Literature DB >> 16453063

Biochemical data in ornithine transcarbamylase deficiency (OTCD) carrier risk estimation: logistic discrimination and combination with genetic information.

Konrad Oexle1.   

Abstract

One-fifth of the gene mutations causing ornithine transcarbamylase deficiency cannot be detected. In such cases carrier risk estimation must refer to biochemical data-such as increased plasma glutamine concentration or increased orotidine excretion after allopurinol load -although these parameters do not yield a definite diagnosis. Here, I derive odds for carrier risk estimation from published data, i.e. from mean and standard deviation of glutamine concentrations in carriers and noncarriers, assuming normal distributions, and from allopurinol test results in individual carriers and noncarriers using logistic regression. I show how such biochemical information may be combined with genetic information, thus demonstrating the usefulness of biochemical data. The necessity to assess individual results in larger proband groups and to consider possible correlations between different parameters is indicated.

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Year:  2006        PMID: 16453063     DOI: 10.1007/s10038-005-0345-6

Source DB:  PubMed          Journal:  J Hum Genet        ISSN: 1434-5161            Impact factor:   3.172


  12 in total

1.  Allopurinol-induced orotidinuria. A test for mutations at the ornithine carbamoyltransferase locus in women.

Authors:  E R Hauser; J E Finkelstein; D Valle; S W Brusilow
Journal:  N Engl J Med       Date:  1990-06-07       Impact factor: 91.245

Review 2.  Mutations and polymorphisms in the human ornithine transcarbamylase gene.

Authors:  Mendel Tuchman; Naser Jaleel; Hiroki Morizono; Lisa Sheehy; Michael G Lynch
Journal:  Hum Mutat       Date:  2002-02       Impact factor: 4.878

3.  Genotype spectrum of ornithine transcarbamylase deficiency: correlation with the clinical and biochemical phenotype.

Authors:  B A McCullough; M Yudkoff; M L Batshaw; J M Wilson; S E Raper; M Tuchman
Journal:  Am J Med Genet       Date:  2000-08-14

4.  Proportions of spontaneous mutations in males and females with ornithine transcarbamylase deficiency.

Authors:  M Tuchman; I Matsuda; A Munnich; S Malcolm; S Strautnieks; T Briede
Journal:  Am J Med Genet       Date:  1995-01-02

5.  Remark on utility and error rates of the allopurinol test in detecting mild ornithine transcarbamylase deficiency.

Authors:  Konrad Oexle; Luisa Bonafé; Beat Steinmann
Journal:  Mol Genet Metab       Date:  2002-05       Impact factor: 4.797

6.  Duchenne muscular dystrophy carrier detection using logistic discrimination: serum creatine kinase and hemopexin in combination.

Authors:  M E Percy; D F Andrews; M W Thompson
Journal:  Am J Med Genet       Date:  1981

7.  The allopurinol loading test for identification of carriers for ornithine carbamoyl transferase deficiency: studies in a healthy control population and females at risk.

Authors:  I Sebesta; L D Fairbanks; P M Davies; H A Simmonds; J V Leonard
Journal:  Clin Chim Acta       Date:  1994-01-14       Impact factor: 3.786

8.  Making the most of multiple measurements in estimating carrier probability in Duchenne muscular dystrophy: the Bayesian incorporation of repeated measurements using logistic discrimination.

Authors:  M E Percy; D F Andrews; P M Brasher; A C Rusk
Journal:  Am J Med Genet       Date:  1987-04

9.  The phenotype of ostensibly healthy women who are carriers for ornithine transcarbamylase deficiency.

Authors:  N E Maestri; C Lord; M Glynn; A Bale; S W Brusilow
Journal:  Medicine (Baltimore)       Date:  1998-11       Impact factor: 1.889

10.  Diagnostic value of orotic acid excretion in heritable disorders of the urea cycle and in hyperammonemia due to organic acidurias.

Authors:  C Bachmann; J P Colombo
Journal:  Eur J Pediatr       Date:  1980-08       Impact factor: 3.183

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  1 in total

1.  Humanized liver mouse model with transplanted human hepatocytes from patients with ornithine transcarbamylase deficiency.

Authors:  Go Sugahara; Chihiro Yamasaki; Ami Yanagi; Suzue Furukawa; Yuko Ogawa; Akinari Fukuda; Shin Enosawa; Akihiro Umezawa; Yuji Ishida; Chise Tateno
Journal:  J Inherit Metab Dis       Date:  2020-12-30       Impact factor: 4.982

  1 in total

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