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CPT2 gene mutations resulting in lethal neonatal or severe infantile carnitine palmitoyltransferase II deficiency

  • Paul J. Isackson
  • , Michael J. Bennett
  • , Uta Lichter-Konecki
  • , Mary Willis
  • , William L. Nyhan
  • , V. Reid Sutton
  • , Ingrid Tein
  • , Georgirene D. Vladutiu
  • SUNY Buffalo
  • University of Pennsylvania
  • George Washington University
  • University of California at San Diego
  • Baylor College of Medicine
  • University of Toronto

Research output: Contribution to journal › Article › peer-review

56 Scopus citations

Abstract

Three distinct clinical manifestations of carnitine palmitoyltransferase II (CPT II) deficiency have been defined including a mild adult onset myopathy, a severe infantile disorder and a lethal neonatal form. In this study we have examined the genomic DNA of five patients, 3 with the lethal neonatal form and 2 with the severe infantile form of the disease and identified two disease-causing mutations in the CPT2 gene for each patient, three of which are novel. In addition, based on currently available structural, biochemical and clinical data, we have classified all 64 known disease-causing mutations into groups with different predicted phenotypes depending on their CPT2 allelic counterparts.

Original languageEnglish
Pages (from-to)422-427
Number of pages6
JournalMolecular Genetics and Metabolism
Volume94
Issue number4
DOIs
StatePublished - Aug 2008

Keywords

  • Expanded newborn screening
  • Fatty acid oxidation defect
  • Genotype/phenotype correlation
  • Molecular diagnostics

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