Crosstalk between cilia and autophagy: implication for human diseases.

Manuela Morleo, Helena L A Vieira, Petra Pennekamp, Alessandro Palma, Liliana Bento-Lopes, Heymut Omran, Susana S Lopes, Duarte C Barral, Brunella Franco

Journal: Autophagy 2023;19(1):24-43

PMID: 35613303

Abstract

Macroautophagy/autophagy is a self-degradative process necessary for cells to maintain their energy balance during development and in response to nutrient deprivation. Autophagic processes are tightly regulated and have been found to be dysfunctional in several pathologies. Increasing experimental evidence points to the existence of an interplay between autophagy and cilia. Cilia are microtubule-based organelles protruding from the cell surface of mammalian cells that perform a variety of motile and sensory functions and, when dysfunctional, result in disorders known as ciliopathies. Indeed, selective autophagic degradation of ciliary proteins has been shown to control ciliogenesis and, conversely, cilia have been reported to control autophagy. Moreover, a growing number of players such as lysosomal and mitochondrial proteins are emerging as actors of the cilia-autophagy interplay. However, some of the published data on the cilia-autophagy axis are contradictory and indicate that we are just starting to understand the underlying molecular mechanisms. In this review, the current knowledge about this axis and challenges are discussed, as well as the implication for ciliopathies and autophagy-associated disorders.

Address: Telethon Institute of Genetics and Medicine (TIGEM), 80078, Pozzuoli, Italy.; Department of Precision Medicine, University of Campania "Luigi Vanvitelli", Naples, Italy.; CEDOC, NOVA Medical School, NMS, Universidade NOVA de Lisboa, Lisboa 1169-056, Portugal.; UCIBIO, Applied Molecular Biosciences Unit, Department of Chemistry, NOVA School of Science and Technology, Universidade NOVA de Lisboa, Caparica, Portugal.; Associate Laboratory i4HB - Institute for Health and Bioeconomy, NOVA School of Science and Technology, Universidade NOVA de Lisboa, Caparica, Portugal.; Department of General Pediatrics, University Hospital Münster, University of Münster, Münster 48149, Germany.; Member of the European Reference Networks ERN-LUNG, Lisbon, Portugal.; Department of Onco-hematology, Gene and Cell Therapy, Bambino Gesù Children's Hospital - IRCCS, Rome, Italy.; Medical Genetics, Department of Translational Medical Science, University of Naples "Federico II", Naples, Italy.; Scuola Superiore Meridionale, School for Advanced Studies, Naples, Italy.
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