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由于 novel BRPF1 variant,来自Çanakkale 的一个家族出现贫血和血小板减少症,伴有智力障碍和畸形面容:病例报告及文献复习。

Anemia and thrombocytopenia due to a novel BRPF1 variant in a family from Çanakkale with intellectual disability and dysmorphic facies: Case report and review of the literature.

机构信息

Department of Medical Genetics, Çanakkale Onsekiz Mart University, Faculty of Medicine, Çanakkale, Turkey.

出版信息

Am J Med Genet A. 2023 Aug;191(8):2209-2214. doi: 10.1002/ajmg.a.63244. Epub 2023 May 15.

DOI:10.1002/ajmg.a.63244
PMID:37190896
Abstract

Intellectual developmental disorder with dysmorphic facies and ptosis (IDDDFP) (MIM#617333) is an autosomal dominant disorder characterized by delayed psychomotor development, intellectual disability (ID), and dysmorphic facial features due to pathogenic variations in the Bromodomain- and PHD Finger-Containing Protein (BRPF1) (MIM#602410) gene. Herein, we report the first Turkish patients with IDDDFP. Additionally, the patients had hematopoietic disorders such as anemia and thrombocytopenia, which have not been previously described in IDDDFP patients. Genetic testing using Whole Exome Sequencing (WES) revealed a novel heterozygous c.1433G > A; p.W478* (NM_004634.3) pathogenic variant on exon 3 of the BRPF1 gene. The patients demonstrated classical features of IDDDFP such as intellectual disability, developmental delay, ptosis, micro and retrognathia, and dysmorphic facial features, in addition to the anemia and thrombocytopenia. Apart from the variant in BRPF1, no additional genomic changes were detected by WES and chromosomal microarray analysis (CMA). Hopefully, our novel report on the hematopoietic anomalies of our patients due to BRPF1 will expand upon the clinical spectrum of IDDDFP, encourage further studies about BRPF1-hematopoietic system relations, and affect the diagnostic and therapeutic schemes of hematopoietic system disorders.

摘要

具有畸形面容和上睑下垂的智力发育障碍 (IDDDFP) (MIM#617333) 是一种常染色体显性疾病,其特征是精神运动发育迟缓、智力障碍 (ID) 和由于 Bromodomain- 和 PHD 指状蛋白 (BRPF1) (MIM#602410) 基因突变导致的畸形面容。在此,我们报告了首例土耳其 IDDDFP 患者。此外,这些患者还存在血液系统疾病,如贫血和血小板减少症,这些疾病以前在 IDDDFP 患者中尚未描述过。使用全外显子组测序 (WES) 的基因检测发现 BRPF1 基因外显子 3 上存在一个新的杂合 c.1433G>A;p.W478* (NM_004634.3) 致病性变异。这些患者表现出 IDDDFP 的典型特征,如智力障碍、发育迟缓、上睑下垂、小颌和后缩以及畸形面容,此外还伴有贫血和血小板减少症。除了 BRPF1 中的变异外,WES 和染色体微阵列分析 (CMA) 未检测到其他基因组变化。希望我们关于 BRPF1 引起的患者血液系统异常的新报告能够扩展 IDDDFP 的临床谱,鼓励进一步研究 BRPF1 与造血系统的关系,并影响血液系统疾病的诊断和治疗方案。

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引用本文的文献

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Bromodomain and PHD Finger-Containing Protein 1: From Functions to a Developmental Disorder, Cancer, and Therapeutics.含溴结构域和PHD结构域蛋白1:从功能到发育障碍、癌症及治疗学
Results Probl Cell Differ. 2025;75:411-434. doi: 10.1007/978-3-031-91459-1_15.
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The Phenotypic and Genotypic Spectrum of BRPF1-Related Disorder: 29 New Patients and Literature Review.BRPF1相关疾病的表型和基因型谱:29例新患者及文献综述
Clin Genet. 2025 May;107(5):527-540. doi: 10.1111/cge.14688. Epub 2024 Dec 29.