Wulfsberg E A, Wassel W C, Polo C A
Clinical Genetics/Dysmorphology, National Naval Medical Center, Bethesda, Maryland.
Clin Genet. 1991 May;39(5):370-5. doi: 10.1111/j.1399-0004.1991.tb03044.x.
Diploid/triploid mosaicism is an uncommon clinical syndrome with a subtle but distinctive phenotype. Characteristic features include prenatal and postnatal asymmetric growth deficiency, triangular and/or asymmetric facies, micrognathia, finger and/or toe syndactyly, clinodactyly, single transverse palmar creases, male genital anomalies, hypotonia and psychomotor retardation. This disorder is underdiagnosed because in 70% of cases the triploid cell line is only seen in fibroblasts. In cases in which a triploid cell line is found in lymphocytes, it usually occurs in less than 5% of cells. While some reports of diploid/triploid mosaicism have mentioned unusual skin pigmentary patterns, including hypomelanosis of Ito, it was only recently recognized that this is a helpful diagnostic clue in mosaic chromosome disorders. We report monozygotic twin girls with diploid/triploid mosaicism whose cutaneous pigmentary dysplasia led to their diagnosis.
二倍体/三倍体嵌合体是一种罕见的临床综合征,具有细微但独特的表型。特征包括产前和产后不对称生长发育迟缓、三角形和/或不对称面容、小颌畸形、手指和/或脚趾并指、指侧弯、单一掌横纹、男性生殖器异常、肌张力减退和精神运动发育迟缓。这种疾病诊断不足,因为在70%的病例中,三倍体细胞系仅在成纤维细胞中可见。在淋巴细胞中发现三倍体细胞系的病例中,其通常出现在不到5%的细胞中。虽然一些关于二倍体/三倍体嵌合体的报告提到了不寻常的皮肤色素沉着模式,包括伊藤色素减退,但直到最近才认识到这是嵌合染色体疾病的一个有用诊断线索。我们报告了一对患有二倍体/三倍体嵌合体的单卵双胞胎女孩,其皮肤色素发育异常导致了她们的诊断。