Maguire Jean Ann, Lu Lin, Mills Jason A, Sullivan Lisa M, Gadue Paul, French Deborah L
Center for Cellular and Molecular Therapeutics, The Children's Hospital of Philadelphia, United States; Department of Pathology and Laboratory Medicine, The Children's Hospital of Philadelphia, United States.
Department of Pathology and Laboratory Medicine, The Children's Hospital of Philadelphia, United States; University of Pennsylvania, United States.
Stem Cell Res. 2016 Mar;16(2):287-9. doi: 10.1016/j.scr.2016.01.015. Epub 2016 Jan 14.
Hermansky-Pudlak syndrome type 2 (HPS2) is a rare autosomal recessive disorder resulting from functional mutations in the adaptor-related protein complex 3, beta 1 subunit (AP3B1) gene. This gene plays a role in organelle biogenesis associated with melanosomes, platelet dense granules, and lysosomes. Here we describe the generation of an HPS2 iPS cell line (CHOPHPS2) using a Cre-excisable polycistronic STEMCCA lentivirus. This line was derived from human fibroblasts isolated from a patient carrying two mutations in the AP3B1 gene. The patient presented with severe neutropenia, ocular albinism, interstitial pulmonary fibrosis, hemorrhagic diathesis, and an absence of platelet-dense granules.
2型赫尔曼斯基-普德拉克综合征(HPS2)是一种罕见的常染色体隐性疾病,由衔接蛋白相关蛋白复合物3β1亚基(AP3B1)基因的功能突变引起。该基因在与黑素小体、血小板致密颗粒和溶酶体相关的细胞器生物发生中起作用。在此,我们描述了使用可经Cre切除的多顺反子STEMCCA慢病毒生成HPS2诱导多能干细胞系(CHOPHPS2)的过程。该细胞系源自从一名携带AP3B1基因两个突变的患者分离出的人成纤维细胞。该患者表现为严重中性粒细胞减少、眼白化病、间质性肺纤维化、出血素质以及血小板致密颗粒缺失。