Therapeutic effect of

Acute therapy Carbamyl phosphate synthetase 1 deficiency Hyperammonemia Liver transplantation N-carbamylglutamate Urea cycle disorder

Journal

Molecular genetics and metabolism reports
ISSN: 2214-4269
Titre abrégé: Mol Genet Metab Rep
Pays: United States
ID NLM: 101624422

Informations de publication

Date de publication:
Sep 2020
Historique:
received: 28 04 2020
revised: 24 06 2020
accepted: 24 06 2020
entrez: 17 7 2020
pubmed: 17 7 2020
medline: 17 7 2020
Statut: epublish

Résumé

The detoxification of ammonia to urea requires a functional hepatic urea cycle, which consists of six enzymes and two mitochondrial membrane transporters. The initial step of the urea cycle is catalyzed by carbamyl phosphate synthetase 1 (CPS1). CPS1 deficiency (CPS1D) is a rare autosomal recessive disorder.

Identifiants

pubmed: 32670798
doi: 10.1016/j.ymgmr.2020.100622
pii: S2214-4269(20)30068-9
pii: 100622
pmc: PMC7347628
doi:

Types de publication

Case Reports

Langues

eng

Pagination

100622

Informations de copyright

© 2020 Published by Elsevier Inc.

Déclaration de conflit d'intérêts

Authors declare no conflict of interest.

Références

Eur J Pediatr. 1985 Jan;143(3):196-7
pubmed: 3987713
Mol Genet Metab. 2014 Jun;112(2):123-32
pubmed: 24813853
Orphanet J Rare Dis. 2016 Aug 19;11(1):116
pubmed: 27538463
Eur J Pediatr. 2003 Jun;162(6):410-6
pubmed: 12684900
J Pediatr. 2014 Aug;165(2):401-403.e3
pubmed: 24880889
JIMD Rep. 2019 May 02;48(1):36-44
pubmed: 31392111
Orphanet J Rare Dis. 2017 Jun 15;12(1):111
pubmed: 28619060
J Pediatr. 2005 Aug;147(2):260-2
pubmed: 16126063
Am J Med Genet. 1991 May 1;39(2):228-9
pubmed: 2063931
Hum Mutat. 2013 Aug;34(8):1149-59
pubmed: 23649895
Sci Rep. 2015 Nov 23;5:16950
pubmed: 26592762
Orphanet J Rare Dis. 2014 Sep 02;9:130
pubmed: 25205257
J Inherit Metab Dis. 2012 Sep;35(5):777-85
pubmed: 22167275
Orphanet J Rare Dis. 2012 May 29;7:32
pubmed: 22642880
J Hum Genet. 2007;52(4):349-354
pubmed: 17310273
Expert Opin Ther Targets. 2017 Apr;21(4):391-399
pubmed: 28281899

Auteurs

Yohei Sugiyama (Y)

Center for Medical Genetics, Chiba Children's Hospital, 579-1 Heta-cho, Midori-ku, Chiba city, Chiba 266-0007, Japan.
Department of Metabolism, Chiba Children's Hospital, 579-1 Heta-cho, Midori-ku, Chiba city, Chiba 266-0007, Japan.
Department of Neonatology, Chiba Children's Hospital, 579-1 Heta-cho, Midori-ku, Chiba city, Chiba 266-0007, Japan.

Masaru Shimura (M)

Center for Medical Genetics, Chiba Children's Hospital, 579-1 Heta-cho, Midori-ku, Chiba city, Chiba 266-0007, Japan.
Department of Metabolism, Chiba Children's Hospital, 579-1 Heta-cho, Midori-ku, Chiba city, Chiba 266-0007, Japan.

Minako Ogawa-Tominaga (M)

Department of Metabolism, Chiba Children's Hospital, 579-1 Heta-cho, Midori-ku, Chiba city, Chiba 266-0007, Japan.

Tomohiro Ebihara (T)

Center for Medical Genetics, Chiba Children's Hospital, 579-1 Heta-cho, Midori-ku, Chiba city, Chiba 266-0007, Japan.
Department of Neonatology, Chiba Children's Hospital, 579-1 Heta-cho, Midori-ku, Chiba city, Chiba 266-0007, Japan.

Yoshina Kinouchi (Y)

Center for Medical Genetics, Chiba Children's Hospital, 579-1 Heta-cho, Midori-ku, Chiba city, Chiba 266-0007, Japan.
Department of Neonatology, Chiba Children's Hospital, 579-1 Heta-cho, Midori-ku, Chiba city, Chiba 266-0007, Japan.

Keitaro Isozaki (K)

Center for Medical Genetics, Chiba Children's Hospital, 579-1 Heta-cho, Midori-ku, Chiba city, Chiba 266-0007, Japan.
Department of Neonatology, Chiba Children's Hospital, 579-1 Heta-cho, Midori-ku, Chiba city, Chiba 266-0007, Japan.

Tetsuro Matsuhashi (T)

Center for Medical Genetics, Chiba Children's Hospital, 579-1 Heta-cho, Midori-ku, Chiba city, Chiba 266-0007, Japan.
Department of Metabolism, Chiba Children's Hospital, 579-1 Heta-cho, Midori-ku, Chiba city, Chiba 266-0007, Japan.

Makiko Tajika (M)

Center for Medical Genetics, Chiba Children's Hospital, 579-1 Heta-cho, Midori-ku, Chiba city, Chiba 266-0007, Japan.
Department of Metabolism, Chiba Children's Hospital, 579-1 Heta-cho, Midori-ku, Chiba city, Chiba 266-0007, Japan.

Takuya Fushimi (T)

Center for Medical Genetics, Chiba Children's Hospital, 579-1 Heta-cho, Midori-ku, Chiba city, Chiba 266-0007, Japan.
Department of Metabolism, Chiba Children's Hospital, 579-1 Heta-cho, Midori-ku, Chiba city, Chiba 266-0007, Japan.

Keiko Ichimoto (K)

Center for Medical Genetics, Chiba Children's Hospital, 579-1 Heta-cho, Midori-ku, Chiba city, Chiba 266-0007, Japan.
Department of Metabolism, Chiba Children's Hospital, 579-1 Heta-cho, Midori-ku, Chiba city, Chiba 266-0007, Japan.

Ayako Matsunaga (A)

Center for Medical Genetics, Chiba Children's Hospital, 579-1 Heta-cho, Midori-ku, Chiba city, Chiba 266-0007, Japan.
Department of Metabolism, Chiba Children's Hospital, 579-1 Heta-cho, Midori-ku, Chiba city, Chiba 266-0007, Japan.

Tomoki Ishida (T)

Department of Neonatology, Kimitsu Chuo Hospital, 1010 sakurai, Kisarazu city, Chiba 292-8535, Japan.

Kayo Mizutani (K)

Department of Neonatology, Kameda Medical Center, 929 Higashi-cho, Kamogawa City, Chiba 296-8602, Japan.

Tomoko Tsuruoka (T)

Center for Medical Genetics, Chiba Children's Hospital, 579-1 Heta-cho, Midori-ku, Chiba city, Chiba 266-0007, Japan.
Department of Neonatology, Chiba Children's Hospital, 579-1 Heta-cho, Midori-ku, Chiba city, Chiba 266-0007, Japan.

Kei Murayama (K)

Center for Medical Genetics, Chiba Children's Hospital, 579-1 Heta-cho, Midori-ku, Chiba city, Chiba 266-0007, Japan.
Department of Metabolism, Chiba Children's Hospital, 579-1 Heta-cho, Midori-ku, Chiba city, Chiba 266-0007, Japan.

Classifications MeSH