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下颌骨-肢端发育不良:一种具有生理老化特征的早衰疾病。

Mandibuloacral dysplasia: A premature ageing disease with aspects of physiological ageing.

机构信息

CNR Institute of Molecular Genetics, Unit of Bologna, Via di Barbiano 1/10, I-40136, Bologna, Italy; Rizzoli Orthopedic Institute, Via di Barbiano 1/10, I-40136, Bologna, Italy.

Tor Vergata University Hospital, Viale Oxford 81, I-00133 Rome, Italy.

出版信息

Ageing Res Rev. 2018 Mar;42:1-13. doi: 10.1016/j.arr.2017.12.001. Epub 2017 Dec 5.

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.

摘要

下颌骨-肢端发育不良(MAD)是一种罕见的遗传疾病,其特征为骨骼异常,包括局部溶骨性和全身性骨质疏松症、皮肤色素沉着、脂肪营养不良迹象和轻度加速老化。与 MAD 相关的分子缺陷是 LMNA 或 ZMPSTE24(FACE1)基因突变,分别导致 A 型或 B 型 MAD。在 LMNA 或 ZMPSTE24 突变的下游,前层粘连蛋白 A 前体 prelamin A 在细胞中积累,并影响染色质动力学和应激反应。最近,下颌骨-肢端发育不良的一种新形式与编码 DNA 聚合酶 delta 的 POLD1 基因突变有关,DNA 聚合酶 delta 是 DNA 复制的主要参与者。值得注意的是,在生理条件下,在前层粘连蛋白 A 参与染色质动力学和招募 DNA 修复因子的情况下,在应激反应和细胞衰老之间的边界也得到了确定。在这里,我们回顾了 MAD 临床和发病机制方面的最新知识,并强调了与生理衰老相关的方面。

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