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Abolishing the prelamin A ZMPSTE24 cleavage site leads to progeroid phenotypes with near-normal longevity in mice

Yuexia Wang, Khurts Shilagardi, Trunee Hsu, Kamsi O. Odinammadu, Takamitsu Maruyama, Wei Wu, Chyuan‐Sheng Lin, Christopher B. Damoci, Eric D. Spear, Ji‐Yeon Shin, Wei Hsu, Susan Michaelis, Howard J. Worman

2022Proceedings of the National Academy of Sciences19 citationsDOIOpen Access PDF

Abstract

Significance The zinc metalloprotease ZMPSTE24 removes the last 15 amino acids of prelamin A, including a farnesylated cysteine, to produce mature lamin A. The premature aging disorder Hutchinson–Gilford progeria syndrome is caused by a permanently farnesylated prelamin A variant lacking the ZMPSTE24 cleavage site. ZMPSTE24 loss of function leads to the accumulation of farnesylated prelamin A and causes progeroid disorders. Some studies have implicated prelamin A in physiological aging. We describe mice with an amino acid substitution in prelamin A that blocks the ZMPSTE24-catalyzed cleavage. These mice develop progeroid phenotypes but, in contrast to those modeling Hutchinson–Gilford progeria syndrome or ZMPSTE24 deficiency, have near-normal lifespans, thus providing a model to study the effects of farnesylated prelamin A during aging.

Topics & Concepts

ProgeriaLaminPhenotypeBiologyPremature agingCleavage (geology)Werner syndromeGeneticsBiochemistryGeneFracture (geology)HelicasePaleontologyRNANuclear Structure and FunctionRNA Research and SplicingGenomics and Chromatin Dynamics
Abolishing the prelamin A ZMPSTE24 cleavage site leads to progeroid phenotypes with near-normal longevity in mice | Litcius