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Alpha-Thalassemia as Cause of Nonimmunological Hydrops

HOWARD A. PEARSON, MD; D. R. SHANKLIN, MD; CHARLES R. BRODINE, MD; BETHESDA, MD
Am J Dis Child. 1965;109(2):168-172. doi:10.1001/archpedi.1965.02090020170016.
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IN 1955 A Filipino couple had a premature fetus with clinical and pathological features of hydrops fetalis, but no iso-immune basis for the disease could be documented. The 1962 report of Lie-Injo and her associates in Indonesia describing homozygous α-thalassemia as a cause of nonimmunological hydrops fetalis1 prompted reinvestigation of this family. Both parents and two living siblings were found to have laboratory features of heterozygous α-thalassemia. Although detailed studies of the hydropic infant's hemoglobin were not performed, we consider it likely that she represented another instance of homozygous α-thalassemia. The α-thalassemia gene reaches a high frequency in persons of southern Asiatic origin and also perhaps in the American Negro. Therefore, in some racial groups, this recently described disorder of hemoglobin synthesis may be a significant cause of fetal wastage.

Materials and Methods  Standard hematological methods were employed for hemoglobin and hematocrit determinations. The Coulter counter was used to

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