Literature DB >> 10518414

A case of human vitamin A deficiency caused by an inherited defect in retinol-binding protein without clinical symptoms except night blindness.

G Wolf1.   

Abstract

Two German siblings were found to suffer from night blindness and mild retinal dystrophy but no other clinical symptoms of vitamin A deficiency. Even though they had no detectable plasma retinal-binding protein (RBP) and their plasma retinol was exceedingly low, they showed normal physiologic functions and growth. Their RBP gene was found to harbor two point mutations. Their post-prandial plasma levels of retinyl esters were normal, and it is likely that they derived their tissue retinol from retinyl esters.

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Year:  1999        PMID: 10518414     DOI: 10.1111/j.1753-4887.1999.tb06955.x

Source DB:  PubMed          Journal:  Nutr Rev        ISSN: 0029-6643            Impact factor:   7.110


  6 in total

1.  Pharmacological inhibition of lipofuscin accumulation in the retina as a therapeutic strategy for dry AMD treatment.

Authors:  Konstantin Petrukhin
Journal:  Drug Discov Today Ther Strateg       Date:  2013

2.  A non-retinoid antagonist of retinol-binding protein 4 rescues phenotype in a model of Stargardt disease without inhibiting the visual cycle.

Authors:  Boglarka Racz; Andras Varadi; Jian Kong; Rando Allikmets; Paul G Pearson; Graham Johnson; Christopher L Cioffi; Konstantin Petrukhin
Journal:  J Biol Chem       Date:  2018-06-05       Impact factor: 5.157

3.  Design, Synthesis, and Preclinical Efficacy of Novel Nonretinoid Antagonists of Retinol-Binding Protein 4 in the Mouse Model of Hepatic Steatosis.

Authors:  Christopher L Cioffi; Boglarka Racz; Andras Varadi; Emily E Freeman; Michael P Conlon; Ping Chen; Lei Zhu; Douglas B Kitchen; Keith D Barnes; William H Martin; Paul G Pearson; Graham Johnson; William S Blaner; Konstantin Petrukhin
Journal:  J Med Chem       Date:  2019-05-28       Impact factor: 7.446

4.  Exome analysis identified a novel mutation in the RBP4 gene in a consanguineous pedigree with retinal dystrophy and developmental abnormalities.

Authors:  Catherine Cukras; Terry Gaasterland; Pauline Lee; Harini V Gudiseva; Venkata R M Chavali; Raghu Pullakhandam; Bruno Maranhao; Lee Edsall; Sandra Soares; G Bhanuprakash Reddy; Paul A Sieving; Radha Ayyagari
Journal:  PLoS One       Date:  2012-11-26       Impact factor: 3.240

5.  Discovery of Bispecific Antagonists of Retinol Binding Protein 4 That Stabilize Transthyretin Tetramers: Scaffolding Hopping, Optimization, and Preclinical Pharmacological Evaluation as a Potential Therapy for Two Common Age-Related Comorbidities.

Authors:  Christopher L Cioffi; Parthasarathy Muthuraman; Arun Raja; Andras Varadi; Boglarka Racz; Konstantin Petrukhin
Journal:  J Med Chem       Date:  2020-09-17       Impact factor: 8.039

6.  Comparative pharmacokinetics and pharmacodynamics of the advanced Retinol-Binding Protein 4 antagonist in dog and cynomolgus monkey.

Authors:  Boglarka Racz; Andras Varadi; Paul G Pearson; Konstantin Petrukhin
Journal:  PLoS One       Date:  2020-01-24       Impact factor: 3.240

  6 in total

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