含 RAI1 的超常标记染色体导致的马赛克波托奇-卢普斯基综合征

Khaliunaa Bayanbold, Noel Tolbanen, John A Bernat, Jaime Nagy
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摘要

波托基-卢普斯基综合征(Potocki-Lupski Syndrome,PTLS)是一种复发性微重复综合征,以发育迟缓、行为异常、轻度面部畸形、肌张力低下和睡眠障碍为特征。我们在此报告了一名被诊断为镶嵌型 PTLS 的 3 岁女孩,她体内有一条含有 RAI1(视黄酸诱导 1)基因的超常标记染色体。细胞遗传学检测(包括染色体微阵列、核型和 FISH 分析)在 85% 的细胞中发现了含有 14 号和 17 号染色体部分的环状染色体。该病例的临床特征包括非典型面容,额部隆起,咬颞部狭窄,耳朵突出,以及轻度语言发育迟缓。本文介绍的是一例与 14 号和 17 号染色体镶嵌增益相关的 PTLS 新病例。由于涉及非同心染色体的小超常标记染色体(sSMC)非常罕见,本病例有助于我们了解与sSMC(17)相关的表型谱系。
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Mosaic Potocki-Lupski Syndrome Due to a Supernumerary Marker Chromosome Containing RAI1
Potocki-Lupski syndrome (PTLS) is a recurrent microduplication syndrome characterized by developmental delay, behavioral abnormalities, mildly dysmorphic facial features, hypotonia, and sleep disorders. We report here a 3-year-old girl diagnosed with mosaic PTLS harboring a supernumerary marker chromosome containing the RAI1 (retinoic acid induced 1) gene. Cytogenetic testing, including chromosomal microarray, karyotype, and FISH analysis, identified a ring chromosome containing portions of chromosomes 14 and 17 in 85% of cells. Clinical features of this individual included atypical facies with frontal bossing, bitemporal narrowing, prominent cupped ears, and mild speech delay. Presented here is a novel case of PTLS associated with mosaic gains of chromosomes 14 and 17. As small supernumerary marker chromosomes (sSMCs) involving non-acrocentric chromosomes are rare, this case contributes to our understanding of phenotypic spectrum associated with sSMC(17).
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