Literature DB >> 34655400

A Double-Blind, Placebo-Controlled, Crossover Study of Magnesium Supplementation in Patients with XMEN Disease.

Samuel D Chauvin1,2, Susan Price3, Juan Zou1, Sally Hunsberger4, Alessandra Brofferio5, Helen Matthews1, Morgan Similuk1, Sergio D Rosenzweig6, Helen C Su3, Jeffrey I Cohen7, Michael J Lenardo8, Juan C Ravell9,10,11,12.   

Abstract

X-linked MAGT1 deficiency with increased susceptibility to Epstein-Barr virus (EBV) infection and N-linked glycosylation defect (XMEN) disease is an inborn error of immunity caused by loss-of-function mutations in the magnesium transporter 1 (MAGT1) gene. The original studies of XMEN patients focused on impaired magnesium regulation, leading to decreased EBV-cytotoxicity and the loss of surface expression of the activating receptor "natural killer group 2D" (NKG2D) on CD8+ T cells and NK cells. In vitro studies showed that supraphysiological supplementation of magnesium rescued these defects. Observational studies in 2 patients suggested oral magnesium supplementation could decrease EBV viremia. Hence, we performed a randomized, double-blind, placebo-controlled, crossover study in 2 parts. In part 1, patients received either oral magnesium L-threonate (MLT) or placebo for 12 weeks followed by 12 weeks of the other treatment. Part 2 began with 3 days of high-dose intravenous (IV) magnesium sulfate (MgSO4) followed by open-label MLT for 24 weeks. One EBV-infected and 3 EBV-naïve patients completed part 1. One EBV-naïve patient was removed from part 2 of the study due to asymptomatic elevation of liver enzymes during IV MgSO4. No change in EBV or NKG2D status was observed. In vitro magnesium supplementation experiments in cells from 14 XMEN patients failed to significantly rescue NKG2D expression and the clinical trial was stopped. Although small, this study indicates magnesium supplementation is unlikely to be an effective therapeutic option in XMEN disease.
© 2021. This is a U.S. government work and not under copyright protection in the U.S.; foreign copyright protection may apply.

Entities:  

Keywords:  Immunodeficiency; MAGT1; NKG2D; XMEN disease; congenital disorder of glycosylation; magnesium

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Year:  2021        PMID: 34655400     DOI: 10.1007/s10875-021-01137-w

Source DB:  PubMed          Journal:  J Clin Immunol        ISSN: 0271-9142            Impact factor:   8.317


  1 in total

1.  Making 'null effects' informative: statistical techniques and inferential frameworks.

Authors:  Christopher Harms; Daniël Lakens
Journal:  J Clin Transl Res       Date:  2018-07-30
  1 in total
  2 in total

1.  Lack of NKG2D in MAGT1-deficient patients is caused by hypoglycosylation.

Authors:  Eline Blommaert; Natalia A Cherepanova; Frederik Staels; Matthew P Wilson; Reid Gilmore; Rik Schrijvers; Jaak Jaeken; François Foulquier; Gert Matthijs
Journal:  Hum Genet       Date:  2022-02-19       Impact factor: 5.881

2.  Further Delineation of the Spectrum of XMEN Disease in Six Chinese Pediatric Patients.

Authors:  Xiaomin Peng; Yi Lu; Huijun Wang; Bingbing Wu; Mingyu Gan; Suzhen Xu; Deyi Zhuang; Jianshe Wang; Jinqiao Sun; Xiaochuan Wang; Wenhao Zhou
Journal:  Front Genet       Date:  2022-01-25       Impact factor: 4.599

  2 in total

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