Genomic mutations associated with mild and severe deficiencies of transcobalamin I (haptocorrin) that cause mildly and severely low serum cobalamin levels.
نویسندگان
چکیده
Transcobalamin (TC) I deficiency, like the function of TC I itself, is incompletely understood. It produces low serum cobalamin levels indistinguishable from those of true cobalamin deficiency. Diagnosis is especially elusive when TC I deficiency is mild. To provide new, more substantive definition, the TCN1 gene was examined in two well-characterised families that included members with both severe and mild TC I deficiencies. A severely deficient proposita with undetectable TC I levels displayed compound heterozygosity for two mutations, each causing a premature stop codon. Relatives in both families who had mildly low or low-normal plasma levels of TC I and cobalamin were heterozygous for one or the other of these mutations. An unrelated patient with mild TC I deficiency and unknown familial TC I and cobalamin status was then tested and found to be similarly heterozygous for one of the mutations. The two nonprivate mutations identify a genetic basis for TC I deficiency for the first time. They also add new approaches to studying mild and severe TC I deficiency and to reducing confusion of its low cobalamin levels with those of cobalamin deficiency and its often dramatically different prognosis and management.
منابع مشابه
Mild transcobalamin I (haptocorrin) deficiency and low serum cobalamin concentrations.
BACKGROUND Low cobalamin concentrations are common, but their causes are often unknown. Transcobalamin I/haptocorrin (TC I/HC) deficiency, viewed as a rare cause, has not been examined systematically in patients with unexplained low serum cobalamin. METHODS Total TC I/HC was measured by RIA in three subgroups of 367, 160, and 38 patients with different categories of low cobalamin concentratio...
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BACKGROUND Two proteins carry vitamin B12 in plasma. Transcobalamin (TC) carries approximately 25% of total plasma vitamin B12 and is 6% to 20% saturated with cobalamin. Haptocorrin (HC) binds approximately 80% of total cobalamin and is largely saturated with cobalamin. METHODS We investigated the distribution and the relationship between concentrations of cobalamin, total and holo forms of T...
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ورودعنوان ژورنال:
- British journal of haematology
دوره 147 3 شماره
صفحات -
تاریخ انتشار 2009