| Literature DB >> 12757706 |
Maria Pia Cosma1, Stefano Pepe, Ida Annunziata, Robert F Newbold, Markus Grompe, Giancarlo Parenti, Andrea Ballabio.
Abstract
In multiple sulfatase deficiency (MSD), a human inherited disorder, the activities of all sulfatases are impaired due to a defect in posttranslational modification. Here we report the identification, by functional complementation using microcell-mediated chromosome transfer, of a gene that is mutated in MSD and is able to rescue the enzymatic deficiency in patients' cell lines. Functional conservation of this gene was observed among distantly related species, suggesting a critical biological role. Coexpression of SUMF1 with sulfatases results in a strikingly synergistic increase of enzymatic activity, indicating that SUMF1 is both an essential and a limiting factor for sulfatases. These data have profound implications on the feasibility of enzyme replacement therapy for eight distinct inborn errors of metabolism.Entities:
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Year: 2003 PMID: 12757706 DOI: 10.1016/s0092-8674(03)00348-9
Source DB: PubMed Journal: Cell ISSN: 0092-8674 Impact factor: 41.582