Maraschio P, Danesino C, Lo Curto F, Zuffardi O, Dalla Fior T, Pedrotti D
Ann Genet. 1984;27(2):96-101.
A 69,XXX female liveborn triploid survived 45 days. The phenotype was consistent with the average clinical picture of liveborn triploids. Autopsy revealed slight atrophy of cerebral cortex and corpus callosum and severe adrenal hypoplasia. Chromosome polymorphisms indicated that the origin of this triploid was dispermy. Replication studies of the X chromosome performed on lymphocytes and fibroblasts showed that the majority of cells had two late replicating X chromosomes. X chromosome inactivation in spontaneous abortuses and liveborn triploids is discussed. Nine enzymes encoded by autosomal genes were tested, five had normal, three increased, and one reduced levels of activity. The reduced activity of alpha-galactosidase, an X-linked enzyme, is in agreement with cytogenetic findings and demonstrated a gene dosage effect.
一名69,XXX活产女性三倍体存活了45天。其表型与活产三倍体的平均临床症状相符。尸检显示大脑皮质和胼胝体轻度萎缩,肾上腺严重发育不全。染色体多态性表明该三倍体的起源是双精受精。对淋巴细胞和成纤维细胞进行的X染色体复制研究表明,大多数细胞有两条晚复制的X染色体。文中讨论了自然流产儿和活产三倍体中的X染色体失活情况。对常染色体基因编码的9种酶进行了检测,其中5种酶活性正常,3种酶活性增加,1种酶活性降低。X连锁酶α-半乳糖苷酶活性降低与细胞遗传学结果一致,证明了基因剂量效应。