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一名患有帕利斯特-基利安综合征女孩的12号染色体短臂12pter-12p12.3部分四体:发现一条异常的无着丝粒、倒位重复标记染色体。

Partial tetrasomy 12pter-12p12.3 in a girl with Pallister-Killian syndrome: extraordinary finding of an analphoid, inverted duplicated marker.

作者信息

Dufke A, Walczak C, Liehr T, Starke H, Trifonov V, Rubtsov N, Schöning M, Enders H, Eggermann T

机构信息

Abteilung Medizinische Genetik, Klinikum der Eberhard-Karls-Universität Tübingen, Germany.

出版信息

Eur J Hum Genet. 2001 Aug;9(8):572-6. doi: 10.1038/sj.ejhg.5200673.

Abstract

Cytogenetic analysis in a girl with multiple congenital anomalies indicating Pallister-Killian syndrome (PKS) showed a supernumerary marker chromosome in 1/76 lymphocytes and 34/75 fibroblast metaphases. GTG-banding pattern was consistent with the chromosomal region 12pter-12q11. While fluorescence-in-situ hybridisation (FISH) with a whole chromosome 12 painting probe confirmed the origin of the marker, a chromosome 12 specific alpha-satellite probe did not hybridise to it. FISH analysis with a specific subtelomeric probe 12p showed hybridisation to both ends of the marker chromosome. High-resolution multicolour-banding (MCB) studies revealed the marker to be a der(12)(pter-->p12.3::p12.3-->pter). Summarising the FISH information, we defined the marker as an inverted duplication of 12pter-12p12.3 leading to partial tetrasomy of chromosome 12p. In skin fibroblasts, cultured at the patient's age of 1 year and 9 years, the marker chromosome was found in similar frequencies, even after several culture passages. Therefore, we consider the marker to have a functional centromere although it lacks detectable centromeric alpha-satellite sequences. To the best of our knowledge, this is the first proven analphoid marker of chromosome 12. Molecular genetic studies indicated that this marker is of paternal origin. The finding of partial tetrasomy 12pter-12p12.3 in our PKS patient allows to narrow down the critical region for PKS.

摘要

对一名患有多种先天性异常、提示帕利斯特-基利安综合征(PKS)的女孩进行的细胞遗传学分析显示,在1/76的淋巴细胞和34/75的成纤维细胞中期相中存在一条额外的标记染色体。GTG显带模式与染色体区域12pter - 12q11一致。虽然用全染色体12涂染探针进行的荧光原位杂交(FISH)证实了该标记的起源,但染色体12特异性α卫星探针并未与其杂交。用特异性亚端粒探针12p进行的FISH分析显示该标记染色体两端均有杂交信号。高分辨率多色带(MCB)研究显示该标记为der(12)(pter→p12.3::p12.3→pter)。综合FISH信息,我们将该标记定义为12pter - 12p12.3的倒位重复,导致染色体12p部分四体性。在患者1岁和9岁时培养的皮肤成纤维细胞中,即使经过多次传代培养,标记染色体的出现频率仍相似。因此,我们认为该标记具有功能性着丝粒,尽管它缺乏可检测到的着丝粒α卫星序列。据我们所知,这是首例经证实的染色体12的无着丝粒标记。分子遗传学研究表明该标记源自父方。在我们的PKS患者中发现12pter - 12p12.3部分四体性,有助于缩小PKS的关键区域。

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