携带来自遗传性染色体内插入的重组X染色体的男性患者的重复。

IF 1.7 4区 生物学 Q4 CELL BIOLOGY Cytogenetic and Genome Research Pub Date : 2023-01-01 Epub Date: 2023-07-21 DOI:10.1159/000532051
Tatiana Mozer Joaquim, Scott David Roy, Clarissa Gondim Picanço de Albuquerque, Carlos Henrique Paiva Grangeiro, Jeremy A Squire, Maisa Yoshimoto, Lucia Martelli
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引用次数: 0

摘要

染色体内插入是复杂的结构重排,用经典的细胞遗传学方法解释是具有挑战性的。我们报告一个男性患者携带重组X染色体源自母系遗传染色体内插入。患者表现出发育迟缓、智力障碍、行为障碍和面部畸形。为了准确识别异常X染色体的重排,进行了额外的细胞遗传学研究,包括荧光原位杂交(FISH),多色带FISH和阵列比较基因组杂交。结果显示重组X染色体,在插入细胞带Xq26.1处的Xp22.33-Xp22.13片段存在13.05 Mb的间质重复。重复区域包含99个基因,其中一些与患者的临床表现有关。我们提出,xp重复基因的综合作用可能有助于患者的表型。
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Xp22.33p22.13 Duplication in a Male Patient Carrying a Recombinant X Chromosome Derived from an Inherited Intrachromosomal Insertion.

Intrachromosomal insertions are complex structural rearrangements that are challenging to interpret using classical cytogenetic methods. We report a male patient carrying a recombinant X chromosome derived from a maternally inherited intrachromosomal insertion. The patient exhibited developmental delay, intellectual disability, behavioral disorder, and dysmorphic facial features. To accurately identify the rearrangements in the abnormal X chromosome, additional cytogenetic studies were conducted, including fluorescence in situ hybridization (FISH), multicolor-banding FISH, and array comparative genomic hybridization. The results showed a recombinant X chromosome, resulting in a 13.05 Mb interstitial duplication of segment Xp22.33-Xp22.13, which was inserted at cytoband Xq26.1. The duplicated region encompasses 99 genes, some of which are associated with the patient's clinical manifestations. We propose that the combined effects of the Xp-duplicated genes may contribute to the patient's phenotype.

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来源期刊
Cytogenetic and Genome Research
Cytogenetic and Genome Research 生物-细胞生物学
CiteScore
3.10
自引率
5.90%
发文量
25
审稿时长
1 months
期刊介绍: During the last decades, ''Cytogenetic and Genome Research'' has been the leading forum for original reports and reviews in human and animal cytogenetics, including molecular, clinical and comparative cytogenetics. In recent years, most of its papers have centered on genome research, including gene cloning and sequencing, gene mapping, gene regulation and expression, cancer genetics, comparative genetics, gene linkage and related areas. The journal also publishes key papers on chromosome aberrations in somatic, meiotic and malignant cells. Its scope has expanded to include studies on invertebrate and plant cytogenetics and genomics. Also featured are the vast majority of the reports of the International Workshops on Human Chromosome Mapping, the reports of international human and animal chromosome nomenclature committees, and proceedings of the American and European cytogenetic conferences and other events. In addition to regular issues, the journal has been publishing since 2002 a series of topical issues on a broad variety of themes from cytogenetic and genome research.
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