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In peripheral neurons\u2014cells with high metabolic demands and long axons\u2014the OMM acts as a vital platform for coordinating bioenergetics, calcium signaling, and redox balance. Ganglioside-induced differentiation-associated protein 1 (GDAP1), an OMM-anchored protein, has emerged as a key regulator of mitochondrial fission and transport, redox homeostasis, and mitochondrial membrane contact sites (MCSs). Genetic variants in GDAP1 cause Charcot\u2013Marie\u2013Tooth disease (CMT), emphasizing its essential role in peripheral nerve function. This review highlights the multifaceted functions of GDAP1 in neuronal physiology and as a model protein that integrates organelle communication and mitochondrial biology. We further discuss how GDAP1 dysfunction leads to structural and functional impairments in peripheral neurons, proposing the OMM and its microenvironment as critical targets for therapeutic intervention in inherited neuropathies.<\/jats:p>","DOI":"10.3390\/biom16020280","type":"journal-article","created":{"date-parts":[[2026,2,10]],"date-time":"2026-02-10T17:10:52Z","timestamp":1770743452000},"page":"280","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Unraveling GDAP1: Bridging Mitochondrial Biology and Peripheral Neuropathy"],"prefix":"10.3390","volume":"16","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-5196-5262","authenticated-orcid":false,"given":"Lara","family":"Cantarero","sequence":"first","affiliation":[{"name":"Laboratory of Neurogenetics and Molecular Medicine, Center for Genomic Sciences in Medicine, Institut de Recerca Sant Joan de D\u00e9u, 08950 Barcelona, Spain"},{"name":"Centro de Investigaci\u00f3n Biom\u00e9dica en Red de Enfermedades Raras (CIBERER), ISCIII, 08950 Barcelona, Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Janet","family":"Hoenicka","sequence":"additional","affiliation":[{"name":"Laboratory of Neurogenetics and Molecular Medicine, Center for Genomic Sciences in Medicine, Institut de Recerca Sant Joan de D\u00e9u, 08950 Barcelona, Spain"},{"name":"Centro de Investigaci\u00f3n Biom\u00e9dica en Red de Enfermedades Raras (CIBERER), ISCIII, 08950 Barcelona, Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Francesc","family":"Palau","sequence":"additional","affiliation":[{"name":"Laboratory of Neurogenetics and Molecular Medicine, Center for Genomic Sciences in Medicine, Institut de Recerca Sant Joan de D\u00e9u, 08950 Barcelona, Spain"},{"name":"Centro de Investigaci\u00f3n Biom\u00e9dica en Red de Enfermedades Raras (CIBERER), ISCIII, 08950 Barcelona, Spain"},{"name":"Hospital Sant Joan de D\u00e9u, 08950 Barcelona, Spain"},{"name":"Division of Pediatrics, Faculty of Medicine and Health Sciences, University of Barcelona, 08007 Barcelona, Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2026,2,10]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"190126","DOI":"10.1098\/rsob.190126","article-title":"Mitochondria-hubs for regulating cellular biochemistry: Emerging concepts and networks","volume":"9","author":"Anderson","year":"2019","journal-title":"Open Biol."},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"1487","DOI":"10.1016\/j.molcel.2025.03.003","article-title":"Mitochondria-organelle crosstalk in establishing compartmentalized metabolic homeostasis","volume":"85","author":"Chen","year":"2025","journal-title":"Mol. 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