Myosin 1b regulates intestinal epithelial morphogenesis via interaction with UNC45A.

Autor:	Revenu C; Institut Curie, PSL Research University, INSERM U934, CNRS UMR3215, 75248 Paris Cedex, France; Sorbonne Université, INSERM, CNRS, Institut de la Vision, 75012 Paris, France., Lebreton C; INSERM, UMR1163, Laboratory of Intestinal Immunity and Institut Imagine, 75015 Paris, France., Cannata Serio M; Institut Curie, PSL Research University, Sorbonne Université, CNRS, UMR 144, Paris, France., Rosello M; Institut Curie, PSL Research University, INSERM U934, CNRS UMR3215, 75248 Paris Cedex, France; Sorbonne Université, INSERM, CNRS, Institut de la Vision, 75012 Paris, France., Duclaux-Loras R; INSERM, UMR1163, Laboratory of Intestinal Immunity and Institut Imagine, 75015 Paris, France., Duroure K; Institut Curie, PSL Research University, INSERM U934, CNRS UMR3215, 75248 Paris Cedex, France; Sorbonne Université, INSERM, CNRS, Institut de la Vision, 75012 Paris, France., Nicolle O; Université de Rennes, CNRS, IGDR (Institut de Génétique et de Développement de Rennes), UMR 6290, 35000 Rennes, France., Eggeler F; Sorbonne Université, INSERM, CNRS, Institut de la Vision, 75012 Paris, France., Prospéri MT; Institut Curie, PSL Research University, Sorbonne Université, CNRS, UMR 144, Paris, France., Stoufflet J; Institut Curie, PSL Research University, INSERM U934, CNRS UMR3215, 75248 Paris Cedex, France., Vougny J; Institut Curie, PSL Research University, INSERM U934, CNRS UMR3215, 75248 Paris Cedex, France., Lépine P; Institut Curie, PSL Research University, Sorbonne Université, CNRS, UMR 144, Paris, France., Michaux G; Université de Rennes, CNRS, IGDR (Institut de Génétique et de Développement de Rennes), UMR 6290, 35000 Rennes, France., Cerf-Bensussan N; INSERM, UMR1163, Laboratory of Intestinal Immunity and Institut Imagine, 75015 Paris, France., Coudrier E; Institut Curie, PSL Research University, Sorbonne Université, CNRS, UMR 144, Paris, France., Perez F; Institut Curie, PSL Research University, Sorbonne Université, CNRS, UMR 144, Paris, France., Parlato M; INSERM, UMR1163, Laboratory of Intestinal Immunity and Institut Imagine, 75015 Paris, France. Electronic address: marianna.parlato@inserm.fr., Del Bene F; Institut Curie, PSL Research University, INSERM U934, CNRS UMR3215, 75248 Paris Cedex, France; Sorbonne Université, INSERM, CNRS, Institut de la Vision, 75012 Paris, France. Electronic address: filippo.del-bene@inserm.fr.
Jazyk:	angličtina
Zdroj:	Cell reports [Cell Rep] 2024 Dec 24; Vol. 43 (12), pp. 114941. Date of Electronic Publication: 2024 Dec 04.
DOI:	10.1016/j.celrep.2024.114941
Abstrakt:	Vesicle trafficking and the establishment of apicobasal polarity are essential processes in epithelial morphogenesis. UNC45A deficiency has been reported in a multi-organ syndrome presenting with severe diarrhea associated with enterocyte polarity defects. Myosin 1b, an actin motor able to bind membranes, regulates membrane shaping and vesicle trafficking. Here, we show that MYO1B is part of the UNC45A interactome. In the absence of UNC45A, myosin 1b is degraded and forms aggregates when proteasome activity is inhibited. In 3D Caco-2 cells, lumen formation is impaired in the absence of myosin 1b, associated with spindle orientation defects, Golgi apparatus fragmentation, and trafficking impairment. In zebrafish larvae, loss of myo1b results in intestinal bulb epithelium folding defects associated with terminal web disorganization and vesicle accumulation, reminiscent of villous atrophy. In conclusion, we show that myosin 1b plays an unexpected role in the development of the intestinal epithelium downstream of UNC45A, establishing its contribution in the gut defects reported in UNC45A patients. Competing Interests: Declaration of interests The authors declare no competing interests. (Copyright © 2024 The Author(s). Published by Elsevier Inc. All rights reserved.)
Databáze:	MEDLINE
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