Erythropoiesis and iron metabolism in dominant erythropoietic protoporphyria

被引:73
作者
Holme, S. Alexander
Worwood, Mark
Anstey, Alexander V.
Elder, George H.
Badminton, Michael N. [1 ]
机构
[1] Cardiff Univ, Dept Med Biochem & Immunol, Cardiff CF14 4XN, S Glam, Wales
[2] Cardiff Univ, Dept Dermatol, Cardiff, S Glam, Wales
[3] Cardiff Univ, Dept Haematol, Cardiff, S Glam, Wales
关键词
D O I
10.1182/blood-2007-04-088120
中图分类号
R5 [内科学];
学科分类号
1002 ; 100201 ;
摘要
Erythropoietic protoporphyria (EPP) results from deficiency of ferrochelatase (FECH). Accumulation of protoporphyrin IX causes life-long acute photosensitivity. Microcytic anemia occurs in 20% to 60% of patients. We investigated 178 patients with dominant EPP confirmed by molecular analysis. Erythropoiesis was impaired in all patients; all had a downward shift in hemoglobin (Hb), and the mean decreased in males by 12 g/L (1.2 g/dL). By World Health Organization criteria, 48% of women and 33% of men were anemic. Iron stores, assessed by serum ferritin (sFn), were decreased by two-thirds, but normal serum soluble transferrin receptor-1 and iron concentrations suggested that erythropoiesis was not limited by iron supply. FECH deficiency in EPP appears to lead to a steady state in which decreased erythropoiesis is matched by reduced iron absorption and supply. This response may in part be mediated by protoporphyrin, but we found no correlation between erythrocyte protoporphyrin and Hb, sFn, total iron-binding capacity, or transferrin saturation.
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页码:4108 / 4110
页数:3
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