نتایج جستجو برای: urea cycle deficiency

تعداد نتایج: 441864  

Journal: :Annals of Clinical Biochemistry: International Journal of Laboratory Medicine 1977

2011
Hyo Jeong Kim Se Jin Park Kook In Park Jin Sung Lee Ho Sun Eun Ji Hong Kim Jae Il Shin

Ornithine transcarbamylase (OTC) deficiency is well known as the most common inherited disorder of the urea cycle, and 1 of the most common causes of hyperammonemia in newborns. We experienced a case of a 3-day-old boy with OTC deficiency who appeared healthy in the first 2 days of life but developed lethargy and seizure soon afterwards. His serum ammonia level was measured as >1700 µg/dL (rang...

Journal: :Nature Biotechnology 2013

Journal: :Archives of neurology 2007
William T Hu Orhun H Kantarci J Lawrence Merritt Pamela McGrann P James B Dyck Claudia F Lucchinetti Maja Tippmann-Peikert

BACKGROUND Neurological complications following bariatric surgery are rare. Whereas nutritional deficiencies are the most common cause of neurological symptoms, the unmasking of previously subclinical metabolic disorders can also lead to significant morbidity. OBJECTIVE To characterize the clinical presentation, serum biochemical fluctuations, and functional enzymatic analysis of a case of fu...

Journal: :Molecular medicine 1999
K P Zimmer M Bendiks M Mori E Kominami M B Robinson X Ye J M Wilson

BACKGROUND The mouse strain sparse fur with abnormal skin and hair (spf(ash)) is a model for the human ornithine transcarbamylase (OTC) deficiency, an X-linked inherited urea cycle disorder. The spf(ash) mouse carries a single base-pair mutation in the OTC gene that leads to the production of OTC enzyme at 10% of the normal level. MATERIALS AND METHODS Recombinant adenoviruses carrying either...

Journal: :The Journal of nutrition 2010
Guillermo Esteban-Pretel M Pilar Marín Francisco Cabezuelo Verónica Moreno Jaime Renau-Piqueras Joaquín Timoneda Teresa Barber

Chronic vitamin A deficiency induces a substantial delay in the rates of weight and height gain in both humans and experimental animals. This effect has been associated with an impaired nutrient metabolism and loss of body protein. Therefore, we analyzed the effect of vitamin A deficiency on endogenous proteolysis and nitrogen metabolism and its reversibility with all-trans retinoic acid (RA). ...

2014
R. Magesh C. George Priya Doss

Ornithine transcarbamylase (OTC) (E.C. 2.1.3.3) is one of the enzymes in the urea cycle, which involves in a sequence of reactions in the liver cells. During protein assimilation in our body surplus nitrogen is made, this open nitrogen is altered into urea and expelled out of the body by kidneys, in this cycle OTC helps in the conversion of free toxic nitrogen into urea. Ornithine transcarbamyl...

2015
Sergio de Cima Luis M. Polo Carmen Díez-Fernández Ana I. Martínez Javier Cervera Ignacio Fita Vicente Rubio

Human carbamoyl phosphate synthetase (CPS1), a 1500-residue multidomain enzyme, catalyzes the first step of ammonia detoxification to urea requiring N-acetyl-L-glutamate (NAG) as essential activator to prevent ammonia/amino acids depletion. Here we present the crystal structures of CPS1 in the absence and in the presence of NAG, clarifying the on/off-switching of the urea cycle by NAG. By bindi...

2016
Caroline Unsinn Anibh Das Vassili Valayannopoulos Eva Thimm Skadi Beblo Alberto Burlina Vassiliki Konstantopoulou Sebene Mayorandan Pascale de Lonlay Jörg Rennecke Jens Derbinski Georg F. Hoffmann Johannes Häberle

BACKGROUND Urea cycle disorders (UCDs) are rare inherited metabolic defects of ammonia detoxification. In about half of patients presenting with a UCD, the first symptoms appear within a few days after birth. These neonatal onset patients generally have a severe defect of urea cycle function and their survival and outcome prognoses are often limited. To understand better the current situation o...

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