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Thiamine Triphosphate Levels and HistopathologyCorrelation in Leigh Disease
Jonathan H. Pincus, MD;
Gilbert B. Solitare, MD;
Jack R. Cooper, PhD
Arch Neurol. 1976;33(11):759-763.
Abstract
• Thiamine and thiamine triphosphate (TTP) values were assayed in various brain regions in 11 controls and 13 patients with subacute necrotizing encephalomyelopathy (SNE, Leigh disease). The TTP values of normal brain were 5% of the total thiamine value. The relative TTP (or % TTP) level was consistently low in the pons, midbrain, and cerebellum of all the SNE brains. Twenty-five percent of the SNE brains had normal TTP levels in the frontal region. The TTP values correlated with the degrees of pathologic involvement in all sampled regions of the brain except the cerebellum. The concentration of thiamine in the mammillary bodies exceeded its concentration elsewhere in both control and SNE brains. The finding of low TTP levels in morphologically abnormal regions supports the hypothesis that TTP deficiency is etiologically related to SNE.
Author Affiliations
From the Departments of Neurology (Dr Pincus) and Pharmacology (Dr Cooper), Yale University School of Medicine, and the Department of Pathology (Dr Solitare), Hospital of St Raphael, New Haven, Conn.
Footnotes
Accepted for publication March 24, 1976.
Reprint requests to Department of Neurology, Yale University School of Medicine, 333 Cedar St, New Haven, CT 06510 (Dr Pincus).
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