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评估 60 例土耳其 NF1 基因变异患者脑磁共振成像中的临床发现和神经纤维瘤病 1 型亮体。

Evaluation of clinical findings and neurofibromatosis type 1 bright objects on brain magnetic resonance images of 60 Turkish patients with NF1 gene variants.

机构信息

Department of Medical Genetics, Dr. Behcet Uz Children's Hospital, Izmir, Turkey.

Department of Pediatric Genetics, Dr. Behcet Uz Children's Hospital, Izmir, Turkey.

出版信息

Neurol Sci. 2021 May;42(5):2045-2057. doi: 10.1007/s10072-020-04988-0. Epub 2021 Jan 14.

DOI:10.1007/s10072-020-04988-0
PMID:33443663
Abstract

Neurofibromatosis type 1 (NF1) is caused by mutations in the NF1 gene. This retrospective study aims to evaluate the clinical manifestations and brain magnetic resonance images (MRI) analysis in 60 genetically confirmed NF1 patients. The results of next-generation sequencing (NGS), Sanger sequencing, and MLPA of NF1 gene were evaluated. A total of 54 different variants were identified. Fourteen out of them were novel variants (25.9%). Patients who complied with NIH criteria had most frequently frameshift variants (11/32 patients), and those with only CALMs had missense variants (9/28 patients). Neurofibromatosis type 1 bright objects (NBOs) on T2-weighted MRI were detected in 42 patients (42/56; 75%). These brain lesions were detected mostly in basal ganglia and in cerebellar vermis. NBOs were detected more in the patients who complied with NIH criteria (80.6%) compared to those who were only CALMs (68%). While frameshift variants (33.3%) were the most common type variants in the patients who had NBOs, the most common variants were splicing (35.7%) and missense (35.7%) variants in the patients whose MRIs were normal. Frameshift variants (11/28 patients; 39.3%) were the most common in the patients with more than one brain locus involvement. Therefore, we consider that frameshift variants may be associated with increased incidence of NBOs and involvement of more than one brain locus. In addition, NBOs may occur less frequently in the patients with splicing variants. To our knowledge, this is the first study evaluated the relationship between NF1 gene variants and NBOs. Future studies may help us understand the etiology of NBOs.

摘要

神经纤维瘤病 1 型(NF1)是由 NF1 基因突变引起的。本回顾性研究旨在评估 60 例经基因证实的 NF1 患者的临床表现和脑部磁共振成像(MRI)分析结果。对 NF1 基因的下一代测序(NGS)、Sanger 测序和 MLPA 结果进行了评估。共发现 54 种不同的变异,其中 14 种为新变异(25.9%)。符合 NIH 标准的患者最常出现移码变异(11/32 例患者),而仅有 CALMs 的患者则为错义变异(9/28 例患者)。在 42 例患者(42/56;75%)的 T2 加权 MRI 上检测到神经纤维瘤病 1 型亮区(NBO)。这些脑部病变主要位于基底节和小脑蚓部。符合 NIH 标准的患者(80.6%)比仅有 CALMs 的患者(68%)更易出现 NBOs。在有 NBOs 的患者中,移码变异(33.3%)是最常见的变异类型,而在 MRI 正常的患者中,最常见的变异类型是剪接(35.7%)和错义(35.7%)变异。在有多个脑部病灶受累的患者中,最常见的变异是移码变异(11/28 例患者;39.3%)。因此,我们认为移码变异可能与 NBOs 的发生率增加和多个脑部病灶受累有关。此外,剪接变异的患者中 NBOs 的发生率可能较低。据我们所知,这是首次评估 NF1 基因突变与 NBOs 之间关系的研究。未来的研究可能有助于我们了解 NBOs 的病因。

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Neurol Sci. 2020 Oct;41(10):2685-2690. doi: 10.1007/s10072-020-04400-x. Epub 2020 May 1.
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Phenotypic expression of a spectrum of Neurofibromatosis Type 1 (NF1) mutations identified through NGS and MLPA.
通过 NGS 和 MLPA 鉴定的一系列神经纤维瘤病 1 型 (NF1) 突变的表型表达。
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