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J Pediatr ; 148(5): 665-670, 2006 May.
Article in English | MEDLINE | ID: mdl-16737882

ABSTRACT

OBJECTIVES: To describe the clinical presentation and long-term follow-up of a large cohort of patients with medium-chain acyl-CoA dehydrogenase (MCAD) deficiency. STUDY DESIGN: A nationwide, retrospective analysis of clinical presentation and follow-up in 155 Dutch patients with MCAD deficiency. RESULTS: Most patients presented between 3 months and 5.1 years of age; 13% had symptoms as neonates not exclusively related to breast-feeding. An acute presentation before the diagnosis was made resulted in a mortality of 22% (25/114), whereas 21% (19/89) developed disabilities after the diagnosis. On follow-up, a total of 44 patients reported fatigue (35%; 28/80), muscle pain (31%; 25/80), and/or reduced exercise tolerance (39%; 31/80). Cardiac evaluation in 11 adult patients revealed no abnormalities in cardiac function explaining these complaints. Children with MCAD deficiency readily become overweight. CONCLUSIONS: Mortality and morbidity were high in undiagnosed children with MCAD deficiency; establishment of the diagnosis significantly improves outcome. Strikingly, after the diagnosis and initiation of treatment, overweight and chronic complaints (fatigue, muscle pain, and reduced exercise tolerance) were prominent.


Subject(s)
Acyl-CoA Dehydrogenase/deficiency , Metabolism, Inborn Errors/complications , Metabolism, Inborn Errors/diagnosis , Acyl-CoA Dehydrogenase/genetics , Adolescent , Adult , Child , Child, Preschool , Cohort Studies , Disease Progression , Humans , Infant , Infant, Newborn , Metabolism, Inborn Errors/genetics , Middle Aged , Mutation/genetics , Netherlands , Prognosis , Retrospective Studies , Severity of Illness Index
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