Long-term outcomes of refractory neurosarcoidosis treated with infliximab

Aubart, F. Cohen; Bouvry, Diane; Galanaud, Damien; Dehais, Caroline; Mathey, Guillaume; Psimaras, Dimitri; Haroche, Julien; Pottier, Corinne; Hié, M.; Mathian, Alexis; Devilliers, H.; Nunès, Hilario; Valeyre, Dominique; Amoura, Zahir

doi:10.1007/s00415-017-8444-9

Cited by 91 publications

(60 citation statements)

References 21 publications

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“…10,33 Most of the data supporting their use is in the form of case series describing infliximab (a chimeric human-murine antibody) or adalimumab (a fully humanized antibody) for treatment of refractory neurosarcoidosis. [92][93][94][95][96] Infliximab is typically used with a cytotoxic medication to prevent formation of antichimeric antibodies. 83,97 Infliximab is more cumbersome, as it must be administered in a monitored infusion setting rather than self-administered subcutaneously.…”

Section: Tnf Antagonistsmentioning

confidence: 99%

Neurosarcoidosis

Neto

Moss

Willis

et al. 2017

Semin Respir Crit Care Med

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Neurosarcoidosis is an uncommon but clinically significant manifestation that will be routinely encountered in sarcoidosis specialty clinics. Overall, neurologic involvement is recognized in 5 to 10% of individuals with sarcoidosis. Neurologic symptoms will be the presenting manifestation of sarcoidosis in approximately one-half of those with neurosarcoidosis. The clinical and imaging features of neurosarcoidosis vary widely, largely depending on the anatomic distribution of the disease. The likelihood of spontaneous resolution is lower than for sarcoidosis in general, and residual functional deficits are not uncommon. Therefore, most patients with neurosarcoidosis require immunosuppressive therapy. Small fiber neuropathy, a recently recognized nongranulomatous parasarcoidosis syndrome, is prevalent in chronic sarcoidosis. The variety of neurologic manifestations, broad differential diagnosis, and complexity of management render neurosarcoidosis an area best served by multidisciplinary teams. Sarcoidologists, neurologists, radiologists, neurosurgeons, endocrinologists, urologists, physiatrists, and physical/occupational therapists all potentially have important roles.

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Section: Tnf Antagonistsmentioning

confidence: 99%

Neurosarcoidosis

Neto

Moss

Willis

et al. 2017

Semin Respir Crit Care Med

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“…In this cohort, infliximab was successfully used in 1 patient as a rescue therapy after glucocorticoids failure. The efficacy of infliximab as a rescue therapy for neurosarcoidosis was previously described in a few case reports and case series [23, 24] but no controlled study is available.…”

Section: Discussionmentioning

confidence: 99%

Characteristics and Long-Term Outcome of Neurosarcoidosis: A Population-Based Study from 1976-2013

Ungprasert

Crowson²,

Matteson³

2017

Neuroepidemiology

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Background/Aims: Neurosarcoidosis is a rare condition with serious health consequences. However, little is known about clinical characteristics and outcome of neurosarcoidosis in the community setting. Methods: Patients with neurosarcoidosis were identified from a previously described cohort of patients with incident sarcoidosis from Olmsted County, Minnesota, United States from 1976 to 2013 using individual medical record review. Data on clinical characteristics, treatment, and outcome were collected. Results: Neurological involvement by sarcoidosis occurred in 11 patients (3% of all patients with sarcoidosis). Cranial neuropathy was the most common type of neurological disease (5 patients; 45%) followed by peripheral neuropathy (3 patients; 27%), and meningitis (3 patients; 27%). Cerebrospinal fluid (CSF) pleocytosis and elevated CSF protein levels were observed in patients with meningitis, intramedullary spinal cord sarcoidosis, intracranial mass lesion and some patients with cranial neuropathy but were normal in patients with peripheral neuropathy. All patients received high-dose glucocorticoids as initial treatment and almost all responded to this therapy. Relapse after glucocorticoid dose reduction necessitated subsequent treatment with steroid-sparing agents in 4 patients. Conclusion: Neurosarcoidosis is an uncommon manifestation of sarcoidosis. Neurosarcoidosis manifestations generally responded well to high-dose glucocorticoids in the majority of patients, but relapse was common.

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“…Anti-TNF-α monoclonal antibodies have been proved to be effective in refractory cases of sarcoidosisrelated optic neuropathy. 170,171 The potential occurrence of demyelinating disease, such as multiple sclerosis, in patients receiving anti-TNF must be known by the treating physicians.…”

Section: Optic Nerve Involvementmentioning

confidence: 99%

Ocular Sarcoidosis

Sève

Jamilloux

Tilikete

et al. 2020

Semin Respir Crit Care Med

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Sarcoidosis is one of the leading causes of inflammatory eye disease. Any part of the eye and its adnexal tissues can be involved. Uveitis and optic neuropathy are the main manifestations, which may require systemic treatment. Two groups of patients with sarcoid uveitis can be distinguished: one of either sex and any ethnicity in which ophthalmological findings are various and another group of elderly Caucasian women with mostly chronic posterior uveitis. Clinically isolated uveitis revealing sarcoidosis remains a strictly ocular condition in a large majority of cases. Although it can be a serious condition involving functional prognosis, early recognition in addition to a growing therapeutic arsenal (including intravitreal implant) has improved the visual prognosis of the disease in recent years. Systemic corticosteroids are indicated when uveitis does not respond to topical corticosteroids or when there is bilateral posterior involvement, especially macular edema. In up to 30% of the cases that require an unacceptable dosage of corticosteroids to maintain remission, additional immunosuppression is used, especially methotrexate. As with other forms of severe noninfectious uveitis, monoclonal antibodies against tumor necrosis factor-α have been used. However, only very rarely does sarcoid uveitis fail to respond to combined corticosteroids and methotrexate therapy, a situation that should suggest either poor adherence or another granulomatous disease. Optic neuropathy often affects women of African and Caribbean origins. Some authors recommend that patients should be treated with high-dose of corticosteroids and concurrent immunosuppression from the onset of this manifestation, which is associated with a poorer outcome.

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Long-term outcomes of refractory neurosarcoidosis treated with infliximab

Cited by 91 publications

References 21 publications

Neurosarcoidosis

Neurosarcoidosis

Characteristics and Long-Term Outcome of Neurosarcoidosis: A Population-Based Study from 1976-2013

Ocular Sarcoidosis

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