Online first
Current issue
Archive
Special Issues
About the Journal
Publication Ethics
Anti-Plagiarism system
Instructions for Authors
Instructions for Reviewers
Editorial Board
Editorial Office
Contact
Reviewers
All Reviewers
2024
2023
2022
2021
2020
2019
2018
2017
2016
General Data Protection Regulation (RODO)
CASE REPORT
IRIS without PML in MS patients – a case report of IRIS after past alemtuzumab therapy with subsequent oral cladribine treatment and review of the literature
1
Department of Neurology, Medical University, Lublin, Poland
2
Interventional Radiology and Neuroradiology, Medical University, Lublin, Poland
Corresponding author
Anna Jamroz-Wiśniewska
Department of Neurology, Medical University of Lublin, Jaczewskiego 8, 20-090, Lublin, Poland
Department of Neurology, Medical University of Lublin, Jaczewskiego 8, 20-090, Lublin, Poland
KEYWORDS
immune reconstitution inflammatory syndrome (IRIS)alemtuzumabhighly-active multiple sclerosisoral cladribine
TOPICS
ABSTRACT
Alemtuzumab is a high-efficacy drug for relapsing-remitting multiple sclerosis (MS), leading to non-selective reconstitution of the immune system. Immune reconstitution inflammatory syndrome (IRIS) develops due to reconstruction of cellular immunity and inflammation in the CNS, mostly after progressive multifocal leukoencephalopathy (PML). A case of a 42-year-old patient is presented treated in the past with alemtuzumab because of highly active MS. He was admitted after 8 years of remission because of aphasia, bulbar syndrome and severe agitation. MRI of the brain showed multiple big confluencing lesions supra- and infratentorially, with mass effect and contrast enhancement. IRIS was diagnosed. Prolonged steroids therapy and plasma exchange were used with success. IRIS can also be a complication of natalizumab or fingolimod withdrawal and arise from the restoration of the previously suppressed immune response. Other cases of IRIS in MS patients without preceding PML are also reviewed.
REFERENCES (21)
1.
Guarnera C, Bramanti P, Mazzon E. Alemtuzumab: a review of efficacy and risks in the treatment of relapsing remitting multiple sclerosis. Ther Clin Risk Manag. 2017;13:871–879. doi:10.2147/TCRM.S134398.
2.
Devonshire V, Phillips R, Wass H, et al. Monitoring and management of autoimmunity in multiple sclerosis patients treated with alemtuzumab: practical recommendations. J Neurol. 2018;265:2494–2505. https://doi.org/10.1007/s00415....
3.
Rolla S, Maglione A, De Mercanti SF, et al. The Meaning of Immune Reconstitution after Alemtuzumab Therapy in Multiple Sclerosis. Cells. 2020;9(6):1396. doi:10.3390/cells9061396.
4.
Bowen L, Nath A, Smith B. CNS immune reconstitution inflammatory syndrome. Handb Clin Neurol. 2018;152:167–176. doi:10.1016/B978 0 444 63849 6.00013 X.
5.
Tugemann B. The risk of PML from natalizumab. Lancet Neurol. 2019;18(3):230. doi:10.1016/S1474-4422(19)30021-3.
6.
Kułakowska A, Mirowska-Guzel D, Adamczyk-Sowa M, et al. Recommendation of Polish clinical experts on the strategy of treatment with cladribine tablets depending on the patient’s characteristic. Experts recommendations. MS Report 2022;4:5–10. https://doi.org/10.24292/01.MS....
7.
Krey L, Raab P, Sherzay R, et al. Severe Progressive Multifocal Leukoencephalopathy (PML) and Spontaneous Immune Reconstitution Inflammatory Syndrome (IRIS) in an Immunocompetent Patient. Front Immunol. 2019;10:1188. doi:10.3389/fimmu.2019.01188.
8.
Patel A, Sul J, Gordon ML, et al. Progressive Multifocal Leukoencephalo-pathy in a Patient With Progressive Multiple Sclerosis Treated With Ocrelizumab Monotherapy. JAMA Neurol. 2021;78(6):736–740. doi:10.1001/jamaneurol.2021.0627.
9.
Gerevini S, Capra R, Bertoli D, et al. Immune profiling of a patient with alemtuzumab-associated progressive multifocal leukoencephalopathy. Mult Scler. 2019;25(8):1196–1201. doi:10.1177/1352458519832259.
10.
Coles AJ, Jones JL, Vermersch P, et al. Autoimmunity and long-term safety and efficacy of alemtuzumab for multiple sclerosis: Benefit/risk following review of trial and post-marketing data. Mult Scler. 2022;28(5):842–846. doi:10.1177/13524585211061335.
12.
N’gbo N’gbo Ikazabo R, Mostosi C, Quivron B, et al. Immune-reconstitution Inflammatory Syndrome in Multiple Sclerosis Patients Treated With Natalizumab: A Series of 4 Cases. Clin Ther. 2016;38(3):670–675. doi:10.1016/j.clinthera.2016.01.010.
13.
Cuascut FX, Alkabie S, Hutton GJ. Fingolimod-related cryptococcal meningoencephalitis and immune reconstitution inflammatory syndrome in a patient with multiple sclerosis. Mult Scler Relat Disord. 2021;53:103072. doi:10.1016/j.msard.2021.1030.
14.
Gundacker ND, Jordan SJ, Jones BA, et al. Acute Cryptococcal Immune Reconstitution Inflammatory Syndrome in a Patient on Natalizumab. Open Forum Infect Dis. 2016;3(1):ofw038. doi:10.1093/ofid/ofw038.
15.
Evangelopoulos ME, Koutoulidis V, Kilidireas K, et al. Immune reconstitution inflammatory syndrome mimicking progressive multifocal leukoencephalopathy in a multiple sclerosis patient treated with natalizumab: a case report and review of the literature. J Clin Med Res. 2015;7(1):65–68. doi:10.14740/jocmr1888w.
16.
Mickeviciene D, Baltusiene A, Afanasjeva B, et al. Progressive multifocal leukoencephalopathy or immune reconstitution inflammatory syndrome after fingolimod cessation? A case report. BMC Neurol. 2022;22(1):306. doi:10.1186/s12883-022-02839-3.
17.
Holmøy T, Beiske AG, Zarnovicky S, et al. Severe inflammatory disease activity 14 months after cessation of Natalizumab in a patient with Leber’s optic neuropathy and multiple sclerosis – a case report. BMC Neurol. 2016;16(1):197. doi:10.1186/s12883-016-0720-2.
18.
Sepúlveda M, Llufriu S, Blanco Y, et al. Intense immunosuppression for the treatment of an immune reconstitution inflammatory syndrome-like exacerbation after natalizumab withdrawal: a case report. J Neurol. 2015;262(1):219–221. doi:10.1007/s00415-014-7574-6.
19.
Massey TH, Smith R, Sadiq S, et al. Rescue of severe brain and cervical cord IRIS by restarting natalizumab in a pregnant MS patient. Neurology. 2017;88(7):711–713. doi:10.1212/WNL.0000000000003604.
20.
Oshima Y, Tanimoto T, Yuji K, et al. Drug-associated progressive multifocal leukoencephalopathy in multiple sclerosis patients. Mult Scler. 2019;25(8):1141–1149. doi:10.1177/1352458518786075.
21.
Stangel M, Becker V, Elias-Hamp B, et al. Oral pulsed therapy of relapsing multiple sclerosis with cladribine tablets – expert opinion on issues in clinical practice. Mult Scler Relat Disord. 2021;54:103075. doi: 10.1016/j.msard.2021.103075.
| eISSN: | 1898-2263 |
| ISSN: | 1232-1966 |
Improvement of visual identification of the journal - task financed under the agreement No. 618/P-DUN/2019 by the Minister of Science and Higher Education allocated to the activities of disseminating science.
Generation of the DOI (Digital Object Identifier) - task financed under the agreement No. 618/P-DUN/2019 by the Minister of Science and Higher
© 2006-2025 Journal hosting platform by Bentus
We process personal data collected when visiting the website. The function of obtaining information about users and their behavior is carried out by voluntarily entered information in forms and saving cookies in end devices. Data, including cookies, are used to provide services, improve the user experience and to analyze the traffic in accordance with the Privacy policy. Data are also collected and processed by Google Analytics tool (more).
You can change cookies settings in your browser. Restricted use of cookies in the browser configuration may affect some functionalities of the website.
You can change cookies settings in your browser. Restricted use of cookies in the browser configuration may affect some functionalities of the website.
