Pub Date : 2024-03-11DOI: 10.1080/27694127.2024.2326402
Francois Singh, Lea Wilhelm, Alan R Prescott, Kevin Ostacolo, Jin-Feng Zhao, Margret H Ogmundsdottir, Ian G Ganley
PINK1, mutated in familial forms of Parkinson's disease, initiates mitophagy following mitochondrial depolarization. However, it is difficult to monitor this pathway physiologically in mice as loss of PINK1 does not alter basal mitophagy levels in most tissues. To further characterize this pathway in vivo, we used mito-QC mice in which loss of PINK1 was combined with the mitochondrial-associated POLGD257A mutation. We focused on skeletal muscle as gene expression data indicates that this tissue has the highest PINK1 levels. We found that loss of PINK1 in oxidative hindlimb muscle significantly reduced mitophagy. Of interest, the presence of the POLGD257A mutation, while having a minor effect in most tissues, restored levels of muscle mitophagy caused by the loss of PINK1. Although our observations highlight that multiple mitophagy pathways operate within a single tissue, we identify skeletal muscle as a tissue of choice for the study of PINK1-dependant mitophagy under basal conditions.
{"title":"PINK1 regulated mitophagy is evident in skeletal muscles.","authors":"Francois Singh, Lea Wilhelm, Alan R Prescott, Kevin Ostacolo, Jin-Feng Zhao, Margret H Ogmundsdottir, Ian G Ganley","doi":"10.1080/27694127.2024.2326402","DOIUrl":"10.1080/27694127.2024.2326402","url":null,"abstract":"<p><p>PINK1, mutated in familial forms of Parkinson's disease, initiates mitophagy following mitochondrial depolarization. However, it is difficult to monitor this pathway physiologically in mice as loss of PINK1 does not alter basal mitophagy levels in most tissues. To further characterize this pathway <i>in vivo</i>, we used <i>mito</i>-QC mice in which loss of PINK1 was combined with the mitochondrial-associated POLG<sup>D257A</sup> mutation. We focused on skeletal muscle as gene expression data indicates that this tissue has the highest PINK1 levels. We found that loss of PINK1 in oxidative hindlimb muscle significantly reduced mitophagy. Of interest, the presence of the POLG<sup>D257A</sup> mutation, while having a minor effect in most tissues, restored levels of muscle mitophagy caused by the loss of PINK1. Although our observations highlight that multiple mitophagy pathways operate within a single tissue, we identify skeletal muscle as a tissue of choice for the study of PINK1-dependant mitophagy under basal conditions.</p>","PeriodicalId":72341,"journal":{"name":"Autophagy reports","volume":"3 1","pages":"2326402"},"PeriodicalIF":0.0,"publicationDate":"2024-03-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7616148/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141581707","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-03-06DOI: 10.1080/27694127.2024.2325265
S. Vega-Rubín-de-Celis
{"title":"GRB2, a protein known for a long time with a new autophagy-regulating function","authors":"S. Vega-Rubín-de-Celis","doi":"10.1080/27694127.2024.2325265","DOIUrl":"https://doi.org/10.1080/27694127.2024.2325265","url":null,"abstract":"","PeriodicalId":72341,"journal":{"name":"Autophagy reports","volume":"44 1","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-03-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140260936","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-02-27DOI: 10.1080/27694127.2024.2320605
Jialing Tang, Eddie Tam, Erfei Song, Aimin Xu, Gary Sweeney
{"title":"Crosstalk between myocardial autophagy and sterile inflammation in the development of heart failure","authors":"Jialing Tang, Eddie Tam, Erfei Song, Aimin Xu, Gary Sweeney","doi":"10.1080/27694127.2024.2320605","DOIUrl":"https://doi.org/10.1080/27694127.2024.2320605","url":null,"abstract":"","PeriodicalId":72341,"journal":{"name":"Autophagy reports","volume":"29 11","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-02-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140425974","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-02-07DOI: 10.1080/27694127.2024.2306086
M. Severino, Lauren S. Richardson, A. Kammala, E. Radnaa, K. Khanipov, Leslie Michelle M. Dalmacio, Indira U. Mysorekar, Marian Kacerovsky, Ramkumar Menon
{"title":"Autophagy Determines Distinct Cell Fates in Human Amnion and Chorion Cells","authors":"M. Severino, Lauren S. Richardson, A. Kammala, E. Radnaa, K. Khanipov, Leslie Michelle M. Dalmacio, Indira U. Mysorekar, Marian Kacerovsky, Ramkumar Menon","doi":"10.1080/27694127.2024.2306086","DOIUrl":"https://doi.org/10.1080/27694127.2024.2306086","url":null,"abstract":"","PeriodicalId":72341,"journal":{"name":"Autophagy reports","volume":"4 2","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-02-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139854555","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-02-07DOI: 10.1080/27694127.2024.2306086
M. Severino, Lauren S. Richardson, A. Kammala, E. Radnaa, K. Khanipov, Leslie Michelle M. Dalmacio, Indira U. Mysorekar, Marian Kacerovsky, Ramkumar Menon
{"title":"Autophagy Determines Distinct Cell Fates in Human Amnion and Chorion Cells","authors":"M. Severino, Lauren S. Richardson, A. Kammala, E. Radnaa, K. Khanipov, Leslie Michelle M. Dalmacio, Indira U. Mysorekar, Marian Kacerovsky, Ramkumar Menon","doi":"10.1080/27694127.2024.2306086","DOIUrl":"https://doi.org/10.1080/27694127.2024.2306086","url":null,"abstract":"","PeriodicalId":72341,"journal":{"name":"Autophagy reports","volume":"112 10","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-02-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139794741","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-02-01eCollection Date: 2024-12-31DOI: 10.1080/27694127.2024.2306766
Hortense de Calbiac, Sebastian Montealegre, Marjolène Straube, Solène Renault, Hugo Debruge, Loïc Chentout, Sorana Ciura, Apolline Imbard, Edouard Le Guillou, Anca Marian, Nicolas Goudin, Laure Caccavelli, Sylvie Fabrega, Arnaud Hubas, Peter van Endert, Nicolas Dupont, Julien Diana, Edor Kabashi, Pascale de Lonlay
Patients with pathogenic variants in the TANGO2 gene suffer from severe and recurrent rhabdomyolysis episodes precipitated by fasting. Autophagy functioning was analyzed in vitro, in primary skeletal myoblasts from TANGO2 patients, in basal and fasting conditions, and TANGO2 mutations were associated with reduced LC3-II levels upon starvation. In zebrafish larvae, tango2 inhibition induced locomotor defects which were exacerbated by exposure to atorvastatin, a compound known to cause rhabdomyolysis. Importantly, rhabdomyolysis features of tango2 knockdown were associated with autophagy and mitophagy defects in zebrafish. Calpeptin treatment was sufficient to rescue the locomotor properties thanks to its beneficial effect on autophagy functioning in zebrafish and to improve LC3-II levels in starved primary muscle cells of TANGO2 patients. Overall, we demonstrated that TANGO2 plays an important role in autophagy thus giving rise to new therapeutic perspectives in the prevention of RM life-threatening episodes.
{"title":"TANGO2-related rhabdomyolysis symptoms are associated with abnormal autophagy functioning.","authors":"Hortense de Calbiac, Sebastian Montealegre, Marjolène Straube, Solène Renault, Hugo Debruge, Loïc Chentout, Sorana Ciura, Apolline Imbard, Edouard Le Guillou, Anca Marian, Nicolas Goudin, Laure Caccavelli, Sylvie Fabrega, Arnaud Hubas, Peter van Endert, Nicolas Dupont, Julien Diana, Edor Kabashi, Pascale de Lonlay","doi":"10.1080/27694127.2024.2306766","DOIUrl":"10.1080/27694127.2024.2306766","url":null,"abstract":"<p><p>Patients with pathogenic variants in the <i>TANGO2</i> gene suffer from severe and recurrent rhabdomyolysis episodes precipitated by fasting. Autophagy functioning was analyzed <i>in vitro</i>, in primary skeletal myoblasts from TANGO2 patients, in basal and fasting conditions, and <i>TANGO2</i> mutations were associated with reduced LC3-II levels upon starvation. In zebrafish larvae, <i>tango2</i> inhibition induced locomotor defects which were exacerbated by exposure to atorvastatin, a compound known to cause rhabdomyolysis. Importantly, rhabdomyolysis features of <i>tango2</i> knockdown were associated with autophagy and mitophagy defects in zebrafish. Calpeptin treatment was sufficient to rescue the locomotor properties thanks to its beneficial effect on autophagy functioning in zebrafish and to improve LC3-II levels in starved primary muscle cells of TANGO2 patients. Overall, we demonstrated that TANGO2 plays an important role in autophagy thus giving rise to new therapeutic perspectives in the prevention of RM life-threatening episodes.</p>","PeriodicalId":72341,"journal":{"name":"Autophagy reports","volume":"3 1","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7617261/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142900737","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-01-08DOI: 10.1080/27694127.2023.2300622
Manuel J. Mallén-Ponce, M. Pérez-Pérez
{"title":"ATG3 is subjected to redox regulation to quarantee ATG8 lipidation under ROS-generating stresses","authors":"Manuel J. Mallén-Ponce, M. Pérez-Pérez","doi":"10.1080/27694127.2023.2300622","DOIUrl":"https://doi.org/10.1080/27694127.2023.2300622","url":null,"abstract":"","PeriodicalId":72341,"journal":{"name":"Autophagy reports","volume":"27 3","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-01-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139445940","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
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