The impact of rituximab infusion protocol on the long-term outcome in anti-MuSK myasthenia gravis


Por: Cortes-Vicente, E, Rojas-Garcia, R, Diaz-Manera, J, Querol, L, Casasnovas, C, Guerrero-Sola, A, Munoz-Blanco, JL, Barcena-Llona, JE, Marquez-Infante, C, Pardo, J, Martinez-Fernandez, EM, Uson, M, Oliva-Nacarino, P, Sevilla, T, Illa, I

Publicada: 1 jun 2018
Resumen:
ObjectiveTo evaluate whether the clinical benefit and relapse rates in anti-muscle-specific kinase (MuSK) myasthenia gravis (MG) differ depending on the protocol of rituximab followed. MethodsThis retrospective multicentre study in patients with MuSK MG compared three rituximab protocols in terms of clinical status, relapse, changes in treatment, and adverse side effects. The primary effectiveness endpoint was clinical relapse requiring a further infusion of rituximab. Survival curves were estimated using Kaplan-Meier methods and survival analyses were undertaken using Cox proportional-hazards models. ResultsTwenty-five patients were included: 11 treated with protocol 4 + 2 (375 mg/m(2)/4 weeks, then monthly for 2 months), five treated with protocol 1 + 1 (two 1 g doses 2 weeks apart), and nine treated with protocol 4 (375 mg/m(2)/4 weeks). Mean follow-up was 5.0 years (SD 3.3). Relapse occurred in 18.2%, 80%, and 33.3%, and mean time to relapse was 3.5 (SD 1.5), 1.1 (SD 0.4), and 2.5 (SD 1.4) years, respectively. Based on Kaplan-Meier estimates, patients treated with protocol 4 + 2 had fewer and later relapses than patients treated with the other two protocols (log-rank test P = 0.0001). Patients treated with protocol 1 + 1 had a higher risk of relapse than patients treated with protocol 4 + 2 (HR 112.8, 95% CI, 5.7-2250.4, P = 0.002). Patients treated with protocol 4 showed a trend to a higher risk of relapse than those treated with protocol 4 + 2 (HR 9.2, 95% CI 0.9-91.8, P = 0.059). InterpretationThis study provides class IV evidence that the 4 + 2 rituximab protocol has a lower clinical relapse rate and produces a more durable response than the 1 + 1 and 4 protocols in patients with MuSK MG.

Filiaciones:
Cortes-Vicente, E:
 Univ Autonoma Barcelona, Hosp Santa Creu & St Pau, Dept Neurol, Neuromuscular Dis Unit, St Antoni Maria Claret 167, Barcelona 08025, Spain

 Ctr Biomed Network Res Rare Dis CIBERER, Madrid, Spain

Rojas-Garcia, R:
 Univ Autonoma Barcelona, Hosp Santa Creu & St Pau, Dept Neurol, Neuromuscular Dis Unit, St Antoni Maria Claret 167, Barcelona 08025, Spain

 Ctr Biomed Network Res Rare Dis CIBERER, Madrid, Spain

Diaz-Manera, J:
 Univ Autonoma Barcelona, Hosp Santa Creu & St Pau, Dept Neurol, Neuromuscular Dis Unit, St Antoni Maria Claret 167, Barcelona 08025, Spain

 Ctr Biomed Network Res Rare Dis CIBERER, Madrid, Spain

Querol, L:
 Univ Autonoma Barcelona, Hosp Santa Creu & St Pau, Dept Neurol, Neuromuscular Dis Unit, St Antoni Maria Claret 167, Barcelona 08025, Spain

 Ctr Biomed Network Res Rare Dis CIBERER, Madrid, Spain

Casasnovas, C:
 Hosp Univ Bellvitge IDIBELL, Neuromuscular Dis Unit, Dept Neurol, Barcelona, Spain

Guerrero-Sola, A:
 Hosp Univ Clin San Carlos, Dept Neurol, Neuromuscular Dis Unit, Madrid, Spain

Munoz-Blanco, JL:
 Hosp Gen Univ Gregorio Maranon, IISGM, Dept Neurol, ALS Neuromuscular Unit, Madrid, Spain

Barcena-Llona, JE:
 Hosp Univ Cruces, Dept Neurol, Baracaldo, Spain

Marquez-Infante, C:
 Hosp Univ Virgen Rocio, Dept Neurol & Neurophysiol, Neuromuscular Dis Unit, Seville, Spain

Pardo, J:
 Hosp Clin, Dept Neurol, Santiago De Compostela, Spain

Martinez-Fernandez, EM:
 Hosp Juan Raman Jimenez, Dept Neurol, Huelva, Spain

Uson, M:
 Hosp Son Llatzer, Dept Neurol, Palma De Mallorca, Spain

Oliva-Nacarino, P:
 Hosp Univ Cent Asturias, Dept Neurol, Oviedo, Spain

Sevilla, T:
 Ctr Biomed Network Res Rare Dis CIBERER, Madrid, Spain

 Hosp Univ & Politecn La Fe, Dept Neurol, Neuromuscular Unit, Valencia, Spain

 Univ Valencia, Dept Med, Valencia, Spain

Illa, I:
 Univ Autonoma Barcelona, Hosp Santa Creu & St Pau, Dept Neurol, Neuromuscular Dis Unit, St Antoni Maria Claret 167, Barcelona 08025, Spain

 Ctr Biomed Network Res Rare Dis CIBERER, Madrid, Spain
ISSN: 23289503
Editorial
WILEY, 111 RIVER ST, HOBOKEN 07030-5774, NJ USA, Estados Unidos America
Tipo de documento: Article
Volumen: 5 Número: 6
Páginas: 710-716
WOS Id: 000434349700005
ID de PubMed: 29928654
imagen Gold, Green Published

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