Clinical characterisation of sensory neuropathy with anti-FGFR3 autoantibodies

Autor(es):Mostrar menos -

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

Afiliação do(s) autor(es):Mostrar menos -

^[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

• 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
• Modalidade de apoio: 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
• Modalidade de apoio: Auxílio à Pesquisa - Jovens Pesquisadores