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PMID: 3998948 Published · ppublish English Case Reports Journal Article

Medium-chain acyl-CoA dehydrogenase deficiency in two siblings with a Reye-like syndrome.

The Journal of pediatrics ·Vol. 106 ·No. 6 ·1985-06-00 ·Pages 918-21

Bougnères PF, Rocchiccioli F, Kølvraa S, Hadchouel M, Lalau-Keraly J, Chaussain JL, Wadman SK, Gregersen N

Abstract

An increasing number of reports indicate that patients with some inherited metabolic diseases may have symptoms resembling those of Reye syndrome. We describe two siblings who developed a Reye-like syndrome at ages 16 and 18 months, respectively, after a viral illness and salicylate therapy. Both had fasting hypoglycemia and hypoketonemia. At the time of the acute episode and after ingestion of a medium-chain triglyceride load, one of them excreted large amounts of abnormal metabolites derived from the omega- and (omega-1)-oxidation of medium-chain fatty acids. Medium-chain acyl-CoA dehydrogenase activity was lower than 20% of control values in fibroblasts from both patients. This enzyme defect should be considered in children with a Reye-like syndrome with these distinctive manifestations.

MeSH Terms
Acyl-CoA Dehydrogenase Acyl-CoA Dehydrogenases/deficiency,genetics,urine Blood Glucose/metabolism Caprylates/blood Carnitine/metabolism Fatty Acids, Nonesterified/metabolism Female Fibroblasts/enzymology Humans Infant Liver/enzymology,pathology Male Reye Syndrome/enzymology,genetics,pathology
Chemicals
Blood Glucose Caprylates Fatty Acids, Nonesterified Acyl-CoA Dehydrogenases Acyl-CoA Dehydrogenase octanoic acid Carnitine
Authors & Affiliations
8 authors, click to expand affiliations / ORCID
Bougnères P F
Rocchiccioli F
Kølvraa S
Hadchouel M
Lalau-Keraly J
Chaussain J L
Wadman S K
Gregersen N
Article Info
Journal
The Journal of pediatrics
Abbr.
J Pediatr
ISSN
0022-3476
Published
1985-06-00
Pages
918-21
Language
English
Region
United States
NLM ID
0375410
Subset
IM
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