Literature DB >> 7254776

Systemic manifestations of gyrate atrophy of the choroid and retina.

M I Kaiser-Kupfer, T Kuwabara, V Askanas, L Brody, K Takki, I Dvoretzky, W K Engel.   

Abstract

In ten patients with gyrate atrophy (GA) and hyperornithinemia, the head hair was fine, straight, and sparse. On microscopic examination, both scalp and pubic hair contained intermittent dark cores within the medullary zone, which was not a deposit, but appeared to be due to the unusual refractive properties of loosely formed macrofilaments amidst wide spaces containing a structureless electron lucent but compact substance, which was insoluble in both water and solvents. Seven of the ten patients had abnormal wave forms on electroencephalography. Three of the five patients who underwent muscle biopsy had tubular aggregates. Of particular interest was the toxicity of exogenous ornithine added to muscle cell culture from GA patients as compared with the lack of toxicity in muscle from control patients. What specific role the hyperornithinemia and absence of OAT is playing in the histopathology of hair and muscle and the EEG changes awaits further biochemical investigation.

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Year:  1981        PMID: 7254776     DOI: 10.1016/s0161-6420(81)35030-1

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


  5 in total

1.  Myopathy with tubular aggregates and gyrate atrophy of the choroid and retina due to hyperornithinaemia.

Authors:  M Fleury; R Barbier; F Ziegler; M Mohr; O Caron; H Dollfus; C Tranchant; J M Warter
Journal:  J Neurol Neurosurg Psychiatry       Date:  2006-11-06       Impact factor: 10.154

2.  Central nervous system involvement in gyrate atrophy of the choroid and retina with hyperornithinaemia.

Authors:  M Valtonen; K Näntö-Salonen; S Jääskeläinen; K Heinänen; A Alanen; O J Heinonen; N Lundbom; M Erkintalo; O Simell
Journal:  J Inherit Metab Dis       Date:  1999-12       Impact factor: 4.982

3.  Gyrate atrophy of the choroid and retina with hyperornithinemia, cystinuria and lysinuria responsive to vitamin B6.

Authors:  Fatos Tanzer; Mehtap Firat; Meral Alagoz; Haydar Erdogan
Journal:  BMJ Case Rep       Date:  2011-03-15

4.  Gyrate atrophy of the choroid and retina: ERG of the neural retina and the pigment epithelium.

Authors:  C Raitta; S Carlson; K Vannas-Sulonen
Journal:  Br J Ophthalmol       Date:  1990-06       Impact factor: 4.638

5.  Multimodal imaging of foveoschisis and macular pseudohole associated with gyrate atrophy: a family report.

Authors:  Imène Zhioua Braham; Imen Ammous; Rim Maalej; Majdi Boukari; Ilhem Mili Boussen; Khalil Errais; Raja Zhioua
Journal:  BMC Ophthalmol       Date:  2018-04-12       Impact factor: 2.209

  5 in total

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