Phenylalanine Hydroxylase Deficiency and Citrin Deficiency in a Chinese Infant

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Tetrahydrobiopterin responsiveness of patients with phenylalanine hydroxylase deficiency.

OBJECTIVE To identify patients responsive to tetrahydrobiopterin (BH4) in a sample of Brazilians with hyperphenylalaninemia due to phenylalanine hydroxylase deficiency (HPA-PAH). METHODS Interventional study, convenience sampling. The inclusion criteria were: diagnosis of HPA-PAH; age ≥ 7 years; phenylalanine-restricted diet and phenylalanine (Phe) levels ≥ 6 mg/dL in all blood tests 1 year b...

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Molecular genetics of tetrahydrobiopterin-responsive phenylalanine hydroxylase deficiency.

Mutations in the phenylalanine hydroxylase (PAH) gene result in phenylketonuria (PKU). Tetrahydrobiopterin (BH(4))-responsive hyperphenylalaninemia has been recently described as a variant of PAH deficiency caused by specific mutations in the PAH gene. It has been suggested that BH(4)-responsiveness may be predicted from the corresponding genotypes. Data from BH(4) loading tests indicated an in...

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Six cases of citrin deficiency in Korea.

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Genotypic and phenotypic features of citrin deficiency: five-year experience in a Chinese pediatric center.

Citrin is a liver-type aspartate/glutamate carrier (AGC) encoded by the gene SLC25A13. Two phenotypes for human citrin deficiency have been described, namely the adult-onset citrullinemia type II (CTLN2) and the neonatal intrahepatic cholestasis caused by citrin deficiency (NICCD). However, citrin deficiency currently remains a perplexing and poorly recognized disorder. In particular, descripti...

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Intrahepatic Cholestasis Secondary to Citrin Deficiency

Citrin deficiency (neonatal-onset type II citrullinemia), is an autosomal recessive metabolic disorder, caused by a mutation of the SLC25A13 gene. The defective transport between the mitochondria and the cytosol, leads to insufficient substrate for argininosuccinate synthetase (ASS) and secondary functional deficiency of ASS activity [1]. Infants with citrin deficiency, have transient intrahepa...

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ژورنال

عنوان ژورنال: Chinese Medical Journal

سال: 2015

ISSN: 0366-6999

DOI: 10.4103/0366-6999.168084