• 文献检索
  • 文档翻译
  • 深度研究
  • 学术资讯
  • Suppr Zotero 插件Zotero 插件
  • 邀请有礼
  • 套餐&价格
  • 历史记录
应用&插件
Suppr Zotero 插件Zotero 插件浏览器插件Mac 客户端Windows 客户端微信小程序
定价
高级版会员购买积分包购买API积分包
服务
文献检索文档翻译深度研究API 文档MCP 服务
关于我们
关于 Suppr公司介绍联系我们用户协议隐私条款
关注我们

Suppr 超能文献

核心技术专利:CN118964589B侵权必究
粤ICP备2023148730 号-1Suppr @ 2026

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验

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

作者信息

Oexle Konrad

机构信息

Institute of Clinical Genetics, Medical Faculty "Carl Gustav Carus", Dresden University of Technology, Fetscherstrasse 74, 01307, Dresden, Germany.

出版信息

J Hum Genet. 2006;51(3):204-208. doi: 10.1007/s10038-005-0345-6. Epub 2006 Feb 2.

DOI:10.1007/s10038-005-0345-6
PMID:16453063
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.

摘要

相似文献

1
Biochemical data in ornithine transcarbamylase deficiency (OTCD) carrier risk estimation: logistic discrimination and combination with genetic information.
J Hum Genet. 2006;51(3):204-208. doi: 10.1007/s10038-005-0345-6. Epub 2006 Feb 2.
2
How reliable is the allopurinol load in detecting carriers for ornithine transcarbamylase deficiency?别嘌呤醇负荷试验在检测鸟氨酸转氨甲酰酶缺乏症携带者方面的可靠性如何?
J Inherit Metab Dis. 2004;27(2):179-86. doi: 10.1023/B:BOLI.0000028727.77454.bd.
3
Calculation of the reliability of the allopurinol load in detecting carriers for ornithine transcarbamylase deficiency.
J Inherit Metab Dis. 2006 Feb;29(1):241. doi: 10.1007/s10545-006-0198-y.
4
The phenotype of ostensibly healthy women who are carriers for ornithine transcarbamylase deficiency.表面健康但为鸟氨酸转氨甲酰酶缺乏症携带者的女性的表型。
Medicine (Baltimore). 1998 Nov;77(6):389-97.
5
Prenatal treatment of ornithine transcarbamylase deficiency.鸟氨酸氨甲酰基转移酶缺陷症的产前治疗。
Mol Genet Metab. 2018 Mar;123(3):297-300. doi: 10.1016/j.ymgme.2018.01.004. Epub 2018 Jan 16.
6
An integrated approach to the diagnosis and prospective management of partial ornithine transcarbamylase deficiency.鸟氨酸转氨甲酰酶部分缺乏症的诊断与前瞻性管理的综合方法。
Pediatrics. 2002 Jan;109(1):150-2. doi: 10.1542/peds.109.1.150.
7
Allopurinol-induced orotidinuria. A test for mutations at the ornithine carbamoyltransferase locus in women.别嘌醇诱发的乳清酸尿症。一项针对女性鸟氨酸氨甲酰基转移酶基因座突变的检测。
N Engl J Med. 1990 Jun 7;322(23):1641-5. doi: 10.1056/NEJM199006073222305.
8
Long-term outcomes in Ornithine Transcarbamylase deficiency: a series of 90 patients.鸟氨酸转氨甲酰酶缺乏症的长期预后:90例患者的系列研究
Orphanet J Rare Dis. 2015 May 10;10:58. doi: 10.1186/s13023-015-0266-1.
9
Remark on utility and error rates of the allopurinol test in detecting mild ornithine transcarbamylase deficiency.
Mol Genet Metab. 2002 May;76(1):71-5. doi: 10.1016/s1096-7192(02)00025-2.
10
Hyperammonemic encephalopathy in a child with ornithine transcarbamylase deficiency due to a novel combined heterozygous mutations.一名因新型复合杂合突变导致鸟氨酸转氨甲酰酶缺乏的儿童发生高氨血症性脑病。
Am J Emerg Med. 2015 Mar;33(3):474.e1-3. doi: 10.1016/j.ajem.2014.08.038. Epub 2014 Aug 23.

引用本文的文献

1
Humanized liver mouse model with transplanted human hepatocytes from patients with ornithine transcarbamylase deficiency.人源化肝脏小鼠模型,移植了来自鸟氨酸转氨甲酰酶缺乏症患者的人肝细胞。
J Inherit Metab Dis. 2021 May;44(3):618-628. doi: 10.1002/jimd.12347. Epub 2020 Dec 30.

本文引用的文献

1
How reliable is the allopurinol load in detecting carriers for ornithine transcarbamylase deficiency?别嘌呤醇负荷试验在检测鸟氨酸转氨甲酰酶缺乏症携带者方面的可靠性如何?
J Inherit Metab Dis. 2004;27(2):179-86. doi: 10.1023/B:BOLI.0000028727.77454.bd.
2
Remark on utility and error rates of the allopurinol test in detecting mild ornithine transcarbamylase deficiency.
Mol Genet Metab. 2002 May;76(1):71-5. doi: 10.1016/s1096-7192(02)00025-2.
3
Mutations and polymorphisms in the human ornithine transcarbamylase gene.人类鸟氨酸转氨甲酰酶基因中的突变与多态性
Hum Mutat. 2002 Feb;19(2):93-107. doi: 10.1002/humu.10035.
4
Genotype spectrum of ornithine transcarbamylase deficiency: correlation with the clinical and biochemical phenotype.鸟氨酸转氨甲酰酶缺乏症的基因型谱:与临床和生化表型的相关性。
Am J Med Genet. 2000 Aug 14;93(4):313-9. doi: 10.1002/1096-8628(20000814)93:4<313::aid-ajmg11>3.0.co;2-m.
5
The allopurinol load test lacks specificity for primary urea cycle defects but may indicate unrecognized mitochondrial disease.
J Inherit Metab Dis. 1999 Apr;22(2):174-84. doi: 10.1023/a:1005406205548.
6
The phenotype of ostensibly healthy women who are carriers for ornithine transcarbamylase deficiency.表面健康但为鸟氨酸转氨甲酰酶缺乏症携带者的女性的表型。
Medicine (Baltimore). 1998 Nov;77(6):389-97.
7
The allopurinol loading test for identification of carriers for ornithine carbamoyl transferase deficiency: studies in a healthy control population and females at risk.
Clin Chim Acta. 1994 Jan 14;224(1):45-54. doi: 10.1016/0009-8981(94)90119-8.
8
Proportions of spontaneous mutations in males and females with ornithine transcarbamylase deficiency.
Am J Med Genet. 1995 Jan 2;55(1):67-70. doi: 10.1002/ajmg.1320550118.
9
Diagnostic value of orotic acid excretion in heritable disorders of the urea cycle and in hyperammonemia due to organic acidurias.乳清酸排泄在尿素循环遗传性疾病及有机酸尿症所致高氨血症中的诊断价值。
Eur J Pediatr. 1980 Aug;134(2):109-13. doi: 10.1007/BF01846026.
10
Duchenne muscular dystrophy carrier detection using logistic discrimination: serum creatine kinase and hemopexin in combination.运用逻辑判别法检测杜氏肌营养不良症携带者:血清肌酸激酶与血浆血红素结合蛋白联合检测
Am J Med Genet. 1981;8(4):397-409. doi: 10.1002/ajmg.1320080406.