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The Autophagy-Cilia Axis: An Intricate Relationship

by 1,2,* and 1,2,*
Telethon Institute of Genetics and Medicine (TIGEM), Via Campi Flegrei, 34, 80078 Pozzuoli, Naples, Italy
Medical Genetics, Department of Translational Medical Sciences, Federico II University of Naples, Via Sergio Pansini 5, 80131 Naples, Italy
Authors to whom correspondence should be addressed.
Cells 2019, 8(8), 905;
Received: 19 July 2019 / Revised: 8 August 2019 / Accepted: 12 August 2019 / Published: 15 August 2019
(This article belongs to the Special Issue Cilia and Flagella: Structure, Function and Beyond)
Primary cilia are microtubule-based organelles protruding from the surface of almost all vertebrate cells. This organelle represents the cell’s antenna which acts as a communication hub to transfer extracellular signals into intracellular responses during development and in tissue homeostasis. Recently, it has been shown that loss of cilia negatively regulates autophagy, the main catabolic route of the cell, probably utilizing the autophagic machinery localized at the peri-ciliary compartment. On the other side, autophagy influences ciliogenesis in a context-dependent manner, possibly to ensure that the sensing organelle is properly formed in a feedback loop model. In this review we discuss the recent literature and propose that the autophagic machinery and the ciliary proteins are functionally strictly related to control both autophagy and ciliogenesis. Moreover, we report examples of diseases associated with autophagic defects which cause cilia abnormalities, and propose and discuss the hypothesis that, at least some of the clinical manifestations observed in human diseases associated to ciliary disfunction may be the result of a perturbed autophagy. View Full-Text
Keywords: primary cilium; autophagy; Hedgehog signaling; ciliopathy primary cilium; autophagy; Hedgehog signaling; ciliopathy
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Morleo, M.; Franco, B. The Autophagy-Cilia Axis: An Intricate Relationship. Cells 2019, 8, 905.

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