Proceedings of the National Academy of Sciences · 2006 · 294 citations · 24 references
Life SciencesGeneticsMolecular GeneticsCardiovascular GeneticsGene ExpressionMedical GeneticsCardiovascular DevelopmentGenetic DisordersMendelian Cardiovascular DisordersLamin A/cMissense MutationsMutationVascular Smooth Muscle DysfunctionVascular Smooth Muscle Regulation
Children with Hutchinson-Gilford progeria syndrome (HGPS) suffer from dramatic acceleration of some symptoms associated with normal aging, most notably cardiovascular disease that eventually leads to death from myocardial infarction and/or stroke usually in their second decade of life. For the vast majority of cases, a de novo point mutation in the lamin A (LMNA) gene is the cause of HGPS. This missense mutation creates a cryptic splice donor site that produces a mutant lamin A protein, termed "progerin," which carries a 50-aa deletion near its C terminus. We have created a mouse model for progeria by generating transgenics carrying a human bacterial artificial chromosome that harbors the common HGPS mutation. These mice develop progressive loss of vascular smooth muscle cells in the medial layer of large arteries, in a pattern very similar to that seen in children with HGPS. This mouse model should prove valuable for testing experimental therapies for this devastating disorder and for exploring cardiovascular disease in general.
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Recurrent de novo point mutations in lamin A cause Hutchinson–Gilford progeria syndrome
Maria Eriksson, W. Ted Brown, Leslie B. Gordon et al. · Nature · 2003 · 2.2K citations · Full text
Edwin M. Horwitz, Patricia Gordon, Winston K. K. Koo et al. · Proceedings of the National Academy of Sciences · 2002 · 1.6K citations · Full text
Medicine, Immunology, Cell Therapy +16
Lamin A Truncation in Hutchinson-Gilford Progeria
Annachiara De Sandre‐Giovannoli, Rafaëlle Bernard, Pierre Cau et al. · Science · 2003 · 1.5K citations