Badens C, Mattei M G, Imbert A M, Lapouméroulie C, Martini N, Michel G, Lena-Russo D
Lancet. 2002 Jan 12;359(9301):132-3. doi: 10.1016/s0140-6736(02)07338-5.
Thalassaemia intermedia is a moderate form of thalassaemia resulting from various genetic defects. We report an undescribed mechanism leading to this condition: a somatic deletion of the beta-globin gene in the haemopoietic lineage of a heterozygous beta-thalassaemic patient. We did molecular studies and haemoglobin analysis of the patient and his parents. We found that the deletion gives rise to a mosaic of cells with either one or no functional beta-globin gene and it extends to a region of frequent loss of heterozygosity called LOH11A, which is located close to the beta-globin locus. Thus, loss of heterozygosity can be a cause of non-malignant genetic disease.
中间型地中海贫血是一种由多种基因缺陷导致的中度地中海贫血。我们报告了一种导致这种病症的未被描述的机制:一名杂合子β地中海贫血患者造血谱系中β珠蛋白基因的体细胞缺失。我们对该患者及其父母进行了分子研究和血红蛋白分析。我们发现这种缺失导致了具有一个或没有功能性β珠蛋白基因的细胞嵌合体,并且它延伸到一个称为LOH11A的杂合性频繁缺失区域,该区域位于β珠蛋白基因座附近。因此,杂合性缺失可能是非恶性遗传疾病的一个原因。