Clinically reversible ustekinumab-induced encephalopathy: case report and review of the literature Article Swipe
Jordi Sarto
,
Berta Caballol
,
Juan Berenguer
,
Ibán Aldecoa
,
Álvaro Carbayo
,
Daniel Santana
,
Iván Archilla
,
Carles Gaig
,
Francesc Graus
,
Julián Panés
,
Albert Saiz
·
YOU?
·
· 2022
· Open Access
·
· DOI: https://doi.org/10.1177/17562864221079682
YOU?
·
· 2022
· Open Access
·
· DOI: https://doi.org/10.1177/17562864221079682
Ustekinumab, a monoclonal antibody against interleukin (IL)-12 and IL-23 approved for the treatment of Crohn’s disease, has shown to be an effective therapy with a favourable safety profile. Clinical trials and real-world studies have reported very few neurological adverse events, including posterior reversible encephalopathy syndrome, idiopathic intracranial hypertension and headache. We describe the case of a 48-year-old man with Crohn’s disease who initiated treatment with ustekinumab on top of ongoing treatment with methotrexate 25 mg/week who presented with an acute-onset encephalopathy that rapidly evolved to severe tetraparesis and akinetic mutism, associated with extensive leukoencephalopathy and restricted diffusion on brain magnetic resonance imaging (MRI), 1 month after the second dose of ustekinumab. Comprehensive in-patient diagnostic testing ruled out vascular, demyelinating, metabolic, tumoral and infectious etiologies. Brain biopsy showed patchy infiltrates of foamy histiocytes with perivascular distribution, associated with edema, diffuse astrocytic gliosis and focal perivascular axonal destruction without demyelination, and ustekinumab-induced neurotoxicity was suspected. After drug discontinuation, the patient presented a complete clinical recovery despite the persistence of leukoencephalopathy. In conclusion, in an era in which biological therapies are continually evolving and expanding, knowledge about the potential neurotoxicity of these new therapies and their management becomes crucial. Although ustekinumab-induced encephalopathy is uncommon, the recognition of this potentially serious side effect is important because prompt withdrawal is associated with a favourable outcome. Whether methotrexate played an additional contributing role is currently unknown, but it is a factor that should be considered.
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- Type
- article
- Language
- en
- Landing Page
- https://doi.org/10.1177/17562864221079682
- https://journals.sagepub.com/doi/pdf/10.1177/17562864221079682
- OA Status
- gold
- Cited By
- 7
- References
- 34
- Related Works
- 10
- OpenAlex ID
- https://openalex.org/W4213446799
All OpenAlex metadata
Raw OpenAlex JSON
- OpenAlex ID
-
https://openalex.org/W4213446799Canonical identifier for this work in OpenAlex
- DOI
-
https://doi.org/10.1177/17562864221079682Digital Object Identifier
- Title
-
Clinically reversible ustekinumab-induced encephalopathy: case report and review of the literatureWork title
- Type
-
articleOpenAlex work type
- Language
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enPrimary language
- Publication year
-
2022Year of publication
- Publication date
-
2022-01-01Full publication date if available
- Authors
-
Jordi Sarto, Berta Caballol, Juan Berenguer, Ibán Aldecoa, Álvaro Carbayo, Daniel Santana, Iván Archilla, Carles Gaig, Francesc Graus, Julián Panés, Albert SaizList of authors in order
- Landing page
-
https://doi.org/10.1177/17562864221079682Publisher landing page
- PDF URL
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https://journals.sagepub.com/doi/pdf/10.1177/17562864221079682Direct link to full text PDF
- Open access
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YesWhether a free full text is available
- OA status
-
goldOpen access status per OpenAlex
- OA URL
-
https://journals.sagepub.com/doi/pdf/10.1177/17562864221079682Direct OA link when available
- Concepts
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Medicine, Ustekinumab, Encephalopathy, Adverse effect, Progressive multifocal leukoencephalopathy, Posterior reversible encephalopathy syndrome, Pathology, Discontinuation, Disease, Internal medicine, Magnetic resonance imaging, Adalimumab, Radiology, Multiple sclerosis, ImmunologyTop concepts (fields/topics) attached by OpenAlex
- Cited by
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7Total citation count in OpenAlex
- Citations by year (recent)
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2025: 3, 2024: 1, 2023: 2, 2022: 1Per-year citation counts (last 5 years)
- References (count)
-
34Number of works referenced by this work
- Related works (count)
-
10Other works algorithmically related by OpenAlex
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| abstract_inverted_index.Comprehensive | 111 |
| abstract_inverted_index.distribution, | 134 |
| abstract_inverted_index.neurotoxicity | 150, 186 |
| abstract_inverted_index.demyelinating, | 118 |
| abstract_inverted_index.demyelination, | 147 |
| abstract_inverted_index.encephalopathy | 43, 80, 198 |
| abstract_inverted_index.discontinuation, | 155 |
| abstract_inverted_index.leukoencephalopathy | 93 |
| abstract_inverted_index.ustekinumab-induced | 149, 197 |
| abstract_inverted_index.leukoencephalopathy. | 167 |
| cited_by_percentile_year.max | 98 |
| cited_by_percentile_year.min | 89 |
| countries_distinct_count | 1 |
| institutions_distinct_count | 11 |
| sustainable_development_goals[0].id | https://metadata.un.org/sdg/3 |
| sustainable_development_goals[0].score | 0.8500000238418579 |
| sustainable_development_goals[0].display_name | Good health and well-being |
| citation_normalized_percentile.value | 0.75417991 |
| citation_normalized_percentile.is_in_top_1_percent | False |
| citation_normalized_percentile.is_in_top_10_percent | False |