Literature DB >> 26711856

Painful small fiber neuropathy with gastroparesis: A new phenotype with a novel mutation in the SCN10A gene.

Ron Dabby1, Menachem Sadeh2, Yelena Broitman3, Keren Yosovich4, Ram Dickman3, Esther Leshinsky-Silver4.   

Abstract

Gain-of-function mutations in the SCN10A gene (encoding the Nav1.8 voltage gated sodium channel) have been reported in a small number of patients. All presented with predominantly painful sensory neuropathy, congruent with the expression of Nav1.8 in nociceptive sensory neurons of the dorsal root ganglion. Only a few had mild autonomic symptoms, including dry eyes and mouth, orthostatic dizziness, palpitations, diarrhea and constipation. The underlying mechanism of the autonomic symptoms in these patients is unclear. We describe a 37-year-old woman with severe progressive gastroparesis and diffuse painful small fiber sensory neuropathy that started at age 32. Due to the severe dysphagia she could not ingest solid food, and lost eight kilograms. The gastroparesis was documented by esophageal manometry and gastric scintigraphy. The neuropathic pain started distally and then intensified and spread to most body areas. The patient harbored a novel heterozygous mutation: c.G4915A:p.D1639N in the SCN10A gene. To the best of our knowledge, this is the first description of such a phenotype due to a Nav1.8 mutation. Thus, our study expands the clinical spectrum of Nav1.8 associated disorders, and suggests that mutations in this sodium channel should be considered in patients with gastrointestinal motility dysfunction and painful neuropathy.
Copyright © 2015 Elsevier Ltd. All rights reserved.

Entities:  

Keywords:  Autonomic neuropathy; Gastroparesis; Na(v)1.8 sodium channel; Neuropathic pain; SCN10A; Small fiber neuropathy

Mesh:

Substances:

Year:  2015        PMID: 26711856     DOI: 10.1016/j.jocn.2015.05.071

Source DB:  PubMed          Journal:  J Clin Neurosci        ISSN: 0967-5868            Impact factor:   1.961


  8 in total

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

Authors:  Luisa Kaluza; Jannis E Meents; Martin Hampl; Corinna Rösseler; Petra A I Hautvast; Silvia Detro-Dassen; Ralf Hausmann; Günther Schmalzing; Angelika Lampert
Journal:  Pflugers Arch       Date:  2018-08-11       Impact factor: 3.657

2.  [Pain and analgesia : Mutations of voltage-gated sodium channels].

Authors:  M J Eberhardt; A Leffler
Journal:  Schmerz       Date:  2017-02       Impact factor: 1.107

3.  Association of small-fiber polyneuropathy with three previously unassociated rare missense SCN9A variants.

Authors:  Mary A Kelley; Anne Louise Oaklander
Journal:  Can J Pain       Date:  2020-02-05

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

Authors:  Laura Solé; Michael M Tamkun
Journal:  Channels (Austin)       Date:  2020-12       Impact factor: 2.581

5.  The Human SCN10AG1662S Point Mutation Established in Mice Impacts on Mechanical, Heat, and Cool Sensitivity.

Authors:  Celeste Chidiac; Yaping Xue; Maria Del Mar Muniz Moreno; Ameer Abu Bakr Rasheed; Romain Lorentz; Marie-Christine Birling; Claire Gaveriaux-Ruff; Yann Herault
Journal:  Front Pharmacol       Date:  2021-12-01       Impact factor: 5.810

6.  Gastrointestinal Symptoms and Channelopathy-Associated Epilepsy.

Authors:  Veronica C Beck; Lori L Isom; Anne T Berg
Journal:  J Pediatr       Date:  2021-06-26       Impact factor: 6.314

7.  Ambroxol for the treatment of fibromyalgia: science or fiction?

Authors:  Kai-Uwe Kern; Myriam Schwickert
Journal:  J Pain Res       Date:  2017-08-16       Impact factor: 3.133

Review 8.  Genetic studies of human neuropathic pain conditions: a review.

Authors:  Katerina Zorina-Lichtenwalter; Marc Parisien; Luda Diatchenko
Journal:  Pain       Date:  2018-03       Impact factor: 7.926

  8 in total

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