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Effect of High-dose Vitamin A Supplementation in Children With Sickle Cell Disease: A Randomized, Double-blind, Dose-finding Pilot Study.
J Pediatr Hematol Oncol. 2020 03; 42(2):83-91.JP

Abstract

Suboptimal vitamin A status (serum retinol <30 µg/dL) is associated with poor clinical outcomes in children with the hemoglobin-SS disease (HbSS), and supplementation with the recommended daily allowance of retinol is ineffective in improving vitamin A status. In a single-center randomized blinded dose-finding pilot study, we compared vitamin A and nutritional status in children with HbSS to healthy children and explored the impact of high-dose supplementation on the primary outcome serum vitamin A status. Exploratory outcomes included hematologic, nutritional, immunologic, and muscle function status in children with HbSS. A mixed-effects linear regression model evaluated associations between vitamin A dose, serum retinol, and exploratory outcomes. Twenty healthy children participated, and 22 subjects with HbSS were randomized to oral 3000 or 6000 IU/d retinol for 8 weeks; 21 subjects completed all evaluations. Serum retinol, growth, and nutritional status were all suboptimal in HbSS subjects at baseline, and supplementation did not change vitamin A status. Fetal hemoglobin (Δ=2.5, 95% confidence interval [CI], 0.5-4.3), mean corpuscular volume (Δ=2.7, 95% CI, 0.7-4.7), mean corpuscular hemoglobin (Δ=1.4, 95% CI, 0.5-2.3), and mean corpuscular hemoglobin concentration (Δ=0.5, 95% CI, 0.1-0.9) all improved with supplementation. Mild improvements in erythrocyte indices, growth status, and muscle function occurred independent of hydroxyurea use.

Authors+Show Affiliations

Division of Gastroenterology, Hepatology, and Nutrition. Department of Pediatrics, University of Pennsylvania Perelman School of Medicine, Philadelphia, PA.Division of Gastroenterology, Hepatology, and Nutrition.Division of Gastroenterology, Hepatology, and Nutrition.Division of Hematology, Children's Hospital of Philadelphia. Department of Pediatrics, University of Pennsylvania Perelman School of Medicine, Philadelphia, PA.Division of Hematology, Children's Hospital of Philadelphia. Department of Pediatrics, University of Pennsylvania Perelman School of Medicine, Philadelphia, PA.Division of Gastroenterology, Hepatology, and Nutrition. Department of Pediatrics, University of Pennsylvania Perelman School of Medicine, Philadelphia, PA.

Pub Type(s)

Journal Article
Randomized Controlled Trial
Research Support, N.I.H., Extramural
Research Support, Non-U.S. Gov't

Language

eng

PubMed ID

31764511

Citation

Brownell, Jefferson N., et al. "Effect of High-dose Vitamin a Supplementation in Children With Sickle Cell Disease: a Randomized, Double-blind, Dose-finding Pilot Study." Journal of Pediatric Hematology/oncology, vol. 42, no. 2, 2020, pp. 83-91.
Brownell JN, Schall JI, Mcanlis CR, et al. Effect of High-dose Vitamin A Supplementation in Children With Sickle Cell Disease: A Randomized, Double-blind, Dose-finding Pilot Study. J Pediatr Hematol Oncol. 2020;42(2):83-91.
Brownell, J. N., Schall, J. I., Mcanlis, C. R., Smith-Whitley, K., Norris, C. F., & Stallings, V. A. (2020). Effect of High-dose Vitamin A Supplementation in Children With Sickle Cell Disease: A Randomized, Double-blind, Dose-finding Pilot Study. Journal of Pediatric Hematology/oncology, 42(2), 83-91. https://doi.org/10.1097/MPH.0000000000001673
Brownell JN, et al. Effect of High-dose Vitamin a Supplementation in Children With Sickle Cell Disease: a Randomized, Double-blind, Dose-finding Pilot Study. J Pediatr Hematol Oncol. 2020;42(2):83-91. PubMed PMID: 31764511.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Effect of High-dose Vitamin A Supplementation in Children With Sickle Cell Disease: A Randomized, Double-blind, Dose-finding Pilot Study. AU - Brownell,Jefferson N, AU - Schall,Joan I, AU - Mcanlis,Carolyn R, AU - Smith-Whitley,Kim, AU - Norris,Cynthia F, AU - Stallings,Virginia A, PY - 2019/11/26/pubmed PY - 2020/8/14/medline PY - 2019/11/26/entrez SP - 83 EP - 91 JF - Journal of pediatric hematology/oncology JO - J Pediatr Hematol Oncol VL - 42 IS - 2 N2 - Suboptimal vitamin A status (serum retinol <30 µg/dL) is associated with poor clinical outcomes in children with the hemoglobin-SS disease (HbSS), and supplementation with the recommended daily allowance of retinol is ineffective in improving vitamin A status. In a single-center randomized blinded dose-finding pilot study, we compared vitamin A and nutritional status in children with HbSS to healthy children and explored the impact of high-dose supplementation on the primary outcome serum vitamin A status. Exploratory outcomes included hematologic, nutritional, immunologic, and muscle function status in children with HbSS. A mixed-effects linear regression model evaluated associations between vitamin A dose, serum retinol, and exploratory outcomes. Twenty healthy children participated, and 22 subjects with HbSS were randomized to oral 3000 or 6000 IU/d retinol for 8 weeks; 21 subjects completed all evaluations. Serum retinol, growth, and nutritional status were all suboptimal in HbSS subjects at baseline, and supplementation did not change vitamin A status. Fetal hemoglobin (Δ=2.5, 95% confidence interval [CI], 0.5-4.3), mean corpuscular volume (Δ=2.7, 95% CI, 0.7-4.7), mean corpuscular hemoglobin (Δ=1.4, 95% CI, 0.5-2.3), and mean corpuscular hemoglobin concentration (Δ=0.5, 95% CI, 0.1-0.9) all improved with supplementation. Mild improvements in erythrocyte indices, growth status, and muscle function occurred independent of hydroxyurea use. SN - 1536-3678 UR - https://www.unboundmedicine.com/medline/citation/31764511/Effect_of_High_dose_Vitamin_A_Supplementation_in_Children_With_Sickle_Cell_Disease:_A_Randomized_Double_blind_Dose_finding_Pilot_Study_ DB - PRIME DP - Unbound Medicine ER -