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Clinical characterisation of sensory neuropathy with anti-FGFR3 autoantibodies

Clinical characterisation of sensory neuropathy with anti-FGFR3 autoantibodies
Clinical characterisation of sensory neuropathy with anti-FGFR3 autoantibodies
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.
0022-3050
1-9
Tholance, Yannick
f36f75bd-8f5a-482c-9224-767b5e90434a
Moritz, Christian Peter
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Rosier, Carole
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Ferraud, Karine
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Lassablière, François
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Reynaud-Federspiel, Evelyne
2203ffda-d053-4421-8f8a-db4d216b06f0
França Jr, Marcondes C.
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Martinez, Alberto R.M.
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Camdessanché, Jean-Philippe
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Antoine, Jean-Christophe
89fa0974-f2f6-4a48-b4c1-ea8739328570
Galea, Ian
66209a2f-f7e6-4d63-afe4-e9299f156f0b
Anti-FGFR3 antibody Study Group
Tholance, Yannick
f36f75bd-8f5a-482c-9224-767b5e90434a
Moritz, Christian Peter
fa896948-260b-48ac-a986-5aeae211fb6d
Rosier, Carole
4f554fbf-f31f-4906-8286-f4f4db73f91d
Ferraud, Karine
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Lassablière, François
f5dae072-6a68-47db-a61f-1146a4034ae4
Reynaud-Federspiel, Evelyne
2203ffda-d053-4421-8f8a-db4d216b06f0
França Jr, Marcondes C.
31e8b208-e5de-4cfb-b008-347b0160c6dd
Martinez, Alberto R.M.
27d665e0-b120-4857-b45b-04d6802cc34b
Camdessanché, Jean-Philippe
14c289af-226f-40b9-9900-c6fea73e6858
Antoine, Jean-Christophe
89fa0974-f2f6-4a48-b4c1-ea8739328570
Galea, Ian
66209a2f-f7e6-4d63-afe4-e9299f156f0b

Tholance, Yannick, Moritz, Christian Peter, Rosier, Carole, Ferraud, Karine, Lassablière, François, Reynaud-Federspiel, Evelyne, França Jr, Marcondes C., Martinez, Alberto R.M., Camdessanché, Jean-Philippe, Antoine, Jean-Christophe and Galea, Ian , Anti-FGFR3 antibody Study Group (2019) Clinical characterisation of sensory neuropathy with anti-FGFR3 autoantibodies. Journal of Neurology Neurosurgery and Psychiatry, 1-9. (doi:10.1136/jnnp-2019-321849).

Record type: Article

Abstract

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.

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Accepted/In Press date: 28 October 2019
e-pub ahead of print date: 5 November 2019

Identifiers

Local EPrints ID: 436041
URI: http://eprints.soton.ac.uk/id/eprint/436041
ISSN: 0022-3050
PURE UUID: ffd4dde7-30ce-4952-b117-b98c7c23389c
ORCID for Ian Galea: ORCID iD orcid.org/0000-0002-1268-5102

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Date deposited: 26 Nov 2019 17:30
Last modified: 10 Jan 2022 02:47

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Contributors

Author: Yannick Tholance
Author: Christian Peter Moritz
Author: Carole Rosier
Author: Karine Ferraud
Author: François Lassablière
Author: Evelyne Reynaud-Federspiel
Author: Marcondes C. França Jr
Author: Alberto R.M. Martinez
Author: Jean-Philippe Camdessanché
Author: Jean-Christophe Antoine
Author: Ian Galea ORCID iD
Corporate Author: Anti-FGFR3 antibody Study Group

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