| Literature DB >> 30099632 |
Luisa Kaluza1, Jannis E Meents1, Martin Hampl1,2, Corinna Rösseler1, Petra A I Hautvast1, Silvia Detro-Dassen3, Ralf Hausmann3, Günther Schmalzing3, Angelika Lampert4.
Abstract
Mutations in voltage-gated sodium channels are associated with altered pain perception in humans. Most of these mutations studied to date present with a direct and intuitive link between the altered electrophysiological function of the channel and the phenotype of the patient. In this study, we characterize a variant of Nav1.8, D1639N, which has been previously identified in a patient suffering from the chronic pain syndrome "small fiber neuropathy". Using a heterologous expression system and patch-clamp analysis, we show that Nav1.8/D1639N reduces current density without altering biophysical gating properties of Nav1.8. Therefore, the D1639N variant causes a loss-of-function of the Nav1.8 sodium channel in a patient suffering from chronic pain. Using immunocytochemistry and biochemical approaches, we show that Nav1.8/D1639N impairs trafficking of the channel to the cell membrane. Neither co-expression of β1 or β3 subunit, nor overnight incubation at 27 °C rescued current density of the D1639N variant. On the other hand, overnight incubation with lidocaine fully restored current density of Nav1.8/D1639N most likely by overcoming the trafficking defect, whereas phenytoin failed to do so. Since lidocaine rescues the loss-of-function of Nav1.8/D1639N, it may offer a future therapeutic option for the patient carrying this variant. These results demonstrate that the D1639N variant, identified in a patient suffering from chronic pain, causes loss-of-function of the channel due to impaired cell surface trafficking and that this trafficking defect can be rescued by lidocaine.Entities:
Keywords: Inherited pain syndromes; Local anesthetics; Mutagenesis; Patch-clamp; Small fiber neuropathy; Trafficking
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Year: 2018 PMID: 30099632 DOI: 10.1007/s00424-018-2189-x
Source DB: PubMed Journal: Pflugers Arch ISSN: 0031-6768 Impact factor: 3.657