Literature DB >> 2095337

Ornithine transcarbamylase deficiency in male adolescence and adulthood.

M Yoshino1, J Nishiyori, F Yamashita, R Kumashiro, H Abe, K Tanikawa, T Ohno, K Nakao, N Kaku, H Fukushima.   

Abstract

A discrete deficiency of hepatic ornithine transcarbamylase (OTC) was found in male patients who were 58, 46 and 17 years old. Each had developed hyperammonemic coma. The mother and a sister of the 17-year-old patient exhibited orotic aciduria either spontaneously or after protein loading, thus demonstrating heterozygosity. A sister of one other patient and a daughter of the third patient showed a smaller orotic aciduria after protein loading. These observations indicate that inherited deficiency of OTC should be included in the differential diagnosis of hyperammonemic states in adult male patients.

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Year:  1990        PMID: 2095337     DOI: 10.1159/000468724

Source DB:  PubMed          Journal:  Enzyme        ISSN: 0013-9432


  7 in total

1.  Rapidly fatal hyperammonemic coma in adults. Urea cycle enzyme deficiency.

Authors:  B E Wilson; W N Hobbs; J J Newmark; S J Farrow
Journal:  West J Med       Date:  1994-08

2.  Phenotypic variability in male patients carrying the mutant ornithine transcarbamylase (OTC) allele, Arg40His, ranging from a child with an unfavourable prognosis to an asymptomatic older adult.

Authors:  I Matsuda; T Matsuura; A Nishiyori; S Komaki; R Hoshide; T Matsumoto; M Funakoshi; K Kiwaki; F Endo; A Hata; M Shimadzu; M Yoshino
Journal:  J Med Genet       Date:  1996-08       Impact factor: 6.318

3.  Female heterozygotes for the hypomorphic R40H mutation can have ornithine transcarbamylase deficiency and present in early adolescence: a case report and review of the literature.

Authors:  Jason R Pinner; Mary-Louise Freckmann; Edwin P Kirk; Makoto Yoshino
Journal:  J Med Case Rep       Date:  2010-11-12

4.  Paternal transmission and slow elimination of mutant alleles associated with late-onset ornithine transcarbamylase deficiency in male patients.

Authors:  Sanae Numata; Eimei Harada; Yasuki Maeno; Isao Ueki; Yoriko Watanabe; Chieko Fujii; Takashi Yanagawa; Satoshi Takenaka; Toshiro Inoue; Shinkai Inoue; Terufumi Goushi; Tsutomu Yasutake; Toshihiko Mizuta; Makoto Yoshino
Journal:  J Hum Genet       Date:  2007-11-20       Impact factor: 3.172

5.  Hyperammonemia-induced encephalopathy: A rare devastating complication of bariatric surgery.

Authors:  Michelle L Kromas; Omar Y Mousa; Savio John
Journal:  World J Hepatol       Date:  2015-05-08

6.  Psychiatric adult-onset of urea cycle disorders: A case-series.

Authors:  Adrien Bigot; Paul Brunault; Christian Lavigne; François Feillet; Sylvie Odent; Elsa Kaphan; Christel Thauvin; Vanessa Leguy; Pierre Broué; Michel C Tchan; François Maillot
Journal:  Mol Genet Metab Rep       Date:  2017-07-06

7.  Common polymorphic OTC variants can act as genetic modifiers of enzymatic activity.

Authors:  Mónica Lopes-Marques; Ana Rita Pacheco; Maria João Peixoto; Ana Rita Cardoso; Catarina Serrano; António Amorim; Maria João Prata; David N Cooper; Luísa Azevedo
Journal:  Hum Mutat       Date:  2021-06-03       Impact factor: 4.878

  7 in total

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