A severe genotype with favourable outcome in very long chain acyl-CoA dehydrogenase deficiency.

Touma, E H; Rashed, M S; Vianey-Saban, C; Sakr, A; Divry, P; Gregersen, N; Andresen, B S · Arch Dis Child · 2001

case_report · Level V

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Abstract

A patient with very long chain acyl-CoA dehydrogenase (VLCAD) deficiency is reported. He had a severe neonatal presentation and cardiomyopathy. He was found to be homozygous for a severe mutation with no residual enzyme activity. Tandem mass spectrometry on dried blood spots revealed increased long chain acylcarnitines. VLCAD enzyme activity was severely decreased to 2% of control levels. Dietary management consisted of skimmed milk supplemented with medium chain triglycerides and L-carnitine. Outcome was good and there was no acute recurrence.

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