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  1. Article ; Online: Use of Compartmental Modeling and Retinol Isotope Dilution to Determine Vitamin A Stores in Young People with Sickle Cell Disease Before and After Vitamin A Supplementation.

    Ford, Jennifer Lynn / Green, Michael H / Brownell, Jefferson N / Green, Joanne Balmer / Oxley, Anthony / Lietz, Georg / Schall, Joan I / Stallings, Virginia A

    The Journal of nutrition

    2023  Volume 153, Issue 9, Page(s) 2762–2771

    Abstract: Background: Suboptimal plasma retinol concentrations have been documented in US children with sickle cell disease (SCD) hemoglobin SS type (SCD-HbSS), but little is known about vitamin A kinetics and stores in SCD.: Objectives: The objectives were to ...

    Abstract Background: Suboptimal plasma retinol concentrations have been documented in US children with sickle cell disease (SCD) hemoglobin SS type (SCD-HbSS), but little is known about vitamin A kinetics and stores in SCD.
    Objectives: The objectives were to quantify vitamin A total body stores (TBS) and whole-body retinol kinetics in young people with SCD-HbSS and use retinol isotope dilution (RID) to predict TBS in SCD-HbSS and healthy peers as well as after vitamin A supplementation in SCD-HbSS subjects.
    Methods: Composite plasma [
    Results: Model-predicted group mean TBS for subjects with SCD-HbSS was 428 μmol, equivalent to ∼11 mo of stored vitamin A; vitamin A disposal rate was 1.3 μmol/d. Model-predicted TBS was similar to that predicted by RID at 3 d postdosing (mean, 389 μmol; ∼0.3 μmol/g liver); TBS predictions at 3 compared with 28 d were not significantly different. Mean TBS in healthy peers was similar (406 μmol). RID-predicted TBS for subjects with SCD-HbSS was not significantly affected by vitamin A supplementation at either dose.
    Conclusions: Despite differences in plasma retinol concentrations, TBS was the same in subjects with SCD-HbSS compared with healthy peers. Because 56 d of vitamin A supplementation at levels 1.2 to 2.6 times the Recommended Dietary Allowance did not increase TBS in these subjects with SCD-HbSS, further work will be needed to understand the effects of SCD on retinol metabolism. This trial was registered as NCT03632876 at clinicaltrials.gov.
    MeSH term(s) Child ; Humans ; Adolescent ; Vitamin A ; Vitamin A Deficiency ; Anemia, Sickle Cell ; Dietary Supplements ; Isotopes
    Chemical Substances Vitamin A (11103-57-4) ; Isotopes
    Language English
    Publishing date 2023-07-17
    Publishing country United States
    Document type Journal Article ; Research Support, Non-U.S. Gov't ; Research Support, N.I.H., Extramural
    ZDB-ID 218373-0
    ISSN 1541-6100 ; 0022-3166
    ISSN (online) 1541-6100
    ISSN 0022-3166
    DOI 10.1016/j.tjnut.2023.07.004
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    Uc I Zs.205: Show issues Location:
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  2. Article ; Online: Pancreatic Function in Chronic Pancreatitis: A Cohort Study Comparing 3 Methods of Detecting Fat Malabsorption and the Impact of Short-term Pancreatic Enzyme Replacement Therapy.

    Brownell, Jefferson N / Schall, Joan I / Stallings, Virginia A

    Pancreas

    2019  Volume 48, Issue 8, Page(s) 1068–1078

    Abstract: Objectives: Reliable pancreatic function tests in patients with chronic pancreatitis (CP) are needed. This cohort study identified malabsorption in people with CP compared with healthy people and then investigated short-term pancreatic enzyme ... ...

    Abstract Objectives: Reliable pancreatic function tests in patients with chronic pancreatitis (CP) are needed. This cohort study identified malabsorption in people with CP compared with healthy people and then investigated short-term pancreatic enzyme replacement therapy (PERT) and fat malabsorption, nutritional status, and quality of life (QOL).
    Methods: Subjects with CP were evaluated before and after PERT and compared with the healthy cohort using coefficient of fat absorption (CFA), stool bomb calorimetry, and the malabsorption blood test (MBT). Anthropometrics, micronutrients, and QOL data were collected. Group means at baseline and after PERT were analyzed.
    Results: The 24 subjects with CP had greater stool energy loss (5668 cal/g [standard deviation {SD}, 753] vs 5152 cal/g [SD, 418], P < 0.01), reduced triglyceride absorption (MBT, 8.3 mg·h/dL [SD, 4.3] vs 17.7 mg·h/dL [SD, 10.3], P < 0.001), lower fat intake, and poorer QOL. Differences in CFA were not significant (90.9% [SD, 12.8] vs 95.4% [SD, 9.3]). After PERT, triglyceride absorption (Δ = 1.7 [SD, 3], P < 0.05) and QOL increased.
    Conclusions: The MBT detected changes in triglyceride absorption in the absence of CFA changes. The MBT may be helpful in guiding PERT initiation in patients with CP before significant morbidity.
    MeSH term(s) Adult ; Cohort Studies ; Enzyme Replacement Therapy/methods ; Exocrine Pancreatic Insufficiency/diagnosis ; Exocrine Pancreatic Insufficiency/physiopathology ; Exocrine Pancreatic Insufficiency/therapy ; Fats/metabolism ; Female ; Humans ; Malabsorption Syndromes/diagnosis ; Malabsorption Syndromes/physiopathology ; Malabsorption Syndromes/therapy ; Male ; Middle Aged ; Nutritional Status ; Outcome Assessment, Health Care/methods ; Outcome Assessment, Health Care/statistics & numerical data ; Pancreas/pathology ; Pancreas/physiopathology ; Pancreatic Function Tests/methods ; Pancreatitis, Chronic/diagnosis ; Pancreatitis, Chronic/physiopathology ; Pancreatitis, Chronic/therapy ; Pancrelipase/metabolism ; Pancrelipase/therapeutic use ; Quality of Life ; Triglycerides/metabolism
    Chemical Substances Fats ; Triglycerides ; Pancrelipase (53608-75-6)
    Language English
    Publishing date 2019-08-30
    Publishing country United States
    Document type Journal Article ; Research Support, N.I.H., Extramural ; Research Support, Non-U.S. Gov't
    ZDB-ID 632831-3
    ISSN 1536-4828 ; 0885-3177
    ISSN (online) 1536-4828
    ISSN 0885-3177
    DOI 10.1097/MPA.0000000000001381
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    Zs.A 2160: Show issues Location:
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  3. Article ; Online: Measures of Dietary Fat and Energy Absorption in Healthy Adults.

    Bashaw, Hillary / Brownell, Jefferson N / Schall, Joan I / Stallings, Virginia A

    Pancreas

    2020  Volume 49, Issue 6, Page(s) 845–854

    Abstract: Objectives: Existing reference ranges for stool fat and energy absorption were developed using subjects in controlled environments on precise diets. This study measured energy and fat absorption in healthy, community-dwelling adults eating a moderate-to- ...

    Abstract Objectives: Existing reference ranges for stool fat and energy absorption were developed using subjects in controlled environments on precise diets. This study measured energy and fat absorption in healthy, community-dwelling adults eating a moderate-to-high fat American diet via stool- and serum-based methods.
    Methods: This was a secondary analysis of healthy subjects recruited as the comparison group in a chronic pancreatitis study. Subjects recorded dietary intake and collected stool over 3-day periods. Stool was analyzed for fat content using the coefficient of fat absorption and for energy content using bomb calorimetry. The malabsorption blood test (MBT) was used to measure dietary fat absorption.
    Results: Nineteen subjects had mean daily stool measures of 143 g wet weight, 4.1 g of fat, and 178 kcal. The mean coefficients of fat and energy absorption were 96% and 93%, respectively. The mean MBT area under the curve cut-point was greater than 8 mg·h/dL.
    Conclusions: This study confirms the historical reference range for the coefficient of fat absorption in contemporary healthy, community-dwelling adults on a moderate-to-high fat diet. The study contributes to the development of reference range values for multiple bomb calorimetry-based outcomes of stool energy losses and to the serum-based MBT as a promising method for measuring fat absorption.
    MeSH term(s) Adult ; Calorimetry/methods ; Cohort Studies ; Dietary Fats/metabolism ; Energy Intake ; Feces/chemistry ; Female ; Healthy Aging/metabolism ; Humans ; Independent Living/statistics & numerical data ; Intestinal Absorption ; Male ; Middle Aged ; Pancreatitis, Chronic/diagnosis ; Pancreatitis, Chronic/metabolism
    Chemical Substances Dietary Fats
    Language English
    Publishing date 2020-06-18
    Publishing country United States
    Document type Journal Article ; Research Support, N.I.H., Extramural ; Research Support, Non-U.S. Gov't
    ZDB-ID 632831-3
    ISSN 1536-4828 ; 0885-3177
    ISSN (online) 1536-4828
    ISSN 0885-3177
    DOI 10.1097/MPA.0000000000001587
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  4. Article ; Online: Correction: Improved residual fat malabsorption and growth in children with cystic fibrosis treated with a novel oral structured lipid supplement: A randomized controlled trial.

    Stallings, Virginia A / Tindall, Alyssa M / Mascarenhas, Maria R / Maqbool, Asim / Schall, Joan I

    PloS one

    2020  Volume 15, Issue 9, Page(s) e0239642

    Abstract: This corrects the article DOI: 10.1371/journal.pone.0232685.]. ...

    Abstract [This corrects the article DOI: 10.1371/journal.pone.0232685.].
    Language English
    Publishing date 2020-09-17
    Publishing country United States
    Document type Journal Article ; Published Erratum
    ISSN 1932-6203
    ISSN (online) 1932-6203
    DOI 10.1371/journal.pone.0239642
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  5. Article ; Online: Vitamin D Supplementation Improves Health-Related Quality of Life and Physical Performance in Children with Sickle Cell Disease and in Healthy Children.

    Dougherty, Kelly A / Schall, Joan I / Bertolaso, Chiara / Smith-Whitley, Kim / Stallings, Virginia A

    Journal of pediatric health care : official publication of National Association of Pediatric Nurse Associates & Practitioners

    2020  Volume 34, Issue 5, Page(s) 424–434

    Abstract: Introduction: No study determined if vitamin D supplementation improves health-related quality of life (HRQL) using pediatric Patient-Reported Outcomes Measurement Information System or physical functioning in type SS sickle cell disease (HbSS).: ... ...

    Abstract Introduction: No study determined if vitamin D supplementation improves health-related quality of life (HRQL) using pediatric Patient-Reported Outcomes Measurement Information System or physical functioning in type SS sickle cell disease (HbSS).
    Method: Subjects with HbSS (n = 21) and healthy subjects (n = 23) were randomized to daily oral doses (4,000 vs. 7,000 IU) of cholecalciferol (vitamin D
    Results: In subjects with HbSS, significant reductions in pain, fatigue, and depressive symptoms and improved upper-extremity function were observed. In healthy subjects, significant reductions in fatigue and improved upper-extremity function were observed. Significant improvements in peak power and dorsiflexion isometric maximal voluntary contraction torques were observed in both groups. In subjects with HbSS, improved plantar flexion isometric maximal voluntary contraction torques were observed. Both groups saw significant improvement in their total Bruininks-Oseretsky Test of Motor Proficiency score.
    Discussion: Daily high-dose vitamin D supplementation for African American children with and without HbSS improved HRQL and physical performance.
    MeSH term(s) Adolescent ; Anemia, Sickle Cell/drug therapy ; Child ; Dietary Supplements ; Female ; Humans ; Male ; Physical Functional Performance ; Quality of Life ; Vitamin D/therapeutic use ; Vitamin D Deficiency/drug therapy
    Chemical Substances Vitamin D (1406-16-2)
    Language English
    Publishing date 2020-06-05
    Publishing country United States
    Document type Journal Article ; Randomized Controlled Trial ; Research Support, N.I.H., Extramural ; Research Support, Non-U.S. Gov't
    ZDB-ID 1036356-7
    ISSN 1532-656X ; 0891-5245
    ISSN (online) 1532-656X
    ISSN 0891-5245
    DOI 10.1016/j.pedhc.2020.04.007
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    Zs.A 2641: Show issues Location:
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  6. Article ; Online: Improved residual fat malabsorption and growth in children with cystic fibrosis treated with a novel oral structured lipid supplement: A randomized controlled trial.

    Stallings, Virginia A / Tindall, Alyssa M / Mascarenhas, Maria R / Maqbool, Asim / Schall, Joan I

    PloS one

    2020  Volume 15, Issue 5, Page(s) e0232685

    Abstract: Background: In the primary analysis of a 12-month double-blind randomized active placebo-controlled trial, treatment of children with cystic fibrosis (CF) and pancreatic insufficiency (PI) with a readily absorbable structured lipid (Encala™, Envara ... ...

    Abstract Background: In the primary analysis of a 12-month double-blind randomized active placebo-controlled trial, treatment of children with cystic fibrosis (CF) and pancreatic insufficiency (PI) with a readily absorbable structured lipid (Encala™, Envara Health, Wayne, PA) was safe, well-tolerated and improved dietary fat absorption (stool coefficient of fat absorption [CFA]), growth, and plasma fatty acids (FA).
    Objective: To determine if the Encala™ treatment effect varied by severity of baseline fat malabsorption.
    Methods: Subjects (n = 66, 10.5±3.0 yrs, 39% female) with baseline CFA who completed a three-month treatment with Encala™ or a calorie and macronutrient-matched placebo were included in this subgroup analysis. Subjects were categorized by median baseline CFA: low CFA (<88%) and high CFA (≥88%). At baseline and 3-month evaluations, CFA (72-hour stool, weighed food record) and height (HAZ), weight (WAZ) and BMI (BMIZ) Z-scores were calculated. Fasting plasma fatty acid (FA) concentrations were also measured.
    Results: Subjects in the low CFA subgroup had significantly improved CFA (+7.5±7.2%, mean 86.3±6.7, p = 0.002), and reduced stool fat loss (-5.7±7.2 g/24 hours) following three months of EncalaTM treatment. These subjects also had increased plasma linoleic acid (+20%), α-linolenic acid (+56%), and total FA (+20%) (p≤0.005 for all) concentrations and improvements in HAZ (0.06±0.08), WAZ (0.17±0.16), and BMIZ (0.20±0.25) (p≤0.002 for all). CFA and FA were unchanged with placebo in the low CFA group, with some WAZ increases (0.14±0.24, p = 0.02). High CFA subjects (both placebo and Encala™ groups) had improvements in WAZ and some FA.
    Conclusions: Subjects with CF, PI and more severe fat malabsorption experienced greater improvements in CFA, FA and growth after three months of Encala™ treatment. Encala™ was safe, well-tolerated and efficacious in patients with CF and PI with residual fat malabsorption and improved dietary energy absorption, weight gain and FA status in this at-risk group.
    MeSH term(s) Administration, Oral ; Child ; Cystic Fibrosis/complications ; Cystic Fibrosis/metabolism ; Cystic Fibrosis/therapy ; Dietary Fats/metabolism ; Dietary Supplements/analysis ; Double-Blind Method ; Exocrine Pancreatic Insufficiency/complications ; Exocrine Pancreatic Insufficiency/metabolism ; Exocrine Pancreatic Insufficiency/therapy ; Female ; Humans ; Lipids/administration & dosage ; Lipids/therapeutic use ; Malabsorption Syndromes/complications ; Malabsorption Syndromes/metabolism ; Malabsorption Syndromes/therapy ; Male ; Placebo Effect
    Chemical Substances Dietary Fats ; Lipids
    Language English
    Publishing date 2020-05-08
    Publishing country United States
    Document type Journal Article ; Randomized Controlled Trial ; Research Support, N.I.H., Extramural
    ISSN 1932-6203
    ISSN (online) 1932-6203
    DOI 10.1371/journal.pone.0232685
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  7. Article ; Online: Genetic Variants, Fat Malabsorption, and Ancestral Background in a Small Chronic Pancreatitis Cohort.

    Brownell, Jefferson N / Haupt, Mark / Orlova, Ekaterina / Schall, Joan I / Stallings, Virginia A

    Pancreas

    2020  Volume 49, Issue 8, Page(s) e76–e78

    MeSH term(s) Adult ; African Americans/genetics ; African Americans/statistics & numerical data ; Aged ; Asian Americans/genetics ; Asian Americans/statistics & numerical data ; Cohort Studies ; Dietary Fats/administration & dosage ; Dietary Fats/metabolism ; Dietary Fats/pharmacokinetics ; Female ; Genetic Predisposition to Disease/genetics ; Hispanic or Latino/genetics ; Hispanic or Latino/statistics & numerical data ; Humans ; Intestinal Absorption ; Male ; Middle Aged ; Mutation ; Pancreatitis, Chronic/ethnology ; Pancreatitis, Chronic/genetics ; Pancreatitis, Chronic/metabolism
    Chemical Substances Dietary Fats
    Language English
    Publishing date 2020-08-24
    Publishing country United States
    Document type Letter ; Research Support, N.I.H., Extramural ; Research Support, Non-U.S. Gov't
    ZDB-ID 632831-3
    ISSN 1536-4828 ; 0885-3177
    ISSN (online) 1536-4828
    ISSN 0885-3177
    DOI 10.1097/MPA.0000000000001623
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  8. Article ; Online: Endocrine-sensitive physical endpoints in newborns: ranges and predictors.

    Shah, Rachana / Alshaikh, Belal / Schall, Joan I / Kelly, Andrea / Ford, Eileen / Zemel, Babette S / Umbach, David M / Adgent, Margaret / Stallings, Virginia A

    Pediatric research

    2020  Volume 89, Issue 3, Page(s) 660–666

    Abstract: Background: In neonates, endocrine-sensitive physical endpoints, including breast and reproductive tissues, may reflect effects of fetal environmental exposure. Studies using standardized measurement techniques that describe demographic and clinical ... ...

    Abstract Background: In neonates, endocrine-sensitive physical endpoints, including breast and reproductive tissues, may reflect effects of fetal environmental exposure. Studies using standardized measurement techniques that describe demographic and clinical variability in these endpoints are lacking.
    Methods: Three hundred and eighty-eight healthy term newborns <3 days old were evaluated, 69% African American and 25% White. Measures included breast bud diameter, anogenital distance (AGD), stretched penile length (SPL), and testicular volume (TV).
    Results: Breast buds were larger in females than males bilaterally (right: 13.0 ± 4.0 vs. 12.0 ± 4.0 mm, p = 0.008; left: 13.0 ± 4.0 vs. 11.0 ± 3.0 mm, p < 0.001). Breast bud size correlated positively with gestational age (regression coefficient = 0.46 ± 0.12 mm, p < 0.001) and weight Z-score (0.59 ± 0.24 mm, p = 0.02), and negatively with White race (-1.00 ± 0.30 mm, p = 0.001). AGD was longer in males (scrotum-to-anus) than females (fourchette-to-anus) (21.0 ± 4.0 vs. 13.0 ± 2.0 mm, p < 0.001) and did not differ by race. SPL was shorter in White infants (35.0 ± 5.0 vs. 36.0 ± 5.0 mm, p = 0.04). Median TV was 0.5 cm
    Impact: This study reports physical measurements for endocrine-sensitive endpoints in healthy US newborns, including breast buds, AGD, SPL, and TV. Associations of measurements to demographic and clinical factors (including race, gestational age, and newborn length and weight) are presented. Contemporary ranges and identification of predictive factors will support further study on effects of pre- and postnatal exposures to endocrine-sensitive tissues in the infant.
    MeSH term(s) African Americans ; Animals ; Breast/anatomy & histology ; Breast/physiology ; Endocrine Disruptors ; Endocrine System/physiology ; Environmental Exposure ; Female ; Humans ; Infant Formula ; Infant, Newborn ; Male ; Milk ; Milk, Human ; Penis/anatomy & histology ; Penis/physiology ; Reproducibility of Results ; Testis/anatomy & histology ; Testis/physiology ; Whites
    Chemical Substances Endocrine Disruptors
    Language English
    Publishing date 2020-05-12
    Publishing country United States
    Document type Journal Article ; Multicenter Study ; Research Support, N.I.H., Extramural ; Research Support, N.I.H., Intramural ; Research Support, Non-U.S. Gov't
    ZDB-ID 4411-8
    ISSN 1530-0447 ; 0031-3998
    ISSN (online) 1530-0447
    ISSN 0031-3998
    DOI 10.1038/s41390-020-0950-2
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    Zs.A 564: Show issues Location:
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  9. Article ; Online: Muscle Strength, Power, and Torque Deficits in Children With Type SS Sickle Cell Disease.

    Dougherty, Kelly A / Bertolaso, Chiara / Schall, Joan I / Smith-Whitley, Kim / Stallings, Virginia A

    Journal of pediatric hematology/oncology

    2018  Volume 40, Issue 5, Page(s) 348–354

    Abstract: In African-American children aged 5 to 17 years with and without type SS sickle cell disease (SCD-SS), dominant hand maximal handgrip strength, peak power, and plantar flexion isometric maximal voluntary contraction (MVC) torque were compared with ... ...

    Abstract In African-American children aged 5 to 17 years with and without type SS sickle cell disease (SCD-SS), dominant hand maximal handgrip strength, peak power, and plantar flexion isometric maximal voluntary contraction (MVC) torque were compared with adjustments for body size and composition. Children with SCD-SS (n=21; age, 11±1 y) compared with healthy control children (n=23; 10±1 y) did not differ by age, sex, or maturation stage, but had significantly lower Z scores for height, weight, body mass index, arm circumference, upper arm muscle area, and lean mass-for-height. Children with SCD-SS had significantly lower unadjusted handgrip strength (16±2 vs. 23±2 kg, P<0.01), peak power (1054±107 vs. 1488±169 W, P<0.04) and MVC torques at 2 angles (10 degrees: 27±3 vs. 42±5 Nm; 20 degrees: 21±3 vs. 34±4 Nm; all P<0.05). Performance decrements persisted when handgrip strength was adjusted for lean body mass and fat mass explaining 66% of the variance; peak power adjusted for age, lean body mass, fat mass, and height explaining 91% of the variance; and the highest MVC torque (10-degree angle) adjusted for left leg length, lean mass-for-height, and fat mass-for-height Z scores explaining 65% of the variance. This suggests additional factors contribute to the attenuated anaerobic performance.
    MeSH term(s) Adolescent ; Age Factors ; Anemia, Sickle Cell/blood ; Anemia, Sickle Cell/physiopathology ; Body Weight ; Calcium/blood ; Child ; Child, Preschool ; Female ; Hand Strength ; Humans ; Male ; Nutritional Status
    Chemical Substances Calcium (SY7Q814VUP)
    Language English
    Publishing date 2018-04-05
    Publishing country United States
    Document type Journal Article ; Multicenter Study ; Randomized Controlled Trial ; Research Support, N.I.H., Extramural ; Research Support, Non-U.S. Gov't
    ZDB-ID 1231152-2
    ISSN 1536-3678 ; 1077-4114 ; 0192-8562
    ISSN (online) 1536-3678
    ISSN 1077-4114 ; 0192-8562
    DOI 10.1097/MPH.0000000000001143
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  10. Article ; Online: Energy Balance and Mechanisms of Weight Gain with Ivacaftor Treatment of Cystic Fibrosis Gating Mutations.

    Stallings, Virginia A / Sainath, Nina / Oberle, Megan / Bertolaso, Chiara / Schall, Joan I

    The Journal of pediatrics

    2018  Volume 201, Page(s) 229–237.e4

    Abstract: Objective: To determine if ivacaftor treatment results in weight gain and improved pulmonary function in people with cystic fibrosis transmembrane conductance regulator gating mutations.: Study design: Children and adults with cystic fibrosis and at ... ...

    Abstract Objective: To determine if ivacaftor treatment results in weight gain and improved pulmonary function in people with cystic fibrosis transmembrane conductance regulator gating mutations.
    Study design: Children and adults with cystic fibrosis and at least 1 cystic fibrosis transmembrane conductance regulator gating mutation were evaluated in this observational study before and after 3 months of ivacaftor treatment. Body size and composition, total energy expenditure, resting energy expenditure (REE%) as percent predicted, coefficient of fat absorption (CFA%), fecal calprotectin, fecal elastase, and quality of life were assessed. Some outcomes were explored by pancreatic status.
    Results: There were 23 patients (5-61 years of age) who completed the study; 70% had pancreatic insufficiency (PI). Patients gained 2.5 ± 2.2 kg (P < .001) with increased (P < .05) fat-free mass (0.9 ± 1.9 kg) and fat mass (1.6 ± 1.5 kg). REE% decreased by 5.5 ± 12.0% (P < .05), fecal calprotectin decreased by 30 ± 40 µg/g stool (P < .01), and total energy expenditure was unchanged. Improvements were greater for PI than patients who were pancreatic-sufficient. CFA% increased significantly only with PI. The change (Δ) in weight was positively correlated with the percent change in forced expiratory volume at 1 second (r = 0.46; P = .028) and ΔCFA% (r = 0.47; P = .032) and negatively with ΔREE% (r = -0.50; P = .017). Together, ΔREE%, ΔCFA%, and the percent change in forced expiratory volume at 1 second explained 58% of the variance in weight gain (adjusted R
    Conclusions: Mechanisms identified for ivacaftor-associated weight gain were decreased REE, gut inflammation, and fat malabsorption (CFA).
    Trial registration: ClinicalTrials.gov: NCT02141464.
    MeSH term(s) Adolescent ; Adult ; Aminophenols/therapeutic use ; Child ; Child, Preschool ; Chloride Channel Agonists/therapeutic use ; Cystic Fibrosis/drug therapy ; Cystic Fibrosis/genetics ; Cystic Fibrosis/metabolism ; Cystic Fibrosis Transmembrane Conductance Regulator/genetics ; Cystic Fibrosis Transmembrane Conductance Regulator/metabolism ; DNA/genetics ; DNA Mutational Analysis ; Energy Metabolism/physiology ; Female ; Humans ; Male ; Middle Aged ; Mutation ; Quality of Life ; Quinolones/therapeutic use ; Treatment Outcome ; Weight Gain/physiology ; Young Adult
    Chemical Substances Aminophenols ; CFTR protein, human ; Chloride Channel Agonists ; Quinolones ; Cystic Fibrosis Transmembrane Conductance Regulator (126880-72-6) ; ivacaftor (1Y740ILL1Z) ; DNA (9007-49-2)
    Language English
    Publishing date 2018-07-18
    Publishing country United States
    Document type Journal Article ; Multicenter Study ; Research Support, N.I.H., Extramural ; Research Support, Non-U.S. Gov't
    ZDB-ID 3102-1
    ISSN 1097-6833 ; 0022-3476
    ISSN (online) 1097-6833
    ISSN 0022-3476
    DOI 10.1016/j.jpeds.2018.05.018
    Database MEDical Literature Analysis and Retrieval System OnLINE

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