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SAT0523 (2020)
BIOLOGICAL THERAPY IN REFRACTORY ATYPICAL OPTIC NEURITIS. MULTICENTER STUDY
A. Herrero Morant1, C. Álvarez Reguera1, V. Calvo del Rio1, O. Maíz Alonso2, A. Blanco3, J. Narváez4, S. Castañeda5, E. Vicente5, S. Romero-Yuste6, R. Demetrio-Pablo7, A. Urruticoechea-Arana8, J. L. García Serrano9, J. L. Callejas Rubio9, N. Ortego9, J. Sánchez10, P. Estrada11, I. Rua-Figueroa12, D. Martínez-López1, J. L. Martín-Varillas1, M. Á. González-Gay1, R. Blanco1
1H. U. M. Valdecilla, Rheumatology, Santander, Spain
2H. Donostia, Rheumatology, Santander, Spain
3H. Donostia, Ophthalmology, San Sebastián, Spain
4H. Bellvitge, Rheumatology, Barcelona, Spain
5H. La princesa, Rheumatology, Madrid, Spain
6H. Pontevedra, Rheumatology, Pontevedra, Spain
7H. U. M. Valdecilla, Ophthalmology, Santander, Spain
8H. Can Misses, Rheumatology, Ibiza, Spain
9H. San Cecilio, Rheumatology, Granada, Spain
10H. 12 de Octubre, Rheumatology, Madrid, Spain
11H. Moises Broggi, Rheumatology, Barcelona, Spain
12H. Dr. Negrín, Rheumatology, Gran Canaria, Spain

Background: Optic Neuritis (ON) is an inflammation of the optic nerve. Its most common presentation is demyelinating typical ON. Atypical ON is rare, severe, non-demyelinating and can be isolated or associated to different diseases including autoimmune diseases. If it is not treated, it can lead to devastating visual results. Conventional treatment includes systemic corticosteroids and conventional immunosuppressants (CIS).


Objectives: Our aim was to assess the efficacy of biological therapy in atypical ON refractory to conventional treatment.


Methods: Open-label multicenter study including 19 patients diagnosed with atypical ON refractory to systemic corticosteroids and at least one CIS. The main outcomes assessed were Best Corrected Visual Acuity (BCVA) and optic nerve and ganglionar cells Optical Coherence Tomography (OCT). These outcome variables were recorded at baseline, 1 week, 2 weeks, 1 month, 3 months and 6 months and 1 year after biological therapy onset.


Results: We studied 19 patients (12 women/7 men); mean age of 34.8 ± 13.9 years. The underlying diseases were idiopathic (n=7), Behçet´s disease (n=5), systemic lupus erythematosus (n=2), neuromyelitis optica (n=3), sarcoidosis (n=1) and relapsing polychondritis (n=1) (TABLE). Before biological therapy and besides systemic corticosteroids, patients had received different CIS. Biological therapy was adalimumab (n=6), rituximab (n=6), infliximab (n=5) and tocilizumab (n=4). After biological therapy, an improvement in ocular parameters was observed: BCVA [0.7±0.3 to 0.8±0.3; p= 0.03], optic nerve OCT [123.2±58.3 to 190.5±175.4; p= 0.11], and ganglionar cells OCT [369.6±137.4 to 270.7±23.2; p= 0.03] at one year (FIGURE ). After a mean follow-up of 29.1 ±19.2 months, there were no severe adverse effects observed.


Conclusion: Biological therapy may be effective in patients with refractory atypical ON.

Case Gender/ Age Underlying disease Laterality IV steroids dose (g) Maximum prednisone oral dose (g) Conventional immunosuppressants Biological therapy Adverse effects
1 F/29 Idiopathic Unilateral 4 60 AZA TCZ No
2 F/26 Idiopathic Bilateral 5.5 30 AZA TCZ No
3 F/13 Idiopathic Bilateral - 10 MTX ADA No
4 F/25 Idiopathic Bilateral 4 60 MTX IFX, TCZ No
5 F/24 Idiopathic Bilateral 0.5 60 MTX, AZA ADA No
6 M/14 Idiopathic Bilateral - 10 MTX ADA No
7 F/30 Vasculitis ANCA+ Unilateral 3 60 AZA, MMF, LFM, CFM RTX Yes
8 M/21 Behçet Bilateral - 60 MTX, AZA ADA Nausea Vomits
9 M/25 Behçet Unilateral 0.5 60 MTX, CyA ADA No
10 M/39 Behçet Unilateral 3 80 MTX, MMF IFX No
11 M/40 Behçet Unilateral - 80 MMF IFX No
12 M/37 Behçet Unilateral - 60 CyA IFX No
13 F/68 NMO Unilateral 2.5 30 CFM, AZA RTX No
14 F/41 NMO Unilateral 3 60 CFM RTX Infection
15 F/43 NMO Bilateral 5 60 AZA RTX Infusion reaction
16 F/56 SLE Unilateral - 60 HCQ, MMF, CFM RTX No
17 F/47 SLE Unilateral 5 60 HCQ, MMF RTX No
18 F/43 Relapsing polychondritis Bilateral 3 60 MTX, CFM IFX, TCZ No
19 M/41 Sarcoidosis Bilateral 3 40 AZA ADA No

Disclosure of Interests: Alba Herrero Morant: None declared, Carmen Álvarez Reguera: None declared, Vanesa Calvo del Rio Grant/research support from: MSD and Roche, Speakers bureau: Abbott, Lilly, Celgene, Grünenthal, UCB Pharma, Olga Maíz Alonso: None declared, Ana Blanco Speakers bureau: Abbvie, J. Narváez: None declared, Santos Castañeda: None declared, Esther Vicente Speakers bureau: BMS, Roche., Susana Romero-Yuste: None declared, Rosalía Demetrio-Pablo: None declared, ANA URRUTICOECHEA-ARANA: None declared, J. L. García Serrano: None declared, J. L. Callejas Rubio: None declared, Norberto Ortego: None declared, Julio Sánchez: None declared, Paula Estrada: None declared, Iñigo Rua-Figueroa: None declared, David Martínez-López: None declared, José Luis Martín-Varillas Grant/research support from: AbbVie, Pfizer, Janssen and Celgene, Speakers bureau: Pfizer and Lilly, Miguel Á. González-Gay Grant/research support from: AbbVie, MSD and Roche, Speakers bureau: AbbVie, MSD and Roche, Ricardo Blanco Grant/research support from: Abbvie, MSD and Roche, Consultant of: Abbvie, Pfizer, Roche, Bristol-Myers, Janssen and MSD, Speakers bureau: Abbvie, Pfizer, Roche, Bristol-Myers, Janssen, Lilly and MSD

Citation: Ann Rheum Dis, volume 79, supplement 1, year 2020, page 1213
Session: Other orphan diseases (Poster Presentations)