Kakela Justin K, Friedman Kenneth D, Haberichter Sandra L, Buchholz Nadine P, Christopherson Pam A, Kroner Philip A, Gill Joan Cox, Montgomery Robert R, Bellissimo Daniel B
BloodCenter of Wisconsin, 638 N. 18th Street, P.O. Box 2178, Milwaukee, WI 53201, USA.
Mol Genet Metab. 2006 Mar;87(3):262-71. doi: 10.1016/j.ymgme.2005.09.016.
Von Willebrand disease (VWD) is a common inherited bleeding disorder caused by quantitative (types 1 and 3) and qualitative (type 2) defects in von Willebrand factor (VWF). The VWF gene is a large gene containing 52 exons; except for type 2 VWD, the majority of mutations causing VWD are not localized to specific exons. We have used denaturing high performance liquid chromatography (DHPLC) to scan the coding region of the VWF gene for sequence variations. Primers were designed to amplify all 52 exons while avoiding amplification of the VWF pseudogene. Exon-specific primers were designed with sequencing primers, allowing direct sequencing of each VWF exon. Sequence variations in 33 previously characterized von Willebrand disease (VWD) samples were all detected using DHPLC demonstrating the high sensitivity of this technique. In addition, we analyzed 42 patients or family members with VWD. Thirty-two novel sequence variations were identified (2 deletions, 2 nonsense, 15 missense, 6 silent, and 7 intronic), some with clear functional consequences. A previously described deletion in exon 18, 2435delC, was also found in two unrelated type 3 patients. This DHPLC and DNA sequencing technique will enable the full length assessment of the VWF gene necessary to detect mutations causing types 1 and 3 VWD.
血管性血友病(VWD)是一种常见的遗传性出血性疾病,由血管性血友病因子(VWF)的数量缺陷(1型和3型)和质量缺陷(2型)引起。VWF基因是一个包含52个外显子的大基因;除2型VWD外,导致VWD的大多数突变并不局限于特定外显子。我们使用变性高效液相色谱(DHPLC)扫描VWF基因的编码区以寻找序列变异。设计引物以扩增所有52个外显子,同时避免扩增VWF假基因。外显子特异性引物与测序引物一起设计,从而能够直接对每个VWF外显子进行测序。使用DHPLC检测到了33个先前已鉴定的血管性血友病(VWD)样本中的序列变异,证明了该技术的高灵敏度。此外,我们分析了42例VWD患者或家庭成员。鉴定出32个新的序列变异(2个缺失、2个无义、15个错义、6个沉默和7个内含子),其中一些具有明确的功能后果。在两名无关的3型患者中也发现了先前描述的外显子18缺失,即2435delC。这种DHPLC和DNA测序技术将能够对检测导致1型和3型VWD的突变所需的VWF基因进行全长评估。