Xue Yang, Ni Jinwen, Zhou Mi, Wang Weiqi, Liu Yuan, Yang Yaowu, Duan Xiaohong
State Key Laboratory of Military Stomatology, Department of Oral Biology, Clinic of Oral Rare and Genetic Diseases, School of Stomatology, the Fourth Military Medical University, 145 West Changle Road, Xi'an 710032, PR China.
State Key Laboratory of Military Stomatology, Department of Oral and Maxillofacial Surgery, School of Stomatology, the Fourth Military Medical University, 145 West Changle Road, Xi'an 710032, PR China.
J Craniomaxillofac Surg. 2015 Sep;43(7):1248-55. doi: 10.1016/j.jcms.2015.05.017. Epub 2015 Jun 3.
To report on the molecular genetic analysis of a Chinese patient with Maffucci syndrome.
Using the genomic DNA extracted from the patient's hemangioma sample, the coding exons and exon/intron splice junctions of the IDH1 and IDH2 genes were amplified by polymerase chain reaction (PCR) and then sequenced. Genomic DNA was extracted from blood and a hemangioma sample from the patient, and also from her mother's blood, for chromosome microarray analysis (CMA) by Affymetrix CytoScan HD array.
None of the known pathogenic mutations in the whole IDH1 or IDH2 genes was found in the patient's hemangioma sample. CMA detected 40 tumor-specific copy number variations (CNVs), and one copy number neutral loss of heterozygosity (LOH) region. Among the 73 known genes included in the 40 CNV regions, only 2 genes, CHEK2 (604373) located in 22q12.1 and EP300 (602700) located in 22q13.2, were found to be related to tumorigenesis. We did not find any CNVs at the IDH1 and IDH2 loci.
This is the first molecular genetic analysis report on a Chinese patient with Maffucci syndrome and our data enrich the understanding of the genetic background of Maffucci syndrome in different ethnic groups. The relationship between CHEK2, EP300 and Maffucci syndrome needs to be further explored.
报告一名中国马富西综合征患者的分子遗传学分析情况。
使用从患者血管瘤样本中提取的基因组DNA,通过聚合酶链反应(PCR)扩增异柠檬酸脱氢酶1(IDH1)和异柠檬酸脱氢酶2(IDH2)基因的编码外显子和外显子/内含子剪接位点,然后进行测序。从患者的血液和血管瘤样本以及其母亲的血液中提取基因组DNA,用于通过Affymetrix CytoScan HD芯片进行染色体微阵列分析(CMA)。
在患者的血管瘤样本中未发现IDH1或IDH2基因的已知致病突变。CMA检测到40个肿瘤特异性拷贝数变异(CNV)和一个拷贝数中性杂合性缺失(LOH)区域。在40个CNV区域包含的73个已知基因中,仅发现位于22q12.1的CHEK2(604373)和位于22q13.2的EP300(602700)这2个基因与肿瘤发生相关。我们在IDH1和IDH2基因座未发现任何CNV。
这是关于一名中国马富西综合征患者的首份分子遗传学分析报告,我们的数据丰富了对不同种族马富西综合征遗传背景的认识。CHEK2、EP300与马富西综合征之间的关系有待进一步探索。