TY - JOUR AU - Rodriguez-Muela, Natalia TI - Autophagy in motor neuron diseases JO - Progress in molecular biology and translational science VL - 172 SN - 1877-1173 CY - Amsterdam [u.a.] PB - Elsevier M1 - DZNE-2021-01287 SP - 157-202 PY - 2020 AB - Motor neuron diseases (MNDs) are a wide group of neurodegenerative disorders characterized by the degeneration of a specific neuronal type located in the central nervous system, the motor neuron (MN). There are two main types of MNs, spinal and cortical MNs and depending on the type of MND, one or both types are affected. Cortical MNs innervate spinal MNs and these control a variety of cellular targets, being skeletal muscle their main one which is also affected in MNDs. A correct functionality of autophagy is necessary for the survival of all cellular types and it is particularly crucial for neurons, given their postmitotic and highly specialized nature. Numerous studies have identified alterations of autophagy activity in multiple MNDs. The scientific community has been particularly prolific in reporting the role that autophagy plays in the most common adult MND, amyotrophic lateral sclerosis, although many studies have started to identify physiological and pathological functions of this catabolic system in other MNDs, such as spinal muscular atrophy and spinal and bulbar muscular atrophy. The degradation of selective cargo by autophagy and how this process is altered upon the presence of MND-causing mutations is currently also a matter of intense investigation, particularly regarding the selective autophagic clearance of mitochondria. Thorough reviews on this field have been recently published. This chapter will cover the current knowledge on the functionality of autophagy and lysosomal homeostasis in the main MNDs and other autophagy-related topics in the MND field that have risen special interest in the research community. KW - Adult KW - Amyotrophic Lateral Sclerosis: genetics KW - Amyotrophic Lateral Sclerosis: pathology KW - Animals KW - Autophagy: drug effects KW - Autophagy: physiology KW - Autophagy-Related Proteins: genetics KW - Autophagy-Related Proteins: physiology KW - C9orf72 Protein: deficiency KW - C9orf72 Protein: genetics KW - C9orf72 Protein: physiology KW - DNA Repeat Expansion KW - Disease Models, Animal KW - Endocytosis KW - Humans KW - Mice, Transgenic KW - Motor Neuron Disease: genetics KW - Motor Neuron Disease: pathology KW - Muscular Atrophy, Spinal: genetics KW - Muscular Atrophy, Spinal: pathology KW - Mutation KW - Neurodegenerative Diseases: genetics KW - Neurodegenerative Diseases: pathology KW - Organelles KW - RNA-Binding Protein FUS: deficiency KW - RNA-Binding Protein FUS: genetics KW - RNA-Binding Protein FUS: physiology KW - TDP-43 Proteinopathies: genetics KW - TDP-43 Proteinopathies: pathology LB - PUB:(DE-HGF)16 C6 - pmid:32620242 DO - DOI:10.1016/bs.pmbts.2020.03.009 UR - https://pub.dzne.de/record/157830 ER -