Mechanisms of obesity in children and adults with phenylketonuria on contemporary treatment

Alghamdi, Noweir Ahmad; Alfheeaid, Hani A.; Cochrane, Barbara; Adam, Sarah; Galloway, Peter; Cozens, Alison; Preston, Tom; Malkova, Dalia; Gerasimidis, Konstantinos

doi:10.1016/j.clnesp.2021.10.012

Cited by 17 publications

(40 citation statements)

References 11 publications

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“…Only one study evaluated the agreement between the REE measured using IC and prediction equations in phenylketonurics. As a result, Quirk et al 15 found that the equations (FAO/WHO/UNU – weight; FAO/WHO/UNU – weight and height; Harris Benedict, Molnár, Schofield – weight; and Schofield – weight and height) underestimated the REE measured using IC, which corroborates our results. However, they evaluated only American female adolescents living with PKU and investigated only six prediction equations.…”

Section: Discussionsupporting

confidence: 91%

“…Both results are higher than those found in this study. However, Alghamdi et al 15 found an REE of 1266 ± 597 kcal (23.9 ± 9.0 kcal/kg) in adolescents (n = 6) in the United Kingdom, a value, in average kcal/kg, lower than that found by us.…”

Section: Discussioncontrasting

confidence: 80%

“…Quirk et al 14 corroborated with Allen et al 13 in the sense that available equations overestimate the REE of adolescent girls living with PKU. Alghamdi et al 15 found no differences in the REE of 6 adolescents and 10 adults living with PKU compared to healthy controls.…”

Section: Introductionmentioning

confidence: 95%

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Accuracy of equations for estimating resting energy expenditure in children and adolescents living with phenylketonuria

Mezzomo

Dias

Vilela

et al. 2023

J Human Nutrition Diet

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BackgroundMeasuring resting energy expenditure (REE) in individuals living with phenylketonuria (PKU) using indirect calorimetry (IC) is unusual in healthcare facilities because it requires specific protocols and expensive equipment. Considering that determining REE is crucial for devising nutritional strategies for the management of PKU, the aim of this study was to identify the predictive equations that provide the best estimates of REE in children and adolescents living with PKU and to propose a predictive equation for determining REE in this population.MethodsAn REE concordance study was conducted with children and adolescents living with PKU. Anthropometric and body composition assessments using bioimpedance and REE assessment using IC were performed. The results were compared to 29 predictive equations.ResultsFifty‐four children and adolescents were evaluated. The REE obtained using IC differed from all estimated REE, except Henry's equation for male children (p = 0.058). Only this equation showed good agreement (0.900) with IC. Eight variables were associated with the REE obtained using IC with emphasis on fat‐free mass (kg) (r = 0.786), weight (r = 0.775), height (r = 0.759) and blood phenylalanine (r = 0.503). With these variables, three REE equations were suggested, with R2 = 0.660, 0.635 and 0.618, respectively, and the third equation, which involves weight and height, showed adequate sample size for a statistical power of 0.942.ConclusionMost equations, not specific for individuals living with PKU, overestimate the REE of this population. We propose a predictive equation for assessing REE for children and adolescents living with PKU to be used in settings where IC is not available.

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Section: Discussionsupporting

confidence: 91%

Section: Discussioncontrasting

confidence: 80%

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Accuracy of equations for estimating resting energy expenditure in children and adolescents living with phenylketonuria

Mezzomo

Dias

Vilela

et al. 2023

J Human Nutrition Diet

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“…Confounding conditions included congenital malformations, brain injury, seizure, technology dependence, cancer, eosinophilic esophagitis or milk protein intolerance, malabsorption syndrome or lactose intolerance, and hypothyroidism or growth hormone deficiency, and other conditions that grouped together conditions with fewer than 100 individuals into 1 category (eTable 2 in Supplement 1). Fisher exact test showed pairwise correlation coefficients to be less than 0.7 for all conditions; hence, no further action was required to account for multicollinearity (eTable 3 in Supplement 1). We performed sensitivity analysis by adjusting for 2 additional conditions—eating disorders and psychiatric conditions—along with 9 conditions used in the fully adjusted model and presented the results of Fisher exact test (eTable 4 in Supplement 1) and marginal cost estimates (eTable 5 in Supplement 1).…”

Section: Methodsmentioning

confidence: 99%

“…in Supplement 1). [32][33][34][35][36][37][38][39][40][41][42][43][44] Fisher exact test showed pairwise correlation coefficients to be less than 0.7 for all conditions; hence, no further action was required to account for multicollinearity (eTable 3 in Supplement 1). We performed sensitivity analysis by adjusting for 2 additional conditions-eating disorders and psychiatric conditions-along with 9 conditions used in the fully adjusted model and presented the results of Fisher exact test (eTable 4 in Supplement 1) and marginal cost estimates (eTable 5 in Supplement 1).…”

Section: Key Pointsmentioning

confidence: 99%

Body Mass Index and Associated Medical Expenditures in the US Among Privately Insured Individuals Aged 2 to 19 Years in 2018

et al. 2023

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ImportanceNearly 40% of US youth aged 2 to 19 years do not have a body mass index (BMI) in the healthy weight category. However, there are no recent estimates for BMI-associated expenditures using clinical or claims data.ObjectiveTo estimate medical expenditures among US youth across all BMI categories along with sex and age groups.Design, Setting, ParticipantsThis cross-sectional study used IQVIA’s ambulatory electronic medical records (AEMR) data set linked with IQVIA’s PharMetrics Plus Claims database from January 2018 through December 2018. Analysis was performed from March 25, 2022, through June 20, 2022. It included a convenience sample of a geographically diverse patient population from AEMR and PharMetrics Plus. The study sample included privately insured individuals with a BMI measurement in 2018 and excluded patients with pregnancy-related visits.ExposureBMI categories.Main Outcomes and MeasuresTotal medical expenditures were estimated using generalized linear model regression with γ distribution and log-link function. For out-of-pocket (OOP) expenditures, a 2-part model was used that included logistic regression to estimate the probability of positive expenditures followed by generalized linear model. Estimates were shown with and without accounting for sex, race and ethnicity, payer type, geographic region, age interacted with sex and BMI categories, and confounding conditions.ResultsThe sample included 205 876 individuals aged 2 to 19 years; 104 066 were male (50.5%) and the median age was 12 years. Compared with those with healthy weight, total and OOP expenditures were higher for all other BMI categories. Differences in total expenditures were highest for those with severe obesity ($909; 95% CI, $600-$1218) followed by underweight ($671; 95% CI, $286-$1055) compared with healthy weight. Differences in OOP expenditures were highest for those with severe obesity ($121; 95% CI, $86-$155) followed by underweight ($117; 95% CI, $78-$157) compared with healthy weight. Having underweight was associated with higher total expenditures at ages 2 to 5 years and 6 to 11 years by $679 (95% CI, $228-$1129) and $1166 (95% CI, $632-$1700), respectively; having severe obesity was associated with higher total expenditures at ages 2 to 5 years, 6 to 11 years, and 12 to 17 years by $1035 (95% CI, $208-$1863), $821 (95% CI, $414-$1227), and $1088 (95% CI, $594-$1582), respectively.Conclusions and RelevanceThe study team found medical expenditures to be higher for all BMI categories when compared with those with healthy weight. These findings may indicate potential economic value of interventions or treatments aimed at reducing BMI-associated health risks.

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Adults with early diagnosis of phenylketonuria have higher resting energy expenditure than adults with late diagnosis

Mezzomo¹,

Dias²,

Pereira³

2023

Clinical Nutrition ESPEN

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Mechanisms of obesity in children and adults with phenylketonuria on contemporary treatment

Cited by 17 publications

References 11 publications

Accuracy of equations for estimating resting energy expenditure in children and adolescents living with phenylketonuria

Accuracy of equations for estimating resting energy expenditure in children and adolescents living with phenylketonuria

Body Mass Index and Associated Medical Expenditures in the US Among Privately Insured Individuals Aged 2 to 19 Years in 2018

Adults with early diagnosis of phenylketonuria have higher resting energy expenditure than adults with late diagnosis

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