In vitro functional correction of Hermansky-Pudlak Syndrome type-1 by lentiviral-mediated gene transfer.


Autoria(s): Ikawa Y.; Hess R.; Dorward H.; Cullinane A.R.; Huizing M.; Gochuico B.R.; Gahl W.A.; Candotti F.
Data(s)

2015

Resumo

Hermansky-Pudlak syndrome (HPS) is a genetic disorder characterized by oculocutaneous albinism, bleeding tendency and susceptibility to pulmonary fibrosis. No curative therapy is available. Genetic correction directed to the lungs, bone marrow and/or gastro-intestinal tract might provide alternative forms of treatment for the diseases multi-systemic complications. We demonstrate that lentiviral-mediated gene transfer corrects the expression and function of the HPS1 gene in patient dermal melanocytes, which opens the way to development of gene therapy for HPS.

Identificador

http://serval.unil.ch/?id=serval:BIB_4EAD5A280AFA

isbn:1096-7206 (Electronic)

pmid:25468649

doi:10.1016/j.ymgme.2014.11.006

isiid:000348178900011

http://my.unil.ch/serval/document/BIB_4EAD5A280AFA.pdf

http://nbn-resolving.org/urn/resolver.pl?urn=urn:nbn:ch:serval-BIB_4EAD5A280AFA0

Idioma(s)

en

Direitos

info:eu-repo/semantics/openAccess

Fonte

Molecular Genetics and Metabolism, vol. 114, no. 1, pp. 62-65

Tipo

info:eu-repo/semantics/article

article