Literature DB >> 23184072

Clinically symptomatic heterozygous carnitine palmitoyltransferase II (CPT II) deficiency.

Pushpa Raj Joshi1, Marcus Deschauer, Stephan Zierz.   

Abstract

Two symptomatic patients with heterozygous carnitine palmitoyltransferase II (CPT II) deficiency are reported. Patient 1, a 21-year-old female professional tennis player, suffered from exercise-induced attacks of muscle pain, burning sensations and proximal weakness. Patient 2, a 30-year-old male amateur marathon runner developed muscle cramps and rhabdomyolysis upon extensive exercise and insolation-induced fever. In both patients, the common p.S113L mutation was found in heterozygote state. No second mutation could be found upon sequencing of all the exons of CPT2 gene including exon-intron boundaries. Biochemically, residual CPT activity in muscle homogenate upon inhibition by malonyl-CoA and Triton-X-100 was intermediate between controls and patients with mutations on both alleles. Although CPT II deficiency is an autosomal recessive disorder, the reported patients indicate that heterozygotes might also have typical attacks of myalgia, pareses or rhabdomyolysis.

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Year:  2012        PMID: 23184072     DOI: 10.1007/s00508-012-0296-9

Source DB:  PubMed          Journal:  Wien Klin Wochenschr        ISSN: 0043-5325            Impact factor:   1.704


  20 in total

1.  Genotype-phenotype correlations in a large series of patients with muscle type CPT II deficiency.

Authors:  Angelica Anichini; Marina Fanin; Christine Vianey-Saban; Denise Cassandrini; Chiara Fiorillo; Claudio Bruno; Corrado Angelini
Journal:  Neurol Res       Date:  2010-08-31       Impact factor: 2.448

2.  Characterization of compound missense mutation and deletion of carnitine palmitoyltransferase II in a patient with adenovirus-associated encephalopathy.

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Journal:  J Med Invest       Date:  2011-08

Review 3.  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

4.  Clinical and biochemical heterogeneity in an Italian family with CPT II deficiency due to Ser 113 Leu mutation.

Authors:  Mubeen F Rafay; E Gordon Murphy; J Denis McGarry; Petra Kaufmann; Salvatore DiMauro; Ingrid Tein
Journal:  Can J Neurol Sci       Date:  2005-08       Impact factor: 2.104

5.  Muscle carnitine palmitoyltransferase II deficiency: clinical and molecular genetic features and diagnostic aspects.

Authors:  Marcus Deschauer; Thomas Wieser; Stephan Zierz
Journal:  Arch Neurol       Date:  2005-01

Review 6.  The syndrome of rhabdomyolysis: complications and treatment.

Authors:  Yiannis S Chatzizisis; Gesthimani Misirli; Apostolos I Hatzitolios; George D Giannoglou
Journal:  Eur J Intern Med       Date:  2008-04-28       Impact factor: 4.487

7.  Malignant hyperthermia-like syndrome and carnitine palmitoyltransferase II deficiency with heterozygous R503C mutation.

Authors:  Kirk J Hogan; Georgirene D Vladutiu
Journal:  Anesth Analg       Date:  2009-10       Impact factor: 5.108

8.  cDNA cloning, sequence analysis, and chromosomal localization of the gene for human carnitine palmitoyltransferase.

Authors:  G Finocchiaro; F Taroni; M Rocchi; A L Martin; I Colombo; G T Tarelli; S DiDonato
Journal:  Proc Natl Acad Sci U S A       Date:  1991-01-15       Impact factor: 11.205

9.  Biochemical evidence for heterozygosity in muscular carnitine palmitoyltransferase deficiency.

Authors:  S Zierz; R R Mundegar; F Jerusalem
Journal:  Clin Investig       Date:  1993-12

10.  Identification of a common mutation in the carnitine palmitoyltransferase II gene in familial recurrent myoglobinuria patients.

Authors:  F Taroni; E Verderio; F Dworzak; P J Willems; P Cavadini; S DiDonato
Journal:  Nat Genet       Date:  1993-07       Impact factor: 38.330

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Journal:  J Hum Genet       Date:  2018-12-04       Impact factor: 3.172

2.  Expanding mutation spectrum in CPT II gene: identification of four novel mutations.

Authors:  Pushpa Raj Joshi; Peter Young; Marcus Deschauer; Stephan Zierz
Journal:  J Neurol       Date:  2013-03-09       Impact factor: 4.849

Review 3.  Muscle Carnitine Palmitoyltransferase II Deficiency: A Review of Enzymatic Controversy and Clinical Features.

Authors:  Diana Lehmann; Leila Motlagh; Dina Robaa; Stephan Zierz
Journal:  Int J Mol Sci       Date:  2017-01-03       Impact factor: 5.923

4.  Metabolic autopsy with next generation sequencing in sudden unexpected death in infancy: Postmortem diagnosis of fatty acid oxidation disorders.

Authors:  Takuma Yamamoto; Hiroyuki Mishima; Hajime Mizukami; Yuki Fukahori; Takahiro Umehara; Takehiko Murase; Masamune Kobayashi; Shinjiro Mori; Tomonori Nagai; Tatsushige Fukunaga; Seiji Yamaguchi; Koh-Ichiro Yoshiura; Kazuya Ikematsu
Journal:  Mol Genet Metab Rep       Date:  2015-10-02

5.  Unrecognized High Occurrence of Genetically Confirmed Hereditary Carnitine Palmitoyltransferase II Deficiency in an Austrian Family Points to the Ongoing Underdiagnosis of the Disease.

Authors:  Christina Zach; Karl Unterkofler; Peter Fraunberger; Heinz Drexel; Axel Muendlein
Journal:  Front Genet       Date:  2019-05-22       Impact factor: 4.599

6.  Infantile onset carnitine palmitoyltransferase 2 deficiency: Cortical polymicrogyria, schizencephaly, and gray matter heterotopias in an adolescent with normal development.

Authors:  Ivan Shelihan; Elsa Rossignol; Jean-Claude Décarie; Jean-Paul Bonnefont; Michèle Brivet; Catherine Brunel-Guitton; Grant A Mitchell
Journal:  JIMD Rep       Date:  2021-09-29

Review 7.  Muscle Carnitine Palmitoyltransferase II (CPT II) Deficiency: A Conceptual Approach.

Authors:  Pushpa Raj Joshi; Stephan Zierz
Journal:  Molecules       Date:  2020-04-13       Impact factor: 4.411

  7 in total

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