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Clinical characterisation of sensory neuropathy with anti-FGFR3 autoantibodies

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Autor(es):
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Tholance, Yannick [1, 2] ; Moritz, Christian Peter [1] ; Rosier, Carole [1, 3] ; Ferraud, Karine [3] ; Lassabliere, Francois [1] ; Reynaud-Federspiel, Evelyne [1] ; Franca, Jr., Marcondes C. [4] ; Martinez, Alberto R. M. [4] ; Camdessanche, Jean-Philippe [1, 3] ; Antoine, Jean-Christophe [1, 3] ; Adams, David [5] ; Cauquil, Cecile [5] ; Attarian, Shahram [6] ; Delmont, Emilien [6] ; Blanchet-Fourcade, Genevieve [7] ; Callias, Celine [8] ; Cassereau, Julien [9] ; Choumert, Ariane [10] ; Clavelou, Pierre [11] ; Creange, Alain [12] ; Di Virgilio, Gabriella [13] ; Echaniz-Laguna, Andoni [14] ; Faucher, Benoit [15] ; Galea, Ian [16] ; Genestet, Steeve [17] ; Gueguen, Antoine [18] ; Ion, Ioana M. [19] ; Juntas-Morales, Raul [20] ; Taieb, Guillaume [20] ; Kuntzer, Thierry [21] ; Lagrange, Emmeline [22] ; Leger, Jean-Marc [23] ; Stojkovic, Tanya [23] ; Lepetit, Maud [24] ; Magy, Laurent [25] ; Mas, Julie [26] ; Michaud, Maud [27] ; Mouthon-Reignier, Capucine [28] ; Neau, Jean-Philippe [29] ; Ozsancak, Canan [30] ; Pereon, Yann [31] ; Perrotte, Paul [32] ; Puma, Angela [33, 34] ; Rajabally, Yusuf A. [35] ; Rinaldi, Simon [36] ; Rouaud, Violaine [37] ; Sole, Guilhem [38] ; Tard, Celine [39] ; Vallet, Anne-Evelyne [40] ; Vial, Christophe [41] ; Grp, Anti-FGFR3 Antibody Study
Número total de Autores: 51
Afiliação do(s) autor(es):
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[1] Inst Neuromyogene, Synaptopathies & Autoanticorps Synatac, St Priest En Jarez - France
[2] CHU St Etienne, Lab Biochem, F-42055 St Etienne - France
[3] CHU St Etienne, Dept Neurol, St Etienne - France
[4] Univ Campinas UNICAMP, Dept Neurol, Campinas, SP - Brazil
[5] Univ Paris Sud, Kremlin Bicetre Hosp, AP HP, FILNEMUS, Dept Neurol, Le Kremlin Bicetre - France
[6] Aix Marseille Univ, Univ Hosp La Timone, Reference Ctr Neuromuscular Disorders & ALS, Marseille - France
[7] Hosp Ctr Narbonne, Dept Neurol, Narbonne - France
[8] Valais Hosp, Cent Inst Hosp ICH, Dept Neurol, Sion - Switzerland
[9] Univ Hosp Angers, ALS Ctr, Dept Neurol, Angers - France
[10] Univ Hosp Reunion, Dept Neurol, St Pierre, Reunion - France
[11] Univ Hosp Clermont Ferrand, Dept Neurol, Clermont Ferrand - France
[12] Henri Mondor Univ Hosp, CHU Creteil, Dept Neurol, Creteil - France
[13] RivieraChablais Hosp, Dept Neurol, Vevey - Switzerland
[14] Univ Hosp Strasbourg, Dept Neurol, Strasbourg - France
[15] Toulon La Seyne Sur Mer Intercommunal Hosp Ctr, Dept Multidisciplinary Med, Toulon - France
[16] Univ Hosp Southampton NHS Fdn Trust, Wessex Neurosci Ctr, Southampton, Hants - England
[17] Univ Hosp Brest, Dept Neurol Funct Explorat, Brest - France
[18] Fdn Ophtalmol Adolphe Rothschild, Dept Neurol, Paris - France
[19] Univ Hosp Nimes, Dept Neurol, Nimes - France
[20] Univ Hosp Montpellier, Dept Neurol, Montpellier - France
[21] CHU Vaudois, Univ Lausanne Hosp, Dept Neurol, Lausanne - Switzerland
[22] Univ Hosp Grenoble, Dept Neurol, Grenoble - France
[23] Univ Hosp Pitie Salpetriere, Natl Referral Ctr Neuromuscular Dis, Paris - France
[24] Cornouailles Hosp Ctr, Dept Neurol, Quimper - France
[25] Univ Hosp Limoges, Reference Ctr Rare Peripheral Neuropathies, Dept Neurol, Limoges - France
[26] St Jean Hosp, Dept Neurol, Perpignan - France
[27] Univ Hosp Nancy, Dept Neurol, Nancy - France
[28] Sud Francilien Hosp Ctr, Dept Neurol, Corbeil Essonnes - France
[29] Univ Poitiers Hosp, Dept Neurol, Poitiers - France
[30] Reg Hosp Ctr Orleans, Dept Neurol, Orleans - France
[31] Hop Hotel Dieu, Ctr Neuromuscular Dis, Nantes - France
[32] Hosp Ctr Le Havre, Dept Neurol, Le Havre - France
[33] Univ Nice, Peripheral Nervous Syst Muscle & ALS Dept, Nice - France
[34] Univ Cote Azur, Nice - France
[35] Univ Hosp Birmingham, Queen Elizabeth Hosp, Reg Neuromuscular Clin, Birmingham, W Midlands - England
[36] Univ Oxford, John Radcliffe Hosp Oxford, Nuffield Dept Clin Neurosci, Oxford Autoimmune Neurol Grp, Oxford - England
[37] Bretagne Atlantique Hosp Ctr, Dept Neurol, Vannes - France
[38] Univ Hosp Bordeaux, Dept Neurol, Bordeaux - France
[39] Lille Univ, Med Ctr, Dept Neurol & Movement Disorders, Lille - France
[40] Hosp Ctr Vienne, Dept Neurol, Vienne - France
[41] Hosp Civils Lyon, Neuromuscular Reference Ctr, Bron - France
Número total de Afiliações: 41
Tipo de documento: Artigo Científico
Fonte: JOURNAL OF NEUROLOGY NEUROSURGERY AND PSYCHIATRY; v. 91, n. 1, p. 49-57, JAN 2019.
Citações Web of Science: 3
Resumo

Objective Sensory neuropathies (SNs) are often classified as idiopathic even if immunological mechanisms can be suspected. Antibodies against the intracellular domain of the fibroblast growth factor receptor 3 (FGFR3) possibly identify a subgroup of SN affecting mostly the dorsal root ganglion (DRG). The aim of this study was to identify the frequency of anti-FGFR3 antibodies and the associated clinical pattern in a large cohort of patients with SN. Methods A prospective, multicentric, European and Brazilian study included adults with pure SN. Serum anti-FGRF3 antibodies were analysed by ELISA. Detailed clinical and paraclinical data were collected for each anti-FGFR3-positive patient and as control for anti-FGFR3-negative patients from the same centres ('center-matched'). Results Sixty-five patients out of 426 (15%) had anti-FGFR3 antibodies, which were the only identified autoimmune markers in 43 patients (66%). The neuropathy was non-length dependent in 89% and classified as sensory neuronopathy in 64%, non-length-dependent small fibre neuropathy in 17% and other neuropathy in 19%. Specific clinical features occurred after 5-6 years of evolution including frequent paresthesia, predominant clinical and electrophysiological involvement of the lower limbs, and a less frequent mixed large and small fibre involvement. Brazilians had a higher frequency of anti-FGFR3 antibodies than Europeans (36% vs 13%, p<0.001), and a more frequent asymmetrical distribution of symptoms (OR 169, 95% CI 3.4 to 8424). Conclusions Anti-FGFR3 antibodies occur in a subgroup of SN probably predominantly affecting the DRG. Differences between Europeans and Brazilians could suggest involvement of genetic or environmental factors. (AU)

Processo FAPESP: 13/26410-0 - Estudo clínico, imunológico e neurofisiológico nas neuronopatias sensitivas
Beneficiário:Alberto Rolim Muro Martinez
Linha de fomento: Bolsas no Brasil - Doutorado Direto
Processo FAPESP: 13/01766-7 - Contribuição ao diagnóstico, à fisiopatologia e à terapêutica das neuronopatias sensitivas
Beneficiário:Marcondes Cavalcante Franca Junior
Linha de fomento: Auxílio à Pesquisa - Apoio a Jovens Pesquisadores