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PMID: 7351973 Published · ppublish English Journal Article Research Support, U.S. Gov't, P.H.S.

Cerebral dysfunction in asymptomatic carriers of ornithine transcarbamylase deficiency.

The New England journal of medicine ·Vol. 302 ·No. 9 ·1980-02-28 ·Pages 482-5

Batshaw ML, Roan Y, Jung AL, Rosenberg LA, Brusilow SW

Abstract

We tested the hypothesis that female carriers of ornithine transcarbamylase (OTC) deficiency have cerebral dysfunction as a consequence of episodic hyperammonemia. Seven such carriers were identified by pedigree analysis and protein-tolerance testing. The Wechsler Adult Intelligence Scale was used for evaluation. All IG scores were above 100, but in the protein-tolerant siblings, the full-scale and performance IQ scores were higher by 5.6 and 9.9 points, respectively (P less than 0.02). The absolute difference between the verbal and performance IQ scores was greater in the OTC carriers by 7.8 points (P less than 0.01). This study suggests that clinically asymptomatic carriers of OTC deficiency may have intellectual deficits, perhaps as a result of episodic hyperammonemia.

MeSH Terms
Amino Acid Metabolism, Inborn Errors/genetics,metabolism,physiopathology Ammonia/blood Brain/physiopathology Dietary Proteins/metabolism Female Genetic Carrier Screening Heterozygote Humans Infant Infant, Newborn Male Ornithine Carbamoyltransferase Deficiency Disease Orotic Acid/urine Pedigree Wechsler Scales
Chemicals
Dietary Proteins Orotic Acid Ammonia
Authors & Affiliations
5 authors, click to expand affiliations / ORCID
Batshaw M L
Roan Y
Jung A L
Rosenberg L A
Brusilow S W
Article Info
Journal
The New England journal of medicine
Abbr.
N Engl J Med
ISSN
0028-4793
Published
1980-02-28
Pages
482-5
Language
English
Region
United States
NLM ID
0255562
Subset
IM
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