Literature DB >> 6111630

Hyperornithinaemia and gyrate atrophy of the retina: improvement of vision during treatment with a low-arginine diet.

R R McInnes, S A Arshinoff, L Bell, E B Marliss, J C McCulloch.   

Abstract

A 15-year-old patient with hyperornithinaemia (0.6--1.2 mmol/l) and gyrate atrophy of the retina was given a low-arginine diet to reduce plasma ornithine to a concentration (0.15--0.25 mmol/l) near the normal range. After five weeks of treatment, there was improvement in the visual function of one eye which had been severely impaired without improvement for 3 years. This improved visual function was maintained until compliance with the diet deteriorated at eight months, after which visual function regressed towards pretreatment status. Overrestriction of dietary arginine produced hyperammonaemia, indicating that arginine is an essential aminoacid in ornithine transaminase deficiency. These results suggest that reduction of plasma ornithine may reverse a metabolic neuroretinopathy in this disease, and offer hope that progression of the retinal atrophy can be arrested as well.

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Year:  1981        PMID: 6111630     DOI: 10.1016/s0140-6736(81)92858-0

Source DB:  PubMed          Journal:  Lancet        ISSN: 0140-6736            Impact factor:   79.321


  6 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.  Studies on the pathway from ornithine to proline in cultured skin fibroblasts with reference to the defect in hyperornithinaemia with hyperammonaemia and homocitrullinuria.

Authors:  R G Gray; S E Hill; R J Pollitt
Journal:  J Inherit Metab Dis       Date:  1983       Impact factor: 4.982

3.  Amino acids in retinitis pigmentosa.

Authors:  S A Arshinoff; J C McCulloch; W Macrae; A N Stein; E B Marliss
Journal:  Br J Ophthalmol       Date:  1981-09       Impact factor: 4.638

4.  Biochemical and therapeutical studies in a case of atrophia gyrata.

Authors:  W Behrens-Baumann; U König; K Schröder; I Hansmann; U Langenbeck
Journal:  Graefes Arch Clin Exp Ophthalmol       Date:  1982       Impact factor: 3.117

5.  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

6.  Low-protein diet and progression of retinal degeneration in gyrate atrophy of the choroid and retina: a twenty-six-year follow-up.

Authors:  R Santinelli; C Costagliola; C Tolone; A D'Aloia; A D'Avanzo; F Prisco; L Perrone; E Miraglia del Giudice
Journal:  J Inherit Metab Dis       Date:  2004       Impact factor: 4.982

  6 in total

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