Literature DB >> 6734670

Ornithine carbamoyl transferase deficiency: a neuropathological study.

B N Harding, J V Leonard, M Erdohazi.   

Abstract

A detailed autopsy study of three children with ornithine carbamoyl transferase (OCT) deficiency is presented. Although variable in extent, a basic pattern of neuropathological lesions is discernible. Case 1 shows gross cerebral atrophy, cases 2 and 3 milder lesions in the basal nuclei but also multiple cerebellar heterotopias and delayed myelination. We suggest that the findings may provide evidence that OCT deficiency can have a teratogenic effect in utero and suggest that there is a need to monitor the pregnancies of carriers of this disorder.

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Year:  1984        PMID: 6734670     DOI: 10.1007/BF00572763

Source DB:  PubMed          Journal:  Eur J Pediatr        ISSN: 0340-6199            Impact factor:   3.183


  22 in total

1.  Neuropathological observations in a patient with carbamylphosphate-synthetase deficiency and in two sibs.

Authors:  E J Ebels
Journal:  Arch Dis Child       Date:  1972-02       Impact factor: 3.791

2.  An experimental model for the study of hepatic encephalopathy.

Authors:  M D Norenberg; L W Lapham; F A Nichols; A G May
Journal:  Arch Neurol       Date:  1974-08

3.  Brain histology and behavior of mice injected with urease.

Authors:  G E Gibson; A Zimber; L Krook; E P Richardson; W J Visek
Journal:  J Neuropathol Exp Neurol       Date:  1974-04       Impact factor: 3.685

4.  Evidence for x-linked dominant inheritance of ornithine transcarbamylase deficiency.

Authors:  E M Short; H O Conn; P J Snodgrass; A G Campbell; L E Rosenberg
Journal:  N Engl J Med       Date:  1973-01-04       Impact factor: 91.245

5.  Pituitary necrosis in patients maintained on mechanical respirators.

Authors:  P M Daniel; E J Spicer; C S Treip
Journal:  J Pathol       Date:  1973-10       Impact factor: 7.996

6.  Hereditary hyperammonaemia.

Authors:  C J Bruton; J A Corsellis; A Russell
Journal:  Brain       Date:  1970       Impact factor: 13.501

7.  Type II Alzheimer change experimentally produced in astrocytes in the rat.

Authors:  J B Cavanagh; M H Kyu
Journal:  J Neurol Sci       Date:  1971-01       Impact factor: 3.181

8.  Treatment of complete ornithine transcarbamylase deficiency with nitrogen-free analogues of essential amino acids.

Authors:  J W McReynolds; S Mantagos; S Brusilow; L E Rosenberg
Journal:  J Pediatr       Date:  1978-09       Impact factor: 4.406

9.  Atypical clinical course of ornithine transcarbamylase deficiency due to a new mutant (comparison with Reye's disease).

Authors:  I Krieger; P J Snodgrass; J Roskamp
Journal:  J Clin Endocrinol Metab       Date:  1979-03       Impact factor: 5.958

10.  Partial ornithine carbamyl transferase deficiency: an inborn error of the urea cycle presenting as orotic aciduria in a male infant.

Authors:  P MacLeod; S Mackenzie; C R Scriver
Journal:  Can Med Assoc J       Date:  1972-09-09       Impact factor: 8.262

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  14 in total

Review 1.  Brain imaging in urea cycle disorders.

Authors:  Andrea Gropman
Journal:  Mol Genet Metab       Date:  2010-02-13       Impact factor: 4.797

2.  Prenatal diagnosis of ornithine carbamoyl transferase deficiency using a gene specific probe.

Authors:  M E Pembrey; J M Old; J V Leonard; C H Rodeck; R Warren; K E Davies
Journal:  J Med Genet       Date:  1985-12       Impact factor: 6.318

3.  Amino acidaemias and brain maturation: interference with sulphate activation and myelin metabolism.

Authors:  F A Hommes
Journal:  J Inherit Metab Dis       Date:  1985       Impact factor: 4.982

4.  Clinical and pathological study of three Tunisian siblings with L-2-hydroxyglutaric aciduria.

Authors:  A Larnaout; F Hentati; S Belal; C Ben Hamida; N Kaabachi; M Ben Hamida
Journal:  Acta Neuropathol       Date:  1994       Impact factor: 17.088

Review 5.  Effects of hyperammonaemia on brain function.

Authors:  R F Butterworth
Journal:  J Inherit Metab Dis       Date:  1998       Impact factor: 4.982

6.  Loss of [3H]MK801 binding sites in brain in congenital ornithine transcarbamylase deficiency.

Authors:  L Ratnakumari; I A Qureshi; R F Butterworth
Journal:  Metab Brain Dis       Date:  1995-09       Impact factor: 3.584

Review 7.  Teratogenic inborn errors of metabolism.

Authors:  J V Leonard
Journal:  Postgrad Med J       Date:  1986-02       Impact factor: 2.401

Review 8.  Neurological implications of urea cycle disorders.

Authors:  A L Gropman; M Summar; J V Leonard
Journal:  J Inherit Metab Dis       Date:  2007-11-23       Impact factor: 4.982

Review 9.  Ammonia toxicity to the brain.

Authors:  Olivier Braissant; Valérie A McLin; Cristina Cudalbu
Journal:  J Inherit Metab Dis       Date:  2012-10-30       Impact factor: 4.982

10.  Regional amino acid neurotransmitter changes in brains of spf/Y mice with congenital ornithine transcarbamylase deficiency.

Authors:  L Ratnakumari; I A Qureshi; R F Butterworth
Journal:  Metab Brain Dis       Date:  1994-03       Impact factor: 3.584

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