differed demographically and geographically.


Subject(s)
Built Environment , Exercise , Pediatric Obesity , Humans , Child, Preschool , Retrospective Studies , Female , Male , Built Environment/statistics & numerical data , Pediatric Obesity/epidemiology , Alberta/epidemiology , Child , Residence Characteristics/statistics & numerical data , Environment Design/statistics & numerical data , Body Mass Index , Cohort Studies
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