Jeffrey M Gelfand1, Michael J Bradshaw1, Barney J Stern1, David B Clifford1, Yunxia Wang1, Tracey A Cho1, Laura L Koth1, Stephen L Hauser1, Jason Dierkhising1, NgocHanh Vu1, Subramaniam Sriram1, Harold Moses1, Francesca Bagnato1, Jeffrey A Kaufmann1, Deidre J Ammah1, Tsion H Yohannes1, Mark J Hamblin1, Nagagopal Venna1, Ari J Green1, Siddharama Pawate2. 1. From the Department of Neurology (J.M.G., S.L.H., J.D., A.J.G.), Division of Pulmonary and Critical Care, Department of Medicine (L.L.K.), and Department of Ophthalmology (A.J.G.), University of California San Francisco; Department of Neurology (M.J.B., N.V., S.S., H.M., F.B., S.P.), Vanderbilt University Medical Center, Nashville, TN; Department of Neurology (B.J.S., J.A.K., D.J.A.), University of Maryland Medical Center, Baltimore; Departments of Neurology and Medicine (D.B.C., T.H.Y.), Washington University in St. Louis, MO; Department of Neurology (Y.W., M.J.H.), University of Kansas Medical Center, Kansas City; and Department of Neurology (T.A.C., N.V.), Massachusetts General Hospital, Boston. 2. From the Department of Neurology (J.M.G., S.L.H., J.D., A.J.G.), Division of Pulmonary and Critical Care, Department of Medicine (L.L.K.), and Department of Ophthalmology (A.J.G.), University of California San Francisco; Department of Neurology (M.J.B., N.V., S.S., H.M., F.B., S.P.), Vanderbilt University Medical Center, Nashville, TN; Department of Neurology (B.J.S., J.A.K., D.J.A.), University of Maryland Medical Center, Baltimore; Departments of Neurology and Medicine (D.B.C., T.H.Y.), Washington University in St. Louis, MO; Department of Neurology (Y.W., M.J.H.), University of Kansas Medical Center, Kansas City; and Department of Neurology (T.A.C., N.V.), Massachusetts General Hospital, Boston. siddharama.pawate@vanderbilt.edu.
Abstract
OBJECTIVE: To describe clinical and imaging responses in neurosarcoidosis to infliximab, a monoclonal antibody against tumor necrosis factor-α. METHODS: Investigators at 6 US centers retrospectively identified patients with CNS sarcoidosis treated with infliximab, including only patients with definite or probable neurosarcoidosis following rigorous exclusion of other causes. RESULTS: Of 66 patients with CNS sarcoidosis (27 definite, 39 probable) treated with infliximab for a median of 1.5 years, the mean age was 47.5 years at infliximab initiation (SD 11.7, range 24-71 years); 56.1% were female; 62.1% were white, 37.0% African American, and 3% Hispanic. Sarcoidosis was isolated to the CNS in 19.7%. Using infliximab doses ranging from 3 to 7 mg/kg every 4-8 weeks, MRI evidence of a favorable treatment response was observed in 82.1% of patients with imaging follow-up (n = 56), with complete remission of active disease in 51.8% and partial MRI improvement in 30.1%; MRI worsened in 1 patient (1.8%). There was clinical improvement in 77.3% of patients, with complete neurologic recovery in 28.8%, partial improvement in 48.5%, clinical stability in 18.2%, worsening in 3%, and 1 lost to follow-up. In 16 patients in remission when infliximab was discontinued, the disease recurred in 9 (56%), typically in the same neuroanatomic location. CONCLUSIONS: Most patients with CNS sarcoidosis treated with infliximab exhibit favorable imaging and clinical treatment responses, including some previously refractory to other immunosuppressive treatments. CLASSIFICATION OF EVIDENCE: This study provides Class IV evidence that for patients with CNS sarcoidosis infliximab is associated with favorable imaging and clinical responses.
OBJECTIVE: To describe clinical and imaging responses in neurosarcoidosis to infliximab, a monoclonal antibody against tumor necrosis factor-α. METHODS: Investigators at 6 US centers retrospectively identified patients with CNS sarcoidosis treated with infliximab, including only patients with definite or probable neurosarcoidosis following rigorous exclusion of other causes. RESULTS: Of 66 patients with CNS sarcoidosis (27 definite, 39 probable) treated with infliximab for a median of 1.5 years, the mean age was 47.5 years at infliximab initiation (SD 11.7, range 24-71 years); 56.1% were female; 62.1% were white, 37.0% African American, and 3% Hispanic. Sarcoidosis was isolated to the CNS in 19.7%. Using infliximab doses ranging from 3 to 7 mg/kg every 4-8 weeks, MRI evidence of a favorable treatment response was observed in 82.1% of patients with imaging follow-up (n = 56), with complete remission of active disease in 51.8% and partial MRI improvement in 30.1%; MRI worsened in 1 patient (1.8%). There was clinical improvement in 77.3% of patients, with complete neurologic recovery in 28.8%, partial improvement in 48.5%, clinical stability in 18.2%, worsening in 3%, and 1 lost to follow-up. In 16 patients in remission when infliximab was discontinued, the disease recurred in 9 (56%), typically in the same neuroanatomic location. CONCLUSIONS: Most patients with CNS sarcoidosis treated with infliximab exhibit favorable imaging and clinical treatment responses, including some previously refractory to other immunosuppressive treatments. CLASSIFICATION OF EVIDENCE: This study provides Class IV evidence that for patients with CNS sarcoidosis infliximab is associated with favorable imaging and clinical responses.
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