Literature DB >> 31373028

Increased risk of sudden death in untreated primary carnitine deficiency.

Jan Rasmussen1,2, Morten Dunø3, Allan M Lund3, Ulrike Steuerwald4, Steen-Holger Hansen5, Høgni D Joensen6, Lars Køber2, Olav W Nielsen7.   

Abstract

Primary carnitine deficiency (PCD) affects fatty acid oxidation and is associated with cardiomyopathy and cardiac arrhythmia, but the risk of sudden death in PCD is unknown. The Faroe Islands have a high prevalence of PCD, 1:300. This study systematically investigated a possible association between untreated PCD and sudden death in young Faroese subjects. We investigated all medico-legal cases of sudden death between 1979 and 2012 among subjects below the age of 45. Stored biomaterial was examined with molecular genetic analysis to reveal PCD. We compared the prevalence of PCD among sudden death cases with that of the background population (0.23%) to calculate the odds ratio (OR) for sudden death with PCD. Biomaterial was available and genetically analyzed from 53 of 65 sudden death cases (82%) in the Faroe Islands. Six (one male and five females) of the 53 cases were homozygous for the PCD related c.95A>G mutation-a prevalence of 11.3% (95% CI 5%-23%) and an OR of 54.3 (95% CI 21-138, P < .0001) for the association between sudden death and untreated PCD. Only 11 of the 53 sudden death cases were women-of whom five were homozygous for the c.95A>G mutation (45.5%) yielding an OR of 348.8 (95% CI 94-1287, P < .0001) for the association between sudden death and untreated PCD in females. This study showed a strong association between sudden death and untreated PCD, especially in females.
© 2019 SSIEM.

Entities:  

Keywords:  Faroe Islands; primary carnitine deficiency; sudden death

Mesh:

Substances:

Year:  2019        PMID: 31373028     DOI: 10.1002/jimd.12158

Source DB:  PubMed          Journal:  J Inherit Metab Dis        ISSN: 0141-8955            Impact factor:   4.982


  5 in total

1.  Primary carnitine deficiency - diagnosis after heart transplantation: better late than never!

Authors:  Sarah C Grünert; Sara Tucci; Anke Schumann; Meike Schwendt; Gwendolyn Gramer; Georg F Hoffmann; Michelle Erbel; Brigitte Stiller; Ute Spiekerkoetter
Journal:  Orphanet J Rare Dis       Date:  2020-04-10       Impact factor: 4.123

2.  Increased detection of primary carnitine deficiency through second-tier newborn genetic screening.

Authors:  Yiming Lin; Weifeng Zhang; Chenggang Huang; Chunmei Lin; Weihua Lin; Weilin Peng; Qingliu Fu; Dongmei Chen
Journal:  Orphanet J Rare Dis       Date:  2021-03-23       Impact factor: 4.123

Review 3.  Clinical characteristics of primary carnitine deficiency: A structured review using a case-by-case approach.

Authors:  Loek L Crefcoeur; Gepke Visser; Sacha Ferdinandusse; Frits A Wijburg; Mirjam Langeveld; Barbara Sjouke
Journal:  J Inherit Metab Dis       Date:  2022-02-03       Impact factor: 4.750

4.  Newborn screening for carnitine transporter defect in Bavaria and the long-term follow-up of the identified newborns and mothers: Assessing the benefit and possible harm based on 19 ½ years of experience.

Authors:  Katharina A Schiergens; Katharina J Weiss; Wulf Röschinger; Amelie S Lotz-Havla; Joachim Schmitt; Robert Dalla Pozza; Sarah Ulrich; Birgit Odenwald; Joachim Kreuder; Esther M Maier
Journal:  Mol Genet Metab Rep       Date:  2021-06-12

5.  Combined primary carnitine deficiency with neonatal intrahepatic cholestasis caused by citrin deficiency in a Chinese newborn.

Authors:  Yiming Lin; Weihua Lin; Yanru Chen; Chunmei Lin; Zhenzhu Zheng; Jianlong Zhuang; Qingliu Fu
Journal:  BMC Pediatr       Date:  2020-10-13       Impact factor: 2.125

  5 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.