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Neurosarcoidosis-like reaction under TNF-α inhibitors: a case report and literature review of a paradoxical immune phenomenon.

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Title: Neurosarcoidosis-like reaction under TNF-α inhibitors: a case report and literature review of a paradoxical immune phenomenon.
Authors: Barbella, Gianvito (AUTHOR), Antenucci, Pietro (AUTHOR), Rosa, Marta (AUTHOR), Gozzi, Andrea (AUTHOR), Padroni, Marina (AUTHOR)
Source: Neurological Sciences. Apr2026, Vol. 47 Issue 4, p1-7. 7p.
Abstract: Background: Neurosarcoidosis (NS) is a rare manifestation of sarcoidosis that oftenrequires long-term immunosuppressive treatment (IST), including tumor necrosis factor-α (TNF-α) inhibitors in refractory cases. Paradoxically, TNF-α blockade has also been associated withsarcoidosis-like reactions (SLRs), granulomatous inflammatory conditions that mimic idiopathicsarcoidosis. Case presentation: We report a case of NS occurring in the context of a TNF-αinhibitor–associated SLR and review previously reported cases during TNF-α inhibitor therapy. Discussion: A 25- year-old man with HLA-B27–negative ankylosing spondylitis developed anacute central nervous system (CNS) inflammatory syndrome during prolonged adalimumabtherapy. The diagnosis was supported by inflammatory cerebrospinal fluid (CSF) findings,including an elevated CD4+/CD8+ ratio, histologically confirmed pulmonary SLR, and sustainedradiological and neurological remission after adalimumab withdrawal. A review of the literatureidentified only seven reported cases of NS during anti-TNF-α therapy across heterogeneousimmune-mediated inflammatory diseases. Clinical and neuroimaging features were variable,whereas CSF analysis consistently showed inflammatory changes. Exposure duration prior toneurological onset and follow-up strategies were inconsistently reported, and acute IST wasfrequently required because of CNS involvement. Conclusions: This case expands the clinicalspectrum of anti-TNF-α– associated SLRs and underscores the importance of considering aniatrogenic etiology in paradoxical neuroinflammatory presentations. Recognition of a“neurosarcoidosis-like reaction” may inform long-term therapeutic decisions, including carefulconsideration of TNF-α inhibitor re-exposure and selection of alternative ISTs for the underlyingdisease. [ABSTRACT FROM AUTHOR]
Copyright of Neurological Sciences is the property of Springer Nature and its content may not be copied or emailed to multiple sites without the copyright holder's express written permission. Additionally, content may not be used with any artificial intelligence tools or machine learning technologies. However, users may print, download, or email articles for individual use. This abstract may be abridged. No warranty is given about the accuracy of the copy. Users should refer to the original published version of the material for the full abstract. (Copyright applies to all Abstracts.)
Database: Psychology and Behavioral Sciences Collection
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DbLabel: Psychology and Behavioral Sciences Collection
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  Data: Neurosarcoidosis-like reaction under TNF-α inhibitors: a case report and literature review of a paradoxical immune phenomenon.
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  Data: <searchLink fieldCode="AR" term="%22Barbella%2C+Gianvito%22">Barbella, Gianvito</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Antenucci%2C+Pietro%22">Antenucci, Pietro</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Rosa%2C+Marta%22">Rosa, Marta</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Gozzi%2C+Andrea%22">Gozzi, Andrea</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Padroni%2C+Marina%22">Padroni, Marina</searchLink> (AUTHOR)
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  Data: <searchLink fieldCode="JN" term="%22Neurological+Sciences%22">Neurological Sciences</searchLink>. Apr2026, Vol. 47 Issue 4, p1-7. 7p.
– Name: Abstract
  Label: Abstract
  Group: Ab
  Data: Background: Neurosarcoidosis (NS) is a rare manifestation of sarcoidosis that oftenrequires long-term immunosuppressive treatment (IST), including tumor necrosis factor-α (TNF-α) inhibitors in refractory cases. Paradoxically, TNF-α blockade has also been associated withsarcoidosis-like reactions (SLRs), granulomatous inflammatory conditions that mimic idiopathicsarcoidosis. Case presentation: We report a case of NS occurring in the context of a TNF-αinhibitor–associated SLR and review previously reported cases during TNF-α inhibitor therapy. Discussion: A 25- year-old man with HLA-B27–negative ankylosing spondylitis developed anacute central nervous system (CNS) inflammatory syndrome during prolonged adalimumabtherapy. The diagnosis was supported by inflammatory cerebrospinal fluid (CSF) findings,including an elevated CD4+/CD8+ ratio, histologically confirmed pulmonary SLR, and sustainedradiological and neurological remission after adalimumab withdrawal. A review of the literatureidentified only seven reported cases of NS during anti-TNF-α therapy across heterogeneousimmune-mediated inflammatory diseases. Clinical and neuroimaging features were variable,whereas CSF analysis consistently showed inflammatory changes. Exposure duration prior toneurological onset and follow-up strategies were inconsistently reported, and acute IST wasfrequently required because of CNS involvement. Conclusions: This case expands the clinicalspectrum of anti-TNF-α– associated SLRs and underscores the importance of considering aniatrogenic etiology in paradoxical neuroinflammatory presentations. Recognition of a“neurosarcoidosis-like reaction” may inform long-term therapeutic decisions, including carefulconsideration of TNF-α inhibitor re-exposure and selection of alternative ISTs for the underlyingdisease. [ABSTRACT FROM AUTHOR]
– Name: AbstractSuppliedCopyright
  Label:
  Group: Ab
  Data: <i>Copyright of Neurological Sciences is the property of Springer Nature and its content may not be copied or emailed to multiple sites without the copyright holder's express written permission. Additionally, content may not be used with any artificial intelligence tools or machine learning technologies. However, users may print, download, or email articles for individual use. This abstract may be abridged. No warranty is given about the accuracy of the copy. Users should refer to the original published version of the material for the full abstract.</i> (Copyright applies to all Abstracts.)
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        Value: 10.1007/s10072-026-08955-z
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        Text: English
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              Text: Apr2026
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