Literature DB >> 35859960

A Case of Carnitine Palmitoyltransferase II Deficiency in Bahrain With a Novel Mutation.

Zahra Alsahlawi1,2, Zainab Fadhul1, Ali Mahmood3, Ali Mohamed4, Mohamed Khalil3, Emtithal Aljishi1,2.   

Abstract

Carnitine palmitoyltransferase II (CPT II) deficiency is a rare genetic metabolic disorder. Three forms of the disease have been described: the lethal neonatal form, the severe infantile hepatocardiomuscular form, and the myopathic form. We report a case of the infantile form of CPT II deficiency with a novel mutation. Our patient is a seven-year-old Bahraini male who was investigated by the pediatric metabolic team following the sudden death of his twin sister in infancy. A fatty acid metabolic disorder was suspected based on his echocardiogram and tandem mass spectrometry (TMS) findings. Genetic analysis was initially inconclusive. Nonetheless, he was started on a fat-free diet, L-carnitine, and medium-chain triglycerides (MCT). At nearly two years of age, the patient had a metabolic crisis precipitated by a viral illness. TMS during this time was consistent with CPT II deficiency. Sanger sequencing then identified the presence of the variant c.161T>G (p.ille54Ser) in a homozygous state, confirming the diagnosis. Although this mutation has not been reported before in previous literature concerning CPT II deficiency, it is extremely likely that this mutation is pathogenic. Although the initial work-up of the patient was inconclusive, our clinical judgment was paramount in managing the patient.
Copyright © 2022, Alsahlawi et al.

Entities:  

Keywords:  carnitine palmitoyltransferase ii deficiency; l-carnitine; metabolic disease; novel mutation; pediatric genetic disease; preimplantation genetic diagnosis

Year:  2022        PMID: 35859960      PMCID: PMC9288837          DOI: 10.7759/cureus.26043

Source DB:  PubMed          Journal:  Cureus        ISSN: 2168-8184


  21 in total

1.  Lethal neonatal and severe late infantile forms of carnitine palmitoyltransferase II deficiency associated with compound heterozygosity for different protein truncation mutations.

Authors:  Georgirene D Vladutiu; Elizabeth J Quackenbush; Bryan E Hainline; Simone Albers; David S Smail; Michael J Bennett
Journal:  J Pediatr       Date:  2002-11       Impact factor: 4.406

2.  Severe infantile type of carnitine palmitoyltransferase II (CPT II) deficiency due to homozygous R503C mutation.

Authors:  R Spiegel; A Shaag; A Gutman; S H Korman; A Saada; O Elpeleg; S A Shalev
Journal:  J Inherit Metab Dis       Date:  2007-02-15       Impact factor: 4.982

Review 3.  Inborn Errors of Metabolism with Myopathy: Defects of Fatty Acid Oxidation and the Carnitine Shuttle System.

Authors:  Areeg El-Gharbawy; Jerry Vockley
Journal:  Pediatr Clin North Am       Date:  2017-12-28       Impact factor: 3.278

Review 4.  Carnitine palmitoyltransferase deficiencies.

Authors:  J P Bonnefont; F Demaugre; C Prip-Buus; J M Saudubray; M Brivet; N Abadi; L Thuillier
Journal:  Mol Genet Metab       Date:  1999-12       Impact factor: 4.797

5.  Muscle carnitine palmityltransferase deficiency and myoglobinuria.

Authors:  S DiMauro; P M DiMauro
Journal:  Science       Date:  1973-11-20       Impact factor: 47.728

Review 6.  Carnitine palmitoyltransferases 1 and 2: biochemical, molecular and medical aspects.

Authors:  Jean-Paul Bonnefont; Fatima Djouadi; Carina Prip-Buus; Stephanie Gobin; Arnold Munnich; Jean Bastin
Journal:  Mol Aspects Med       Date:  2004 Oct-Dec

7.  Selective screening for fatty acid oxidation disorders by tandem mass spectrometry: difficulties in practical discrimination.

Authors:  Yosuke Shigematsu; Satoko Hirano; Ikue Hata; Yukie Tanaka; Masakatsu Sudo; Tsuyoshi Tajima; Nobuo Sakura; Seiji Yamaguchi; Masaki Takayanagi
Journal:  J Chromatogr B Analyt Technol Biomed Life Sci       Date:  2003-07-15       Impact factor: 3.205

8.  Effect of supplementation with L-carnitine at a small dose on acylcarnitine profiles in serum and urine and the renal handling of acylcarnitines in a patient with multiple acyl-coenzyme A dehydrogenation defect.

Authors:  Makoto Yoshino; Yasuykuki Tokunaga; Yoriko Watanabe; Ichiro Yoshida; Miki Sakaguchi; Ikue Hata; Yosuke Shigematsu; Masahiko Kimura; Seiji Yamaguchi
Journal:  J Chromatogr B Analyt Technol Biomed Life Sci       Date:  2003-07-15       Impact factor: 3.205

9.  Neonatal Carnitine Palmitoyltransferase II Deficiency: A Lethal Entity.

Authors:  Sushma Malik; Ashutosh Abhimanyu Paldiwal; Charusheela Sujit Korday; Shruti Sudhir Jadhav
Journal:  J Clin Diagn Res       Date:  2015-10-01

10.  Two CPT2 mutations in three Japanese patients with carnitine palmitoyltransferase II deficiency: functional analysis and association with polymorphic haplotypes and two clinical phenotypes.

Authors:  K Wataya; J Akanuma; P Cavadini; Y Aoki; S Kure; F Invernizzi; I Yoshida; J Kira; F Taroni; Y Matsubara; K Narisawa
Journal:  Hum Mutat       Date:  1998       Impact factor: 4.878

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