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Abstract Number: 1885

Biological Therapy in Refractory Neurobehçet’s Disease. Multicenter Study of 42 Patients

Alba Herrero-Morant1, José Luis Martin-Varillas2, Vanesa Calvo-Río3, Santos Castañeda4, Olga Maíz5, Ana Blanco6, Julio Sánchez7, Norberto Ortego8, Enrique Raya9, Anahy Maria Brandy-Garcia10, Alejandro Olive-Marques11, Agueda Prior-Español12, Clara Moriano13, Elvira Díez14, Rafael Melero15, Jenaro Enrique Grana Gil16, Álvaro Seijas-López17, Ana Urruticoechea-Arana18, Ángel Ramos-Calvo19, Concepcion Delgado-Beltran20, Marta Loredo-Martinez20, Eva Salgado21, Francisca Sivera22, Ignacio Torre23, Javier Narvaez24, Jose Luis Andreu25, Olga Martinez26, Ricardo Gómez de la Torre27, Sabela Fernandez-Aguado28, Susana Romero-Yuste29, Gerard Espinosa30, Miguel Ángel gonzalez-Gay31 and Ricardo Blanco3, 1Hospital Universitario Marqués de Valdecilla, Santander, Spain, 2Hospital Sierrallana, Torrelavega, Spain, 3Hospital Universitario Marqués de Valdecilla, IDIVAL, Santander, Spain, 4Hospital Universitario de la Princesa, Madrid, Spain, 5Hospital Universitario de Donostia, San Sebastián, Spain, 6Hospital Universitario de Donostia, San Sebastián, 7Hospital Universitario 12 de Octubre, Madrid, Spain, 8Medicine Department, Universidad de Granada, Granada, Spain, 9Hospital San Cecilio, Granada, Spain, 10Hospital Germans Trias i Pujol, Badalona, Spain, 11Rheumatology Service. Hospital Germans Trias i Pujol, Barcelona, Spain, 12Department of Rheumatology, Germans Trias i Pujol. University Hospital, Badalona, Spain, 13Hospital Universitario de León, León, Spain, 14Hospital de León, León, Spain, 15Complexo Hospitalario Universitario de Vigo, Vigo, Galicia, Spain, 16Hospital Universitario de A Coruña, A Corua, Spain, 17Hospital Universitario de A Coruña, A Coruña, Spain, 18H. Can Misses, Elvissa, Spain, 19Complejo Hospitalario de Soria, Soria, Spain, 20Hospital Clínico Lozano Blesa, Zaragoza, Spain, 21Complejo Hospitalario Universitario de Ourense, Ourense, Spain, 22Hospital Universitario de Elda, Alicante, Spain, 23Hospital Universitario de Basurto, Bilbao, Spain, 24Division of Rheumatology, Hospital Universitario de Bellvitge, Barcelona, Spain, 25Hospital Universitario Puerta de Hierro, Madrid, Spain, 26H. Salamanca, Zamora, Spain, 27H. Asturias, Oviedo, Spain, 28Hospital Universitario de Cabueñes, Gijón, Spain, 29Complejo Hospitalario Universitario de Pontevedra, Pontevedra, Spain, 30Hospital Clinic, Barcelona, Spain, 31Research group on Genetic Epidemiology and Atherosclerosis in Systemic Diseases and in Metabolic Bone Diseases of the Musculoskeletal System, IDIVAL, Division of Rheumatology, Hospital Universitario Marqués de Valdecilla; School of Medicine, Universidad de Cantabria, Santander, Spain. Cardiovascular Pathophysiology and Genomics Research Unit, School of Physiology, Faculty of Health Sciences, University of the Witwatersrand, Johannesburg, South Africa

Meeting: ACR Convergence 2021

Keywords: Behçet's Syndrome, Biologicals, neurology, Vasculitis

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Session Information

Date: Tuesday, November 9, 2021

Session Title: Vasculitis – Non-ANCA-Associated & Related Disorders Poster II (1862–1888)

Session Type: Poster Session D

Session Time: 8:30AM-10:30AM

Background/Purpose: Neurobehçet’s disease (NBD) is a severe complication of Behcet’s disease (BD). Despite well-established therapies, with glucocorticoids and conventional immunosuppressants (cIS) a significant proportion of patients are refractory. The aim of this study is to assess efficacy and safety of biologic therapy (BT) in NBD refractory to glucocorticoids and at least one cIS.

Methods: Open-label multicenter study of refractory NBD from 22 different referral Spanish Hospitals. The main outcome variables were safety and clinical response measured at baseline, 6, 12 and 24 months. Other outcome variables were improvement in analytical parameters and corticosteroid-sparing effect of biological therapy.

Results: We studied 42 patients (21 women/21 men; mean age 40.4±10.8 years). HLA B51 was positive in 15 (40.5%) out of 37 patients tested. Non-neurological manifestations were oral ulcers (n=41, 97.6%), genital ulcers (n=31, 73.8%), skin lesions (n=28, 66.7%), arthralgia (n=27, 64.3%), uveitis (n=21, 50.0%), arthritis (n=9, 21.4%), venous thrombosis (n=9, 21.4%) and arterial thrombosis (n=4, 9.5%). The underlying neurologic manifestation were parenchymal (n=34, 81 %) and non-parenchymal (n=17, 40.5%) involvement (TABLE). The first BT used was infliximab (n=20), adalimumab (n=13), golimumab (n=3), tocilizumab (n=3) and etanercept (n=2).

After 58.2±51.4 months since initiation of BT, neurological response was complete (n=27; 64.3%), or partial (11, 26.1%). Only 4 (9.5%) patients did not respond (FIGURE). After 6 months of BT, ESR improved from.31.5±25.6 to 15.3±11.9 mg/L (p=0.005) and CRP from 1.4 [0.2-12.8] to 0.3 [0.1-3] mg/L (p= 0.002). Likewise, a decrease in oral prednisone dose was also achieved from 45.6±17.3 mg/day at baseline to 5.17±2.85 mg/day after 24 months (p< 0.0001).

Primary failure was observed in 16 (38.1%) patients due to inefficacy (n=11, 68.8%) or adverse effects (n=5, 31.3%). Similarly, secondary failure was detected in 6 (14.3%) patients due to inefficacy (n=5, 83.3%) or adverse effects (n=1, 16.7%). No serious adverse effects were observed.

Conclusion: BT, especially monoclonal anti-TNF drugs, seems effective and safe in patients with refractory NBD.

TABLE: Underlying neurologic manifestation of 41 patients with refractory neurobehçet’s disease treated with biologic therapy

FIGURE: Neurological response after initiation of biological therapy


Disclosures: A. Herrero-Morant, None; J. Martin-Varillas, None; V. Calvo-Río, None; S. Castañeda, None; O. Maíz, None; A. Blanco, None; J. Sánchez, None; N. Ortego, None; E. Raya, None; A. Brandy-Garcia, None; A. Olive-Marques, None; A. Prior-Español, None; C. Moriano, None; E. Díez, None; R. Melero, None; J. Grana Gil, None; . Seijas-López, None; A. Urruticoechea-Arana, None; . Ramos-Calvo, None; C. Delgado-Beltran, None; M. Loredo-Martinez, None; E. Salgado, None; F. Sivera, None; I. Torre, None; J. Narvaez, None; J. Andreu, None; O. Martinez, None; R. Gómez de la Torre, None; S. Fernandez-Aguado, None; S. Romero-Yuste, Abbvie, 2, 6, MSD, 5, Pfizer, 5, Novartis, 5, Biogen, 2, 6, Amgen, 6, Grünenthal, 6, Kern Pharma, 6, Lilly, 2, 6, Roche, 6, Sandoz, 6, Sanofi, 6, UCB, 6, Janssen, 2, 6, Fresenius Kabi, 2, Gebro, 2, BMS, 2, 5, Galapagos, 2; G. Espinosa, None; M. gonzalez-Gay, None; R. Blanco, Brystol Myers Squibb, 6.

To cite this abstract in AMA style:

Herrero-Morant A, Martin-Varillas J, Calvo-Río V, Castañeda S, Maíz O, Blanco A, Sánchez J, Ortego N, Raya E, Brandy-Garcia A, Olive-Marques A, Prior-Español A, Moriano C, Díez E, Melero R, Grana Gil J, Seijas-López , Urruticoechea-Arana A, Ramos-Calvo , Delgado-Beltran C, Loredo-Martinez M, Salgado E, Sivera F, Torre I, Narvaez J, Andreu J, Martinez O, Gómez de la Torre R, Fernandez-Aguado S, Romero-Yuste S, Espinosa G, gonzalez-Gay M, Blanco R. Biological Therapy in Refractory Neurobehçet’s Disease. Multicenter Study of 42 Patients [abstract]. Arthritis Rheumatol. 2021; 73 (suppl 9). https://acrabstracts.org/abstract/biological-therapy-in-refractory-neurobehcets-disease-multicenter-study-of-42-patients/. Accessed February 2, 2023.
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