Nodes of ranvier and paranodes in chronic acquired neuropathies.

Chronic acquired neuropathies of unknown origin are classified as chronic inflammatory demyelinating polyneuropathies (CIDP) and chronic idiopathic axonal polyneuropathies (CIAP). The diagnosis can be very difficult, although it has important therapeutic implications since CIDP can be improved by im...

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Autores principales:	Carmen Cifuentes-Diaz, Odile Dubourg, Theano Irinopoulou, Marc Vigny, Sylvie Lachkar, Laurence Decker, Patrick Charnay, Natalia Denisenko, Thierry Maisonobe, Jean-Marc Léger, Karine Viala, Jean-Jacques Hauw, Jean-Antoine Girault
Formato:	article
Lenguaje:	EN
Publicado:	Public Library of Science (PLoS) 2011
Materias:	Medicine R Science Q
Acceso en línea:	https://doaj.org/article/81f02b68dbe042d4bebd1756b1e9fb2a
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spelling	oai:doaj.org-article:81f02b68dbe042d4bebd1756b1e9fb2a2021-11-18T07:00:22ZNodes of ranvier and paranodes in chronic acquired neuropathies.1932-620310.1371/journal.pone.0014533https://doaj.org/article/81f02b68dbe042d4bebd1756b1e9fb2a2011-01-01T00:00:00Zhttps://www.ncbi.nlm.nih.gov/pmc/articles/pmid/21267074/pdf/?tool=EBIhttps://doaj.org/toc/1932-6203Chronic acquired neuropathies of unknown origin are classified as chronic inflammatory demyelinating polyneuropathies (CIDP) and chronic idiopathic axonal polyneuropathies (CIAP). The diagnosis can be very difficult, although it has important therapeutic implications since CIDP can be improved by immunomodulating treatment. The aim of this study was to examine the possible abnormalities of nodal and paranodal regions in these two types of neuropathies. Longitudinal sections of superficial peroneal nerves were obtained from biopsy material from 12 patients with CIDP and 10 patients with CIAP and studied by immunofluorescence and in some cases electron microscopy. Electron microscopy revealed multiple alterations in the nodal and paranodal regions which predominated in Schwann cells in CIDP and in axons in CIAP. In CIDP paranodin/Caspr immunofluorescence was more widespread than in control nerves, extending along the axon in internodes where it appeared intense. Nodal channels Nav and KCNQ2 were less altered but were also detected in the internodes. In CIAP paranodes, paranodin labeling was irregular and/or decreased. To test the consequences of acquired primary Schwann cells alteration on axonal proteins, we used a mouse model based on induced deletion of the transcription factor Krox-20 gene. In the demyelinated sciatic nerves of these mice we observed alterations similar to those found in CIDP by immunofluorescence, and immunoblotting demonstrated increased levels of paranodin. Finally we examined whether the alterations in paranodin immunoreactivity could have a diagnosis value. In a sample of 16 biopsies, the study of paranodin immunofluorescence by blind evaluators led to correct diagnosis in 70 ± 4% of the cases. This study characterizes for the first time the abnormalities of nodes of Ranvier in CIAP and CIDP, and the altered expression and distribution of nodal and paranodal proteins. Marked differences were observed between CIDP and CIAP and the alterations in paranodin immunofluorescence may be an interesting tool for their differential diagnosis.Carmen Cifuentes-DiazOdile DubourgTheano IrinopoulouMarc VignySylvie LachkarLaurence DeckerPatrick CharnayNatalia DenisenkoThierry MaisonobeJean-Marc LégerKarine VialaJean-Jacques HauwJean-Antoine GiraultPublic Library of Science (PLoS)articleMedicineRScienceQENPLoS ONE, Vol 6, Iss 1, p e14533 (2011)
institution	DOAJ
collection	DOAJ
language	EN
topic	Medicine R Science Q
spellingShingle	Medicine R Science Q Carmen Cifuentes-Diaz Odile Dubourg Theano Irinopoulou Marc Vigny Sylvie Lachkar Laurence Decker Patrick Charnay Natalia Denisenko Thierry Maisonobe Jean-Marc Léger Karine Viala Jean-Jacques Hauw Jean-Antoine Girault Nodes of ranvier and paranodes in chronic acquired neuropathies.
description	Chronic acquired neuropathies of unknown origin are classified as chronic inflammatory demyelinating polyneuropathies (CIDP) and chronic idiopathic axonal polyneuropathies (CIAP). The diagnosis can be very difficult, although it has important therapeutic implications since CIDP can be improved by immunomodulating treatment. The aim of this study was to examine the possible abnormalities of nodal and paranodal regions in these two types of neuropathies. Longitudinal sections of superficial peroneal nerves were obtained from biopsy material from 12 patients with CIDP and 10 patients with CIAP and studied by immunofluorescence and in some cases electron microscopy. Electron microscopy revealed multiple alterations in the nodal and paranodal regions which predominated in Schwann cells in CIDP and in axons in CIAP. In CIDP paranodin/Caspr immunofluorescence was more widespread than in control nerves, extending along the axon in internodes where it appeared intense. Nodal channels Nav and KCNQ2 were less altered but were also detected in the internodes. In CIAP paranodes, paranodin labeling was irregular and/or decreased. To test the consequences of acquired primary Schwann cells alteration on axonal proteins, we used a mouse model based on induced deletion of the transcription factor Krox-20 gene. In the demyelinated sciatic nerves of these mice we observed alterations similar to those found in CIDP by immunofluorescence, and immunoblotting demonstrated increased levels of paranodin. Finally we examined whether the alterations in paranodin immunoreactivity could have a diagnosis value. In a sample of 16 biopsies, the study of paranodin immunofluorescence by blind evaluators led to correct diagnosis in 70 ± 4% of the cases. This study characterizes for the first time the abnormalities of nodes of Ranvier in CIAP and CIDP, and the altered expression and distribution of nodal and paranodal proteins. Marked differences were observed between CIDP and CIAP and the alterations in paranodin immunofluorescence may be an interesting tool for their differential diagnosis.
format	article
author	Carmen Cifuentes-Diaz Odile Dubourg Theano Irinopoulou Marc Vigny Sylvie Lachkar Laurence Decker Patrick Charnay Natalia Denisenko Thierry Maisonobe Jean-Marc Léger Karine Viala Jean-Jacques Hauw Jean-Antoine Girault
author_facet	Carmen Cifuentes-Diaz Odile Dubourg Theano Irinopoulou Marc Vigny Sylvie Lachkar Laurence Decker Patrick Charnay Natalia Denisenko Thierry Maisonobe Jean-Marc Léger Karine Viala Jean-Jacques Hauw Jean-Antoine Girault
author_sort	Carmen Cifuentes-Diaz
title	Nodes of ranvier and paranodes in chronic acquired neuropathies.
title_short	Nodes of ranvier and paranodes in chronic acquired neuropathies.
title_full	Nodes of ranvier and paranodes in chronic acquired neuropathies.
title_fullStr	Nodes of ranvier and paranodes in chronic acquired neuropathies.
title_full_unstemmed	Nodes of ranvier and paranodes in chronic acquired neuropathies.
title_sort	nodes of ranvier and paranodes in chronic acquired neuropathies.
publisher	Public Library of Science (PLoS)
publishDate	2011
url	https://doaj.org/article/81f02b68dbe042d4bebd1756b1e9fb2a
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Nodes of ranvier and paranodes in chronic acquired neuropathies.

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