Spencer K Hutto1,2, Kevin Kyle3, Julien J Cavanagh4,3, Haatem Reda3, Nagagopal Venna3. 1. Division of Hospital Neurology, Department of Neurology, Emory University School of Medicine, 12 Executive Park Dr NE, Atlanta, GA, 30329, USA. shutto@emory.edu. 2. Division of Neuroimmunology and Neuroinfectious Diseases, Department of Neurology, Massachusetts General Hospital, Harvard Medical School, Boston, MA, USA. shutto@emory.edu. 3. Division of Neuroimmunology and Neuroinfectious Diseases, Department of Neurology, Massachusetts General Hospital, Harvard Medical School, Boston, MA, USA. 4. Division of Hospital Neurology, Department of Neurology, Emory University School of Medicine, 12 Executive Park Dr NE, Atlanta, GA, 30329, USA.
Abstract
BACKGROUND: Tumor necrosis factor (TNF) alpha is critical in the development of granulomas and multiple recent reports have highlighted the role of infliximab, an infused TNF alpha inhibitor, in the treatment of neurosarcoidosis. As a self-injected TNF alpha inhibitor, adalimumab has certain advantages over infused medications, including greater patient freedom and autonomy. Experience with adalimumab is not well reported in the literature. OBJECTIVE: To report clinical experience with adalimumab in the treatment of central nervous system (CNS) sarcoidosis by combining observations in our center with those that have been reported in the literature. METHODS: Patients were identified from the Mass General Brigham Research Patient Data Registry and in the literature by searching PubMed. Patients with CNS manifestations of sarcoidosis treated with adalimumab were included for retrospective review and analyzed for baseline characteristics, treatment indications, outcomes, and adverse effects. RESULTS: Adalimumab was commonly started after failure of or intolerance to infliximab and methotrexate. Of those with adequate follow-up, 5/10 ultimately improved, remission was maintained in 3/10, and 2/10 with active disease remained stable without further worsening. One patient suffered a relapse, likely multifactorial in etiology, but has remained relapse free on adalimumab for 10 months subsequently. Three patients ultimately discontinued adalimumab. CONCLUSIONS: Preliminary evidence suggests that adalimumab may be a reasonable therapeutic option for patients with neurosarcoidosis affecting the CNS, including those with medically refractory disease.
BACKGROUND: Tumor necrosis factor (TNF) alpha is critical in the development of granulomas and multiple recent reports have highlighted the role of infliximab, an infused TNF alpha inhibitor, in the treatment of neurosarcoidosis. As a self-injected TNF alpha inhibitor, adalimumab has certain advantages over infused medications, including greater patient freedom and autonomy. Experience with adalimumab is not well reported in the literature. OBJECTIVE: To report clinical experience with adalimumab in the treatment of central nervous system (CNS) sarcoidosis by combining observations in our center with those that have been reported in the literature. METHODS: Patients were identified from the Mass General Brigham Research Patient Data Registry and in the literature by searching PubMed. Patients with CNS manifestations of sarcoidosis treated with adalimumab were included for retrospective review and analyzed for baseline characteristics, treatment indications, outcomes, and adverse effects. RESULTS: Adalimumab was commonly started after failure of or intolerance to infliximab and methotrexate. Of those with adequate follow-up, 5/10 ultimately improved, remission was maintained in 3/10, and 2/10 with active disease remained stable without further worsening. One patient suffered a relapse, likely multifactorial in etiology, but has remained relapse free on adalimumab for 10 months subsequently. Three patients ultimately discontinued adalimumab. CONCLUSIONS: Preliminary evidence suggests that adalimumab may be a reasonable therapeutic option for patients with neurosarcoidosis affecting the CNS, including those with medically refractory disease.
Authors: L Riancho-Zarrabeitia; M Delgado-Alvarado; J Riancho; A Oterino; M J Sedano; J Rueda-Gotor; I Pérez-Martín; M C González-Vela; J Berciano; M A González-Gay; R Blanco Journal: Clin Exp Rheumatol Date: 2013-12-09 Impact factor: 4.473
Authors: Jeanne Gosselin; Chantal Roy-Hewitson; Sean S M Bullis; John C DeWitt; Bruno P Soares; Sidarth Dasari; Alana Nevares Journal: Curr Rheumatol Rep Date: 2022-10-12 Impact factor: 4.686