Literature DB >> 6696649

Familial systemic carnitine deficiency.

R P Cruse, S Di Mauro, J Towfighi, C Trevisan.   

Abstract

Two sisters with systemic carnitine deficiency showed the heterogenicity of this condition and a lack of correlation between measurable carnitine levels and clinical manifestations. One child experienced recurrent metabolic encephalopathy, which was diagnosed as Reye's syndrome. The older child, despite lower carnitine concentrations, was asymptomatic. Lipid inclusion myopathy was shown in both children. Serum, muscle, and liver carnitine levels were reduced. The parents had normal serum carnitine concentrations. Both children had significantly abnormal renal handling of carnitine, and their mother, a mild alteration. This family provided evidence for genetic transmission and presumed autosomal recessive inheritance in this disease. Both children were treated with carnitine without clear benefit; no side effects were noted. Previously reported cases of systemic carnitine deficiency were reviewed.

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Year:  1984        PMID: 6696649     DOI: 10.1001/archneur.1984.04050150079021

Source DB:  PubMed          Journal:  Arch Neurol        ISSN: 0003-9942


  5 in total

1.  Multiple acyl-CoA dehydrogenation deficiency as decreased acyl-carnitine profile in serum.

Authors:  Bing Wen; Duoling Li; Wei Li; Yuying Zhao; Chuanzhu Yan
Journal:  Neurol Sci       Date:  2015-04-01       Impact factor: 3.307

2.  Familial carnitine deficiency: further evidence for autosomal recessive transmission with variable expression.

Authors:  E Shahar; N Brand; Y Shapira; V Barash; A Gutman
Journal:  J Neurol Neurosurg Psychiatry       Date:  1988-02       Impact factor: 10.154

Review 3.  Medium chain acyl-CoA dehydrogenase deficiency.

Authors:  E H Touma; C Charpentier
Journal:  Arch Dis Child       Date:  1992-01       Impact factor: 3.791

4.  Left ventricular noncompaction in primary systemic carnitine deficiency: A rare association.

Authors:  Deepanjan Bhattacharya; Deepa Sasikumar; Harikrishnan Kurup; K M Krishnamoorthy
Journal:  Ann Pediatr Cardiol       Date:  2020-11-19

Review 5.  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

  5 in total

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