Journal of Clinical Investigation · 1970 · 119 citations · 29 references
Life SciencesGeneticsHematologyBiochemical GeneticsHuman GeneticsMedical GeneticsAnemiaHematologic DisordersHemolytic DisordersRed Blood Cell DisordersHematological AnalysisHematological PhenotypingHemoglobin Synthesis DefectsImbalance
Homozygous thalassemia is due to inherited unbalanced synthesis of the alpha- or beta-chains of hemoglobin. Clinical severity may be in part related to the extent of alpha:beta imbalance. Two families are presented that illustrate this concept. Thalassemia in these individuals was evaluated by clinical and genetic criteria. The relative rates of alpha- and beta-chain synthesis in their reticulocytes were estimated by the extent of incorporation of 1-leucine-U-(14)C into the chains. Unusual combinations of clinical and hematological data and biosynthetic ratios were obtained in certain individuals which indicated the presence of combinations of alpha- and beta-thalassemia genes. The propositus of the first family had mild Cooley's anemia and was believed to have one alpha- as well as two beta-thalassemia genes. Presumably the alpha-thalassemia gene interfered with alpha-chain production which lead to less accumulation of alpha-chains and a reduced rate of intramedullary and peripheral hemolysis. In the second family two individuals were believed to have an alpha-thalassemia, a "silent carrier," and a beta-thalassemia gene. Despite the fact that they appeared to have the genotype of hemoglobin H disease, their cells contained no hemoglobin H and had a normal lifespan presumably because excess beta-chain production was inhibited by the beta-thalessemia gene. These family studies suggest that the alpha:beta imbalance observed in thalassemia may be favorably influenced by combinations of alpha- and beta-thalassemia genes.
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Globin Synthesis in Thalassaemia: An in vitro Study
D. J. Weatherall, J. B. Clegg, Michael A. Naughton · Nature · 1965 · 236 citations
Genetic Basis of the Thalassæmia Diseases
Vernon M. Ingram, Antony O.W. Stretton · Nature · 1959 · 218 citations
Inclusions of Hemoglobin in Erythroblasts and Erythrocytes of Thalassemia
Phaedon Fessas · Blood · 1963 · 169 citations · Full text
Medicine, Genetics, Hematology +10