Literature DB >> 30099632

Loss-of-function of Nav1.8/D1639N linked to human pain can be rescued by lidocaine.

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

Mesh:

Substances:

Year:  2018        PMID: 30099632     DOI: 10.1007/s00424-018-2189-x

Source DB:  PubMed          Journal:  Pflugers Arch        ISSN: 0031-6768            Impact factor:   3.657


  59 in total

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Authors:  P Chevrier; K Vijayaragavan; M Chahine
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  4 in total

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4.  Trafficking mechanisms underlying Nav channel subcellular localization in neurons.

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