Suppr超能文献

patched-1(PTCH1)基因的新突变及更新后的数据库

New mutations and an updated database for the patched-1 (PTCH1) gene.

作者信息

Reinders Marie G, van Hout Antonius F, Cosgun Betûl, Paulussen Aimée D, Leter Edward M, Steijlen Peter M, Mosterd Klara, van Geel Michel, Gille Johan J

机构信息

Department of Dermatology, Maastricht University Medical Center, Maastricht, The Netherlands.

GROW Research School for Oncology and Developmental Biology, Maastricht University Medical Center, Maastricht, The Netherlands.

出版信息

Mol Genet Genomic Med. 2018 May;6(3):409-415. doi: 10.1002/mgg3.380. Epub 2018 Mar 25.

Abstract

BACKGROUND

Basal cell nevus syndrome (BCNS) is an autosomal dominant disorder characterized by multiple basal cell carcinomas (BCCs), maxillary keratocysts, and cerebral calcifications. BCNS most commonly is caused by a germline mutation in the patched-1 (PTCH1) gene. PTCH1 mutations are also described in patients with holoprosencephaly.

METHODS

We have established a locus-specific database for the PTCH1 gene using the Leiden Open Variation Database (LOVD). We included 117 new PTCH1 variations, in addition to 331 previously published unique PTCH1 mutations. These new mutations were found in 141 patients who had a positive PTCH1 mutation analysis in either the VU University Medical Centre (VUMC) or Maastricht University Medical Centre (MUMC) between 1995 and 2015.

RESULTS

The database contains 331 previously published unique PTCH1 mutations and 117 new PTCH1 variations.

CONCLUSION

We have established a locus-specific database for the PTCH1 gene using the Leiden Open Variation Database (LOVD). The database provides an open collection for both clinicians and researchers and is accessible online at http://www.lovd.nl/PTCH1.

摘要

背景

基底细胞痣综合征(BCNS)是一种常染色体显性疾病,其特征为多发性基底细胞癌(BCC)、上颌角化囊肿和脑钙化。BCNS最常见由patched-1(PTCH1)基因的种系突变引起。全前脑畸形患者中也有PTCH1突变的描述。

方法

我们使用莱顿开放变异数据库(LOVD)建立了PTCH1基因的位点特异性数据库。除了331个先前发表的独特PTCH1突变外,我们纳入了117个新的PTCH1变异。这些新突变是在1995年至2015年间于VU大学医学中心(VUMC)或马斯特里赫特大学医学中心(MUMC)进行PTCH1突变分析呈阳性的141名患者中发现的。

结果

该数据库包含331个先前发表的独特PTCH1突变和117个新的PTCH1变异。

结论

我们使用莱顿开放变异数据库(LOVD)建立了PTCH1基因的位点特异性数据库。该数据库为临床医生和研究人员提供了一个开放的集合,可在http://www.lovd.nl/PTCH1在线访问。

相似文献

7
Unexpected phenotype in a frameshift mutation of PTCH1.PTCH1 移码突变导致的意外表型。
Mol Genet Genomic Med. 2020 Jan;8(1):e987. doi: 10.1002/mgg3.987. Epub 2019 Oct 2.

引用本文的文献

1
Germline variants in CDKN2A wild-type melanoma prone families.CDKN2A野生型黑色素瘤易感家族中的种系变异
Mol Oncol. 2025 May;19(5):1493-1507. doi: 10.1002/1878-0261.70020. Epub 2025 Mar 12.
2
Nevoid basal cell carcinoma syndrome with anophthalmia: a case report.伴无眼畸形的痣样基底细胞癌综合征:一例报告
Hua Xi Kou Qiang Yi Xue Za Zhi. 2022 Mar 25;40(2):240-245. doi: 10.7518/hxkq.2022.02.018.
8
Oral Cancer Stem Cells: Therapeutic Implications and Challenges.口腔癌干细胞:治疗意义与挑战
Front Oral Health. 2021 Jul 21;2:685236. doi: 10.3389/froh.2021.685236. eCollection 2021.
10
Molecular alterations in basal cell carcinoma subtypes.基底细胞癌亚型中的分子改变。
Sci Rep. 2021 Jun 24;11(1):13206. doi: 10.1038/s41598-021-92592-3.

本文引用的文献

1
Postzygotic mosaicism in basal cell naevus syndrome.基底层细胞痣综合征中的合子后镶嵌性。
Br J Dermatol. 2017 Jul;177(1):249-252. doi: 10.1111/bjd.15082. Epub 2017 Jun 6.
6
"PTCH"-ing it together: a basal cell nevus syndrome review.PTCH 整合在一起:基底细胞痣综合征综述。
Dermatol Surg. 2013 Nov;39(11):1557-72. doi: 10.1111/dsu.12241. Epub 2013 May 31.

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验