Infliximab for the treatment of CNS sarcoidosis: A multi-institutional series

Jeffrey M Gelfand; Michael J Bradshaw; Barney J Stern; David B Clifford; Yunxia Wang; Tracey A Cho; Laura L Koth; Stephen L Hauser; Jason Dierkhising; NgocHanh Vu; Subramaniam Sriram; Harold Moses; Francesca Bagnato; Jeffrey A Kaufmann; Deidre J Ammah; Tsion H Yohannes; Mark J Hamblin; Nagagopal Venna; Ari J Green; Siddharama Pawate

doi:10.1212/WNL.0000000000004644

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Infliximab for the treatment of CNS sarcoidosis: A multi-institutional series

Journal article

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Infliximab for the treatment of CNS sarcoidosis: A multi-institutional series

Jeffrey M Gelfand, Michael J Bradshaw, Barney J Stern, David B Clifford, Yunxia Wang, Tracey A Cho, Laura L Koth, Stephen L Hauser, Jason Dierkhising, NgocHanh Vu, …

Neurology, Vol.89(20), pp.2092-2100

11/14/2017

DOI: 10.1212/WNL.0000000000004644

PMCID: PMC5711506

PMID: 29030454

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Abstract

To describe clinical and imaging responses in neurosarcoidosis to infliximab, a monoclonal antibody against tumor necrosis factor-α. 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. 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. Most patients with CNS sarcoidosis treated with infliximab exhibit favorable imaging and clinical treatment responses, including some previously refractory to other immunosuppressive treatments. This study provides Class IV evidence that for patients with CNS sarcoidosis infliximab is associated with favorable imaging and clinical responses.

Outcome Assessment (Health Care)

Humans

Middle Aged

Sarcoidosis - drug therapy

Male

Infliximab - pharmacology

Infliximab - administration & dosage

Young Adult

Infliximab - adverse effects

Central Nervous System Diseases - drug therapy

Tumor Necrosis Factor-alpha - immunology

Adult

Female

Immunosuppressive Agents - adverse effects

Aged

Immunosuppressive Agents - pharmacology

Immunosuppressive Agents - administration & dosage

Details

Title: Subtitle: Infliximab for the treatment of CNS sarcoidosis: A multi-institutional series
Creators: Jeffrey M Gelfand - 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
Michael J Bradshaw - 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
Barney J Stern - 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
David B Clifford - 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
Yunxia Wang - 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
Tracey A Cho - 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
Laura L Koth - 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
Stephen L Hauser - 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
Jason Dierkhising - 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
NgocHanh Vu - 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
Subramaniam Sriram - 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
Harold Moses - 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
Francesca Bagnato - 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
Jeffrey A Kaufmann - 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
Deidre J Ammah - 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
Tsion H Yohannes - 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
Mark J Hamblin - 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
Nagagopal Venna - 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
Ari J Green - 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 - 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
Resource Type: Journal article
Publication Details: Neurology, Vol.89(20), pp.2092-2100
Publisher: United States
DOI: 10.1212/WNL.0000000000004644
PMID: 29030454
PMCID: PMC5711506
ISSN: 0028-3878
eISSN: 1526-632X
Language: English
Date published: 11/14/2017
Academic Unit: Neurology; Iowa Neuroscience Institute
Record Identifier: 9984013114102771

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