日本 1 型神经纤维瘤病患者的基因型和表型:遗传性肿瘤队列研究》。

IF 1 Q4 GENETICS & HEREDITY Human Genome Variation Pub Date : 2024-11-26 DOI:10.1038/s41439-024-00299-4
Mashu Futagawa, Tetsuya Okazaki, Eiji Nakata, Chika Fukano, Risa Osumi, Fumino Kato, Yusaku Urakawa, Hideki Yamamoto, Toshifumi Ozaki, Akira Hirasawa
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引用次数: 0

摘要

神经纤维瘤病 1 型(NF1)有多种临床表现,包括肿瘤发生和高血压的风险增加。有关 NF1 患者基因型与表型相关性的综合数据十分有限。因此,在本研究中,我们旨在阐明遗传性肿瘤队列中 NF1 的详细遗传和临床特征。我们在临床实验室进行了测序和拷贝数检测,并分析了 44 名疑似 NF1 患者的临床数据。在36名患者(81.8%)中检测到了种系致病变异,其中20.7%为新型变异。值得注意的是,40.0%的成年患者患有恶性肿瘤;20.0%的患者患有女性乳腺癌,这一比例高于之前的报道。30.6%的成年患者患有高血压,其中一名患者猝死,另一名患者罹患嗜铬细胞瘤。三名 NF1 基因大缺失患者表现出明显的皮肤、骨骼和神经系统症状。这些结果凸显了定期监测的重要性,尤其是对恶性肿瘤和高血压患者。我们的研究结果为遗传咨询和临床管理提供了有价值的见解,强调了与NF1相关的多种健康风险以及全面和多学科护理的必要性。
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Genotypes and phenotypes of neurofibromatosis type 1 patients in Japan: A Hereditary Tumor Cohort Study.

Neurofibromatosis type 1 (NF1) presents with a broad spectrum of clinical manifestations, including an increased risk of tumor development and hypertension. Comprehensive data on genotype‒phenotype correlations in patients with NF1 are limited. Therefore, in this study, we aimed to elucidate the detailed genetic and clinical characteristics of NF1 in a hereditary tumor cohort. We performed sequencing and copy number assays in a clinical laboratory and analyzed the clinical data of 44 patients with suspected NF1. Germline pathogenic variants were detected in 36 patients (81.8%), and 20.7% of the variants were novel. Notably, 40.0% of adult patients presented with malignancies; female breast cancer occurred in 20.0% of patients, which was a higher rate than that previously reported. Hypertension was observed in 30.6% of the adult patients, with one patient experiencing sudden death and another developing pheochromocytoma. Three patients with large deletions in NF1 exhibited prominent cutaneous, skeletal, and neurological manifestations. These results highlight the importance of regular surveillance, particularly for patients with malignancies and hypertension. Our findings provide valuable insights for genetic counseling and clinical management, highlighting the multiple health risks associated with NF1 and the need for comprehensive and multidisciplinary care.

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来源期刊
Human Genome Variation
Human Genome Variation Biochemistry, Genetics and Molecular Biology-Genetics
CiteScore
2.30
自引率
0.00%
发文量
39
审稿时长
13 weeks
期刊最新文献
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