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IFT172 和 KIFAP3 中的新突变可能会引发基底细胞癌。

Novel alterations in IFT172 and KIFAP3 may induce basal cell carcinoma.

机构信息

Department of Biochemistry, Tokyo Dental College, 2-9-18, Kanda Misakichou, Chiyoda, Tokyo, 101-0061, Japan.

Department of Oral Medicine and Hospital Dentistry, Tokyo Dental College, 5-11-13, Sugano, Ichikawa, Chiba, 272-8513, Japan.

出版信息

Orphanet J Rare Dis. 2021 Oct 21;16(1):443. doi: 10.1186/s13023-021-02033-7.

Abstract

BACKGROUND

Basal cell carcinoma (BCC) is the most commonly occurring neoplasm in patients with Gorlin syndrome. It is widely accepted that multiple basal cell carcinomas simultaneously develop in middle-aged patients with this syndrome. However, the presence of driver genes other than the PTCH1 in Gorlin syndrome has not been explored. This study aimed to identify common gene mutations other than PTCH1 in simultaneously occurring basal cell carcinomas in patients with Gorlin syndrome via exome sequencing analysis.

METHODS

Next-generation sequencing analysis was performed using four basal cell carcinoma samples, one dental keratinocyte sample, and two epidermoid cyst samples, which were surgically resected from one patient with Gorlin syndrome on the same day.

RESULTS

Overall, 282 somatic mutations were identified in the neoplasms. No additional somatic mutations in PTCH1, PTCH2, TP53, and SMO were identified. However, enrichment analysis showed that multiple genes, such as IFT172 and KIFAP3, could regulate ciliary functions important for Hedgehog signaling.

CONCLUSION

The development of BCCs in patients with Gorlin syndrome may be triggered by mutations that cause substantial dysfunction of cilia.

摘要

背景

基底细胞癌(BCC)是神经纤维瘤病患者中最常见的肿瘤。人们普遍认为,患有这种综合征的中年患者会同时出现多个基底细胞癌。然而,神经纤维瘤病中除了 PTCH1 以外的驱动基因尚未被探索。本研究旨在通过外显子组测序分析,确定神经纤维瘤病患者同时发生的基底细胞癌中除了 PTCH1 以外的常见基因突变。

方法

对同一天从一名神经纤维瘤病患者身上手术切除的四个基底细胞癌样本、一个牙角质细胞样本和两个表皮样囊肿样本进行下一代测序分析。

结果

在肿瘤中总共鉴定出 282 个体细胞突变。在 PTCH1、PTCH2、TP53 和 SMO 中没有发现其他体细胞突变。然而,富集分析表明,多个基因,如 IFT172 和 KIFAP3,可调节对 Hedgehog 信号传导至关重要的纤毛功能。

结论

神经纤维瘤病患者的 BCC 发生可能是由导致纤毛功能严重失调的突变引发的。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/893f/8529737/6a65631a6d81/13023_2021_2033_Fig1_HTML.jpg

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