Figure 4.

Key regulators of mitochondrial transport implicated in peripheral neuropathy. Anterograde and retrograde mitochondrial transport along the microtubule cytoskeleton is mediated by specific machinery. Protein names highlighted in red are linked to peripheral neuropathy phenotypes. The outer mitochondrial membrane homologs MIRO1 and MIRO2 interact with the homologous TRAK1 and TRAK2 adaptors. Kinesins, including the KIF5A implicated in hereditary spastic paraplegia and peripheral neuropathy, interact with TRAK1/2 to mediate anterograde mitochondrial transport (+). Meanwhile, retrograde mitochondrial transport (−) is mediated by the dynein complex, including DYNC1H1, which is implicated in CMT2O.