Cristina Scarpazza1, Luca Prosperini2, Chiara R Mancinelli1, Nicola De Rossi1, Alessandra Lugaresi3, Marco Capobianco4, Lucia Moiola5, Paola Naldi6, Luisa Imberti7, Simonetta Gerevini8, Ruggero Capra9. 1. Multiple Sclerosis Centre, Spedali Civili di Brescia, Via Ciotti 154, 25018 Montichiari, Brescia, Italy. 2. Department of Neurology and Psychiatry, Sapienza University, Viale Dell'Università, 30, 00185 Rome, Italy. 3. Department of Biomedical and NeuroMotor Sciences (DIBINEM), Alma Mater Studiorum, Università di Bologna, Italy; IRCCS Istituto delle Scienze, "UOSI Riabilitazione Sclerosi Multipla", Via Altura 3, 40138 Bologna, Italy. 4. SCDO Neurologia 2, Centro di Riferimento Regionale Sclerosi Multipla, AOU San Luigi Gonzaga, Regione Gonzole 10, 10043 Orbassano, Torino, Italy. 5. Department of Neurology, San Raffaele Scientific Institute, Vita-Salute San Raffaele University, Via Olgettina 48, 20132 Milan, Italy. 6. Multiple Sclerosis Centre, Clinica Neurologica, AOU "Maggiore della Carità" Novara, Corso Mazzini 18, 28100 Novara, Italy. 7. Centro Ricerca Emato-oncologia AIL (CREA), Diagnostics Department, Spedali Civili of Brescia, Piazzale Spedali Civili, 1, 25123 Brescia, Italy. 8. Department of Neuroradiology, San Raffaele Scientific Institute, Vita-Salute San Raffaele University, Via Olgettina 60, 20132 Milan, Italy. 9. Multiple Sclerosis Centre, Spedali Civili di Brescia, Via Ciotti 154, 25018 Montichiari, Brescia, Italy. Electronic address: ruggero.capra@gmail.com.
Abstract
BACKGROUND: Despite the recent advances in the understanding of natalizumab (NTZ) related progressive multifocal leukoencephalopathy (PML) and its associated immune reconstitution inflammatory syndrome (PML-IRIS), the therapeutic options are still under investigated. In this context, the beneficial use of maraviroc is still an anecdotal observation. OBJECTIVE: To evaluate the impact of maraviroc in modifying the course of PML preventing IRIS or blunting IRIS manifestations. METHODS: Three patients with NTZ PML included in the Italian dataset of PML were treated with maraviroc. Their longitudinal clinical and radiological course was described in detail. RESULTS: The three patients were characterized by a steady clinical worsening not controlled by maraviroc. All the three patients manifested PML-IRIS, which emerged, respectively, at 62, 64 and 90days post NTZ withdrawal. This is in accordance with the data of the Italian dataset. Clinical and radiological stabilization of PML-IRIS occurred only after corticosteroids administration. CONCLUSION: In these three cases, maraviroc did not show any clear effect in modulating the clinical course of PML preventing IRIS. Moreover, once PML-IRIS emerged, the clinical stabilization was achieved only with the use of corticosteroids. Thus, the use of maraviroc should be regarded with extreme caution due the potential adverse events associated with its use.
BACKGROUND: Despite the recent advances in the understanding of natalizumab (NTZ) related progressive multifocal leukoencephalopathy (PML) and its associated immune reconstitution inflammatory syndrome (PML-IRIS), the therapeutic options are still under investigated. In this context, the beneficial use of maraviroc is still an anecdotal observation. OBJECTIVE: To evaluate the impact of maraviroc in modifying the course of PML preventing IRIS or blunting IRIS manifestations. METHODS: Three patients with NTZ PML included in the Italian dataset of PML were treated with maraviroc. Their longitudinal clinical and radiological course was described in detail. RESULTS: The three patients were characterized by a steady clinical worsening not controlled by maraviroc. All the three patients manifested PML-IRIS, which emerged, respectively, at 62, 64 and 90days post NTZ withdrawal. This is in accordance with the data of the Italian dataset. Clinical and radiological stabilization of PML-IRIS occurred only after corticosteroids administration. CONCLUSION: In these three cases, maraviroc did not show any clear effect in modulating the clinical course of PML preventing IRIS. Moreover, once PML-IRIS emerged, the clinical stabilization was achieved only with the use of corticosteroids. Thus, the use of maraviroc should be regarded with extreme caution due the potential adverse events associated with its use.