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Original Article |
-tocopherol metabolites in
-tocopherol transfer protein-deficient patients
Correspondence to: Regina Brigelius-Flohé
Patients with
-tocopherol transfer protein (
-TTP) defects experience neurological symptoms characteristic of vitamin E deficiency and depend on continuous high
-tocopherol supplements. We investigated the excretion of 2,5,7,8-tetramethyl-2(2'-carboxyethyl)-6-hydroxychroman (
-CEHC), a urinary metabolite of
-tocopherol, as a putative marker for the
-tocopherol status of
-TTP-deficient patients and control subjects. In three patients vitamin E supplementation was stopped for short periods of time, during which plasma
-tocopherol concentrations and urinary
-CEHC excretion were measured. In the patients, plasma
-tocopherol decreased below normal (<5 µmol/l) but
-CEHC excretion remained above the range of unsupplemented control subjects (0.118;0.306 mg/day, n = 6). In healthy subjects, however,
-CEHC excretion was increased only after surpassing a plasma
-tocopherol threshold of 30;40 µmol/l. Such a threshold did not exist in patients. The general mechanism of
-tocopherol degradation did not appear to differ between patients and control subjects.
The presumed mechanism of
- and subsequent ß-oxidation was supported by the detection of
-CPHC, an
-CEHC homolog with a side chain longer by 3 carbon atoms, both in supplemented patients and in control subjects. Schuelke, M., A. Elsner, B. Finckh, A. Kohlschütter, C. Hübner, and R. Brigelius-Flohé. Urinary
-tocopherol metabolites in
-tocopherol transfer protein-deficient patients. J. Lipid Res. 2000. 41: 1543;1551.
Supplementary key words:
-tocopherol,
-tocopherol metabolites,
-CEHC,
-tocopherol transfer protein, AVED patients
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