Literature DB >> 7254777

Ocular findings in patients with gyrate atrophy on pyridoxine and low-protein, low-arginine diets.

E L Berson, V E Shih, P L Sullivan.   

Abstract

Five patients, ages 12 to 30, with gyrate atrophy have shown substantial (60% or greater) decreases in plasma ornithine concentrations within four to eight weeks when placed on a therapeutic trial of low-protein (10-15 g/day), low-arginine diets supplemented with essential amino acids (EAA) and pyridoxine hydrochloride. Four of five patients have continued on modified protein restriction (20-35 g/day) and one on pyridoxine (300 mg/day) alone with maintenance of plasma ornithine in the range of 30 to 60% below pretherapeutic trial levels. After one year, four of five patients have shown no significant improvement in visual acuity, fields, final dark-adapted thresholds, electroretinograms, or fundus appearance. One patient with the poorest control of plasma ornithine has developed a decrease in ERG amplitudes and a new area of chorioretinal atrophy. These patients continue in this trial to determine whether or not any reductions in hyperornithinemia will modify the course of the ocular disease.

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Year:  1981        PMID: 7254777     DOI: 10.1016/s0161-6420(81)35029-5

Source DB:  PubMed          Journal:  Ophthalmology        ISSN: 0161-6420            Impact factor:   12.079


  4 in total

1.  Correction of ornithine accumulation prevents retinal degeneration in a mouse model of gyrate atrophy of the choroid and retina.

Authors:  T Wang; G Steel; A H Milam; D Valle
Journal:  Proc Natl Acad Sci U S A       Date:  2000-02-01       Impact factor: 11.205

2.  A far advanced case of gyrate atrophy in a 12-year-old girl.

Authors:  H D Bakker; N G Abeling; M J van Schooneveld; R J Wanders; A H van Gennip
Journal:  J Inherit Metab Dis       Date:  1991       Impact factor: 4.982

Review 3.  B6-responsive disorders: a model of vitamin dependency.

Authors:  Peter T Clayton
Journal:  J Inherit Metab Dis       Date:  2006 Apr-Jun       Impact factor: 4.982

4.  Clinical trials of vitamin B6 and proline supplementation for gyrate atrophy of the choroid and retina.

Authors:  S Hayasaka; T Saito; H Nakajima; O Takahashi; K Mizuno; K Tada
Journal:  Br J Ophthalmol       Date:  1985-04       Impact factor: 4.638

  4 in total

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