ArticleEuropean journal of pediatrics2009
Vitamin E supplementation improves neutropenia and reduces the frequency of infections in patients with glycogen storage disease type 1b.
Article in European journal of pediatrics, 2009. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 8 papers.
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Who cites it
8 citing papers in PubMed, 29 citations in OpenAlex.
- Exploring the role of antioxidants in sepsis-associated oxidative stress: a comprehensive review.Frontiers in cellular and infection microbiology · 2024Review
- Glycogen storage diseases: An update.World journal of gastroenterology · 2023Review
- Glycogen storage disease type I: Genetic etiology, clinical manifestations, and conventional and gene therapies.Pediatric discovery · 2023Article
- Vitamin E as promising adjunct treatment option in the combat of infectious diseases caused by bacterial including multi-drug resistant pathogens - Results from a comprehensive literature survey.European journal of microbiology & immunology · 2020Article
- Vitamin E Improves Clinical Outcome of Patients Affected by Glycogen Storage Disease Type Ib.JIMD reports · 2016Article
- Glycogen storage disease type Ia (GSDIa) but not Glycogen storage disease type Ib (GSDIb) is associated to an increased risk of metabolic syndrome: possible role of microsomal glucose 6-phosphate accumulation.Orphanet journal of rare diseases · 2015Article
- Involvement of endocrine system in a patient affected by glycogen storage disease 1b: speculation on the role of autoimmunity.Italian journal of pediatrics · 2014Article
- Neutropenia in type Ib glycogen storage disease.Current opinion in hematology · 2010Review
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Authors and funding
10 authors at 3 institutions in 1 country.
Funding
Abstract
backgroundNeutropenia and/or neutrophil dysfunction are part of glycogen storage disease type 1b (GSD1b) phenotype. Recent studies indicated that activation of apoptosis and increased reactive oxygen species are implicated in the pathogenesis of neutropenia in GSD1b.
methodsWe studied seven GSD1b patients over a 2-year-period to evaluate the efficacy of vitamin E, a known antioxidant, in preventing or improving the clinical manifestations associated with neutropenia and neutrophil dysfunction. Frequency and severity of infections, neutrophil counts and function, ileocolonoscopy and intestinal histology, were monitored. During the first year, patients did not assume vitamin E; during the second year of the study, vitamin E supplementation was added to their therapeutic regimens.
resultsDuring vitamin E supplementation, the mean values of neutrophil counts were significantly higher (p < 0.05) and neutrophil counts lower than 500/mm(3) were found less frequently (p < 0.05); the frequency and severity of infections, mouth ulcers and perianal lesions, was reduced (p < 0.05); ileocolonoscopy and histology showed a mild improvement. Vitamin E supplementation did not result in changes in neutrophil function.
conclusionsThese results suggest that vitamin E supplementation might be beneficial in GSD1b patients and may alleviate disease manifestations associated with neutropenia.
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