| Literature DB >> 29208544 |
Vittoria Cenni1, Maria Rosaria D'Apice2, Paolo Garagnani3, Marta Columbaro4, Giuseppe Novelli5, Claudio Franceschi6, Giovanna Lattanzi7.
Abstract
Mandibuloacral dysplasia (MAD) is a rare genetic condition characterized by bone abnormalities including localized osteolysis and generalized osteoporosis, skin pigmentation, lipodystrophic signs and mildly accelerated ageing. The molecular defects associated with MAD are mutations in LMNA or ZMPSTE24 (FACE1) gene, causing type A or type B MAD, respectively. Downstream of LMNA or ZMPSTE24 mutations, the lamin A precursor, prelamin A, is accumulated in cells and affects chromatin dynamics and stress response. A new form of mandibuloacral dysplasia has been recently associated with mutations in POLD1 gene, encoding DNA polymerase delta, a major player in DNA replication. Of note, involvement of prelamin A in chromatin dynamics and recruitment of DNA repair factors has been also determined under physiological conditions, at the border between stress response and cellular senescence. Here, we review current knowledge on MAD clinical and pathogenetic aspects and highlight aspects typical of physiological ageing.Entities:
Keywords: Aging; Lamin A/C gene (LMNA); Mandibuloacral dysplasia (MAD); Prelamin A; Progeroid syndromes; ZMPSTE24
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Year: 2017 PMID: 29208544 DOI: 10.1016/j.arr.2017.12.001
Source DB: PubMed Journal: Ageing Res Rev ISSN: 1568-1637 Impact factor: 10.895