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Eur J Haematol ; 94(6): 491-7, 2015 Jun.
Artículo en Inglés | MEDLINE | ID: mdl-25251786

RESUMEN

Congenital erythropoietic porphyria (CEP) is a rare genetic disease that is characterized by a severe cutaneous photosensitivity causing unrecoverable deformities, chronic hemolytic anemia requiring blood transfusion program, and by fatal systemic complications. A correct and early diagnosis is required to develop a management plan that is appropriate to the patient's needs. Recently only one case of X-linked CEP had been reported, describing the trans-acting GATA1-R216W mutation. Here, we have characterized two novel X-linked CEP patients, both with misleading hematological phenotypes that include dyserythropoietic anemia, thrombocytopenia, and hereditary persistence of fetal hemoglobin. We compare the previously reported case to ours and propose a diagnostic paradigm for this variant of CEP. Finally, a correlation between phenotype variability and the presence of modifier mutations in loci related to disease-causing gene is described.


Asunto(s)
Sustitución de Aminoácidos , Factor de Transcripción GATA1/genética , Estudios de Asociación Genética , Mutación , Porfiria Eritropoyética/diagnóstico , Porfiria Eritropoyética/genética , Biopsia , Médula Ósea/patología , Niño , Preescolar , Análisis Mutacional de ADN , Índices de Eritrocitos , Genes Ligados a X , Humanos , Masculino , Linaje , Fenotipo , Porfirinas/sangre , Porfirinas/orina
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