Top

Neurological Sciences

Published in:

Open Access 18-09-2023 | Multiple Sclerosis | Review Article

Glucocorticoid-dependent multiple sclerosis overlapping anti-NMDA receptor encephalitis: a case report and literature review update

Authors: Bo Yang, Nengwei Yu

Published in: Neurological Sciences | Issue 1/2024

Abstract

Background

Previous studies suggest a relationship between central nervous system inflammatory demyelinating diseases and anti-N-methyl-d-aspartate receptor (NMDAR) encephalitis. Also, the overlap between anti-NMDAR encephalitis and multiple sclerosis (MS) has been reported. However, the pathogenesis and clinical characteristics are still obscure.

Case presentation

A 33-year-old woman presented with diplopia and sensory ataxia at the onset. The cerebrospinal fluid (CSF) anti-NMDAR antibodies were positive (1:3.2), and nuclear magnetic resonance imaging (MRI) showed bilateral centrum ovale and lateral ventricle demyelinating lesions. Therefore, she was diagnosed with anti-NMDAR encephalitis. After administering intravenous immunoglobulin and oral prednisone, her lesions disappeared, and symptoms were relieved. The condition was maintained with a low dose of prednisone, but her lesions reappeared on MRI. Consequently, immunomodulatory therapy of mycophenolate mofetil was initiated. However, she developed dysarthria and right limb ataxia after 10 months with a positive CSF anti-NMDAR antibody (1:1) and positive oligoclonal band. The MRI showed symmetrical multiple demyelinating lesions. Considering the MS diagnosis, her neurological dysfunction again improved significantly after intravenous methylprednisolone. Unfortunately, her symptoms aggravated for the second time when teriflunomide was started. Finally, her condition was controlled again with oral prednisone.

Conclusions

Consistent with previous cases of overlapping anti-NMDAR encephalitis and MS, patients often show atypical symptoms on MRIs and immunological tests. The overlap cannot be arbitrarily treated because of the recurrence of previous diseases. Long-term follow-up, dynamic antibody monitoring, and MRI examination are crucial for these patients. The special dependency of the patient on glucocorticoids in this study has been rarely reported, which may guide the treatment of insensitivity to disease-modifying therapy in recurrent overlapping anti-NMDAR encephalitis and MS.

Dobson R, Giovannoni G (2019) Multiple sclerosis - a review. Eur J Neurol 26(1):27–40.https://doi.org/10.1111/ene.13819CrossRefPubMed

Oh J, Vidal-Jordana A, Montalban X (2018) Multiple sclerosis: clinical aspects. Curr Opin Neurol 31(6):752–759.https://doi.org/10.1097/WCO.0000000000000622CrossRefPubMed

Guasp M, Dalmau J (2018) Encephalitis associated with antibodies against the NMDA receptor. Encefalitis por anticuerpos contra el receptor de NMDA. Med Clin (Barc) 151(2):71–79.https://doi.org/10.1016/j.medcli.2017.10.015CrossRefPubMed

Ramberger M, Bsteh G, Schanda K, Höftberger R, Rostásy K, Baumann M et al (2015) NMDA receptor antibodies: a rare association in inflammatory demyelinating diseases. Neurol Neuroimmunol Neuroinflamm 2(5):e141.https://doi.org/10.1212/NXI.0000000000000141CrossRefPubMedPubMedCentral

Dalmau J, Lancaster E, Martinez-Hernandez E, Rosenfeld MR, Balice-Gordon R (2011) Clinical experience and laboratory investigations in patients with anti-NMDAR encephalitis. Lancet Neurol 10(1):63–74.https://doi.org/10.1016/S1474-4422(10)70253-2CrossRefPubMedPubMedCentral

Dalmau J, Tüzün E, Wu HY, Masjuan J, Rossi JE, Voloschin A et al (2007) Paraneoplastic anti-N-methyl-D-aspartate receptor encephalitis associated with ovarian teratoma. Ann Neurol 61(1):25–36.https://doi.org/10.1002/ana.21050CrossRefPubMedPubMedCentral

Stangel M, Fredrikson S, Meinl E, Petzold A, Stüve O, Tumani H (2013) The utility of cerebrospinal fluid analysis in patients with multiple sclerosis. Nat Rev Neurol 9(5):267–276.https://doi.org/10.1038/nrneurol.2013.41CrossRefPubMed

Ramachandran PS, Wilson MR (2020) Metagenomics for neurological infections-expanding our imagination. Nat Rev Neurol 16(10):547–556.https://doi.org/10.1038/s41582-020-0374-yCrossRefPubMedPubMedCentral

Brusaferri F, Candelise L (2000) Steroids for multiple sclerosis and optic neuritis: a meta-analysis of randomized controlled clinical trials. J Neurol 247(6):435–442.https://doi.org/10.1007/s004150070172CrossRefPubMed

10.

Vedula SS, Brodney-Folse S, Gal RL, Beck R (2007) Corticosteroids for treating optic neuritis. Cochrane Database Syst Rev (1):CD001430. Published 2007 Jan 24.https://doi.org/10.1002/14651858.CD001430.pub2

11.

Thrower BW (2009) Relapse management in multiple sclerosis. Neurologist 15(1):1–5.https://doi.org/10.1097/NRL.0b013e31817acf1aCrossRefPubMed

12.

Zhao YY, Wang HX, Wang M, Yuan B, Sun TW, Xu YM (2022) Relapse factors of patients of anti-N-methyl-D-aspartate receptor encephalitis. Acta Neurol Scand 145(4):434–441.https://doi.org/10.1111/ane.13568CrossRefPubMed

13.

Zhong R, Chen Q, Zhang X, Zhang H, Lin W (2022) Relapses of Anti-NMDAR, anti-GABABR and anti-LGI1 encephalitis: a retrospective cohort study. Front Immunol 13:918396. Published 2022 Jun 9.https://doi.org/10.3389/fimmu.2022.918396CrossRefPubMedPubMedCentral

14.

Oliveri RL, Valentino P, Russo C, Sibilia G, Aguglia U, Bono F et al (1998) Randomized trial comparing two different high doses of methylprednisolone in MS: a clinical and MRI study. Neurology 50(6):1833–1836.https://doi.org/10.1212/wnl.50.6.1833CrossRefPubMed

15.

Baldassari LE, Fox RJ (2018) Therapeutic advances and challenges in the treatment of progressive multiple sclerosis. Drugs 78(15):1549–1566.https://doi.org/10.1007/s40265-018-0984-5CrossRefPubMed

16.

Held U, Heigenhauser L, Shang C, Kappos L, Polman C, Sylvia Lawry Centre for MS Research (2005) Predictors of relapse rate in MS clinical trials. Neurology 65(11):1769–1773.https://doi.org/10.1212/01.wnl.0000187122.71735.1fCrossRefPubMed

17.

Jokubaitis VG, Li V, Kalincik T, Izquierdo G, Hodgkinson S, Alroughani R et al (2014) Fingolimod after natalizumab and the risk of short-term relapse. Neurology 82(14):1204–1211.https://doi.org/10.1212/WNL.0000000000000283CrossRefPubMedPubMedCentral

18.

Rovaris M, Comi G, Ladkani D, Wolinsky JS, Filippi M, European/Canadian Glatiramer Acetate Study Group (2003) Short-term correlations between clinical and MR imaging findings in relapsing-remitting multiple sclerosis. AJNR Am J Neuroradiol 24(1):75–81PubMedPubMedCentral

19.

Sormani MP, Rovaris M, Comi G, Filippi M (2007) A composite score to predict short-term disease activity in patients with relapsing-remitting MS. Neurology 69(12):1230–1235.https://doi.org/10.1212/01.wnl.0000276940.90309.15CrossRefPubMed

20.

Titulaer MJ, Höftberger R, Iizuka T, Leypoldt F, McCracken L, Cellucci T et al (2014) Overlapping demyelinating syndromes and anti–N-methyl-D-aspartate receptor encephalitis. Ann Neurol 75(3):411–428.https://doi.org/10.1002/ana.24117CrossRefPubMedPubMedCentral

21.

Hacohen Y, Deiva K, Pettingill P, Waters P, Siddiqui A, Chretien P et al (2014) N-methyl-D-aspartate receptor antibodies in post-herpes simplex virus encephalitis neurological relapse. Mov Disord 29(1):90–96.https://doi.org/10.1002/mds.25626CrossRefPubMed

22.

Lipton SA (2006) NMDA receptors, glial cells, and clinical medicine. Neuron 50(1):9–11.https://doi.org/10.1016/j.neuron.2006.03.026CrossRefPubMed

23.

Leypoldt F, Titulaer MJ, Aguilar E, Walther J, Bönstrup M, Havemeister S et al (2013) Herpes simplex virus-1 encephalitis can trigger anti-NMDA receptor encephalitis: case report. Neurology 81(18):1637–1639.https://doi.org/10.1212/WNL.0b013e3182a9f531CrossRefPubMedPubMedCentral

24.

Johnston J, Ali K, Pearson OR, Rickards C, Vincent A (2010) POI14 Multiple sclerosis: a potential association with anti-N-methyl-d-aspartate receptor encephalitis. J Neurol Neurosurg Psychiatry 81(11):e56.https://doi.org/10.1136/jnnp.2010.226340.155

25.

Uzawa A, Mori M, Takahashi Y, Ogawa Y, Uchiyama T, Kuwabara S (2012) Anti-N-methyl D-aspartate-type glutamate receptor antibody-positive limbic encephalitis in a patient with multiple sclerosis. Clin Neurol Neurosurg 114(4):402–404.https://doi.org/10.1016/j.clineuro.2011.10.047CrossRefPubMed

26.

Waschbisch A, Kallmünzer B, Schwab S, Gölitz P, Vincent A, Lee DH et al (2014) Demyelinating disease and anti-N-methyl-D-aspartate receptor immunoglobulin G antibodies: a case report. BMC Res Notes 7:948. Published 2014 Dec 23.https://doi.org/10.1186/1756-0500-7-948CrossRefPubMedPubMedCentral

27.

Fleischmann R, Prüss H, Rosche B, Bahnemann M, Gelderblom H, Deuschle K et al (2015) Severe cognitive impairment associated with intrathecal antibodies to the NR1 subunit of the N-methyl-D-aspartate receptor in a patient with multiple sclerosis. JAMA Neurol 72(1):96–99.https://doi.org/10.1001/jamaneurol.2014.1817CrossRefPubMed

28.

Huang G, Zou XQ, Wang YM, Wu XM (2016) A case of recurrent anti-NMDA receptor encephalitis misdiagnosed as multiple sclerosis. Chin J Neuroimmunol Neurol 23(05):379–380

29.

Baheerathan A, Brownlee WJ, Chard DT, Shields K, Gregory R, Trip SA (2017) Antecedent anti-NMDA receptor encephalitis in two patients with multiple sclerosis. Mult Scler Relat Disord 12:20–22.https://doi.org/10.1016/j.msard.2016.12.009CrossRefPubMed

30.

Suleman S, Javed Q (2018) NMDAR (N-methyl-D-aspartate receptor) encephalitis in a patient with MS (multiple sclerosis): a rare and challenging case. BMJ Case Rep 2018:bcr2017222086. Published 2018 Feb 17.https://doi.org/10.1136/bcr-2017-222086CrossRefPubMedPubMedCentral

31.

Gulec B, Kurucu H, Bozbay S, Dikmen Y, Sayman H, Tuzun E et al (2020) Co-existence of multiple sclerosis and anti-NMDA receptor encephalitis: a case report and review of literature. Mult Scler Relat Disord 42:102075.https://doi.org/10.1016/j.msard.2020.102075CrossRefPubMed

32.

Huang Y, Wang Q, Zeng S, Zhang Y, Zou L, Fu X et al (2020) Case report: overlapping multiple sclerosis with anti-N-methyl-D-aspartate receptor encephalitis: a case report and review of literature. Front Immunol 11:595417. Published 2020 Dec 9.https://doi.org/10.3389/fimmu.2020.595417CrossRefPubMedPubMedCentral

33.

Tappatà M, Riguzzi P, Volpi L, Albini Riccioli L, Pantieri R, Michelucci R (2021) Long-term persistence of NMDAR antibodies after encephalitis with de novo occurrence of demyelinating disorder. Neurol Sci 42(1):301–303.https://doi.org/10.1007/s10072-020-04729-3CrossRefPubMed

34.

Chahal K, Endeman T, Scapinello S, Sapieha M (2021) Anti-N-methyl-D-aspartate receptor encephalitis presenting as atypical psychosis in multiple sclerosis: a case report. BMC Psychiatry 21(1):347. Published 2021 Jul 12.https://doi.org/10.1186/s12888-021-03351-7CrossRefPubMedPubMedCentral

35.

Seyed Ahadi M, Ghadiri F, Sahraian MA, Naser MA (2021) Anti-N-methyl-d-aspartate receptor encephalitis in a patient with multiple sclerosis on dimethyl fumarate: a case report. Neurol Sci 42(9):3929–3931.https://doi.org/10.1007/s10072-021-05385-xCrossRefPubMed

36.

Zhou R, Jiang F, Cai H, Zeng Q, Yang H (2021) Case Report: Antibodies to the N-methyl-D-aspartate receptor in a patient with multiple sclerosis. Front Immunol 12:664364. Published 2021 Apr 23.https://doi.org/10.3389/fimmu.2021.664364CrossRefPubMedPubMedCentral

37.

Karunaratne K, Ahrabian D, Monoghan B, Campion T, Yousry T, Lunn MP et al (2021) Bortezomib for anti-NMDAR encephalitis following daclizumab treatment in a patient with multiple sclerosis. BMJ Neurol Open 3(1):e000096.https://doi.org/10.1136/bmjno-2020-00009CrossRefPubMedPubMedCentral

38.

Etemadifar M, Nouri H, Salari M, Sedaghat N (2022) Detection of anti-NMDA receptor antibodies following BBIBP-CorV COVID-19 vaccination in a rituximab-treated person with multiple sclerosis presenting with manifestations of an acute relapse. Hum Vaccin Immunother 18(1):2033540.https://doi.org/10.1080/21645515.2022.2033540CrossRefPubMedPubMedCentral

39.

Liu P, Yan H, Li H, Zhang C, Li Y (2023) Overlapping anti-NMDAR encephalitis and multiple sclerosis: a case report and literature review. Front Immunol 14:1088801. Published 2023 Jan 30.https://doi.org/10.3389/fimmu.2023.1088801CrossRefPubMedPubMedCentral

Title: Glucocorticoid-dependent multiple sclerosis overlapping anti-NMDA receptor encephalitis: a case report and literature review update
Authors: Bo Yang
Nengwei Yu
Publication date: 18-09-2023
Publisher: Springer International Publishing
Keywords: Multiple Sclerosis
Anti-NMDA Receptor Encephalitis
Glucocorticoid
Magnetic Resonance Imaging
Magnetic Resonance Imaging
Diplopia
Teriflunomide
Demyelinating Disease
Published in: Neurological Sciences / Issue 1/2024
Print ISSN: 1590-1874
Electronic ISSN: 1590-3478
DOI: https://doi.org/10.1007/s10072-023-07034-x

Keynote webinar | Spotlight on medication adherence

Springer Medicine

Glucocorticoid-dependent multiple sclerosis overlapping anti-NMDA receptor encephalitis: a case report and literature review update

Abstract

Background

Case presentation

Conclusions

Keynote webinar | Spotlight on medication adherence

Springer Medicine

Abstract

Background

Case presentation

Conclusions

Please log in to get access to this content

Other articles of this Issue 1/2024

Arg4810Lys mutation in RNF213 among Eastern Indian non-MMD ischemic stroke patients: a genotype–phenotype correlation

Repeated intravenous thrombolysis in early recurrent ischemic stroke: a case report of an elderly low weight female

A review and meta-analysis of stem cell therapies in stroke patients: effectiveness and safety evaluation

Lithium-induced subacute diencephalic angio endotheliopathy

Dizziness and vertigo in patients with tension-type headache: A case–control study

Epworth sleepiness scale is associated with increased striatal dopamine uptake in Parkinson’s disease: a cross-sectional study