Top

BMC Neurology

Published in:

Open Access 01-12-2022 | Levodopa | Case report

Neuroleptic malignant-like syndrome associated multiple system atrophy: report on three cases

Authors: Yan Lin, Lin Ma, Nan Zhang, Ruihua Li, Wenjing Jiang

Published in: BMC Neurology | Issue 1/2022

Abstract

Background

Multiple system atrophy (MSA) associated with neuroleptic malignant-like syndrome (NMLS) is rare and few cases have been described in the literature.

Case presentation

In the present study, three patients with MSA associated with NMLS were analyzed from January 2012 to January 2020 to characterize their clinical presentations. Data collected from the patients for analysis included general patient history, the fluctuation and severity of disease symptoms, the indicated therapies and disease progression at follow-up. All patients had histories of sudden withdrawal or reduction of levodopa prior to the onset of symptoms. Clinical presentations were characterized by hyperthermia, autonomic dysfunction, worsening of extrapyramidal symptoms, and elevated serum creatine kinase (CK) levels. During hospitalization, one patient rapidly progressed and died, while the other two patients were successfully treated.

Conclusions

Early diagnosis and treatment are very important for patient outcomes in NMLS. Notably, the correct dose and time of administration of dopaminergic medication may be key in treating NMLS.

Available only for authorised users

Delay J, Deniker P. Drug-induced extrapyramidal syndromes. In: Vinken PJ, Bruyn GW, editors. Handbook of Clinical Neurology; Diseases of the Basal Ganglia, vol. 6; 1968. p. 248–66.

Oruch R, Pryme IF, Engelsen BA, Lund A. Neuroleptic malignant syndrome: an easily overlooked neurologic emergency. Neuropsychiatr Dis Treat. 2017;13:161–75. https://doi.org/10.2147/ndt.S118438.CrossRefPubMedPubMedCentral

Gillman PK. Neuroleptic malignant syndrome: mechanisms, interactions, and causality. Mov Disord. 2010;25(12):1780–90. https://doi.org/10.1002/mds.23220.CrossRefPubMed

Toru M, Matsuda O, Makiguchi K, Sugano K. Neuroleptic malignant syndrome-like state following a withdrawal of antiparkinsonian drugs. J Nerv Ment Dis. 1981;169(5):324–7. https://doi.org/10.1097/00005053-198105000-00011.CrossRefPubMed

Modi S, Dharaiya D, Schultz L, Varelas P. Neuroleptic malignant syndrome: complications, outcomes, and mortality. Neurocrit Care. 2016;24(1):97–103. https://doi.org/10.1007/s12028-015-0162-5.CrossRefPubMed

Velamoor R. Neuroleptic malignant syndrome: a neuro-psychiatric emergency: recognition, prevention, and management. J Asian J Psychiatry. 2017;29:106–9. https://doi.org/10.1016/j.ajp.2017.05.004.CrossRef

Ueda M, Hamamoto M, Nagayama H, Okubo S, Amemiya S, Katayama Y. Biochemical alterations during medication withdrawal in Parkinson's disease with and without neuroleptic malignant-like syndrome. J Neurol Neurosurg Psychiatry. 2001;71(1):111–3. https://doi.org/10.1136/jnnp.71.1.111.CrossRefPubMedPubMedCentral

Jussen D, Sprung C, Buchert R, Braun JS. Hydrocephalus-induced neuroleptic malignant-like syndrome with reduced dopamine transporters. J Neurol. 2013;260(8):2182–4. https://doi.org/10.1007/s00415-013-7026-8.CrossRefPubMed

Eiden C, Capdevielle D, Deddouche C, Boulenger JP, Blayac JP, Peyrière H. Neuroleptic malignant syndrome-like reaction precipitated by antipsychotics in a patient with gamma-butyrolactone withdrawal. J Addict Med. 2011;5(4):302–3. https://doi.org/10.1097/ADM.0b013e3182236730.CrossRefPubMed

10.

Bonnici A, Ruiner CE, St-Laurent L, Hornstein D. An interaction between levodopa and enteral nutrition resulting in neuroleptic malignant-like syndrome and prolonged ICU stay. Ann Pharmacother. 2010;44(9):1504–7. https://doi.org/10.1345/aph.1P242.CrossRefPubMed

11.

Caroff SN, Campbell EC. Risk of neuroleptic malignant syndrome in patients with NMDAR encephalitis. Neurol Sci. 2015;36(3):479–80. https://doi.org/10.1007/s10072-014-2022-z.CrossRefPubMed

12.

Koga S, Dickson DW. Recent advances in neuropathology, biomarkers and therapeutic approach of multiple system atrophy. J Neurol Neurosurg Psychiatry. 2018;89(2):175–84. https://doi.org/10.1136/jnnp-2017-315813.CrossRefPubMed

13.

Gilman S, Wenning GK, Low PA, Brooks DJ, Mathias CJ, Trojanowski JQ, et al. Second consensus statement on the diagnosis of multiple system atrophy. 2008;71(9):670–6. https://doi.org/10.1212/01.wnl.0000324625.00404.15.

14.

Lieto M, Roca A, Bruzzese D, Antenora A, Alfieri G, Saccà F, et al. Longitudinal study of a cohort of MSA-C patients in South Italy: survival and clinical features. Neurol Sci. 2019;40(10):2105–9. https://doi.org/10.1007/s10072-019-03948-7.CrossRefPubMed

15.

Gurrera RJ, Mortillaro G, Velamoor V, Caroff SN. A validation study of the international consensus diagnostic criteria for neuroleptic malignant syndrome. J Clin Psychopharmacol. 2017;37(1):67–71. https://doi.org/10.1097/jcp.0000000000000640.CrossRefPubMed

16.

Ghosh R, Liddle BJ. Emergency presentations of Parkinson's disease: early recognition and treatment are crucial for optimum outcome. Postgrad Med J. 2011;87(1024):125–31. https://doi.org/10.1136/pgmj.2010.104976.CrossRefPubMed

17.

Wei L, Chen Y. Neuroleptic malignant-like syndrome with a slight elevation of creatine-kinase levels and respiratory failure in a patient with Parkinson's disease. Patient Preference Adherence. 2014;8:271–3. https://doi.org/10.2147/ppa.S59150.CrossRefPubMedPubMedCentral

18.

Serrano-Dueñas M. Neuroleptic malignant syndrome-like, or--dopaminergic malignant syndrome--due to levodopa therapy withdrawal. Clinical features in 11 patients. Parkinsonism Relat Disord. 2003;9(3):175–8. https://doi.org/10.1016/s1353-8020(02)00035-4.CrossRefPubMed

19.

Reimer J, Kuhlmann A, Müller T. Neuroleptic malignant-like syndrome after rapid switch from bromocriptine to pergolide. Parkinsonism Relat Disord. 2002;9(2):115–6. https://doi.org/10.1016/s1353-8020(01)00045-1.CrossRefPubMed

20.

Jellinger KA. Multiple system atrophy: an Oligodendroglioneural Synucleinopathy1. J Alzheimers Dis. 2018;62(3):1141–79. https://doi.org/10.3233/jad-170397.CrossRefPubMedPubMedCentral

21.

Glasmacher SA, Leigh PN, Saha RA. Predictors of survival in progressive supranuclear palsy and multiple system atrophy: a systematic review and meta-analysis. J Neurol Neurosurg Psychiatry. 2017;88(5):402–11. https://doi.org/10.1136/jnnp-2016-314956.CrossRefPubMed

22.

Curry-Hyde A, Chen BJ, Ueberham U, Arendt T, Janitz M. Multiple system atrophy: many lessons from the Transcriptome. Neuroscientist. 2018;24(3):294–307. https://doi.org/10.1177/1073858417723915.CrossRefPubMed

23.

Guerrero-Ferreira R, Kovacik L, Ni D, Stahlberg H. New insights on the structure of alpha-synuclein fibrils using cryo-electron microscopy. Curr Opin Neurobiol. 2020;61:89–95. https://doi.org/10.1016/j.conb.2020.01.014.CrossRefPubMed

24.

Douglas A, Morris J. It was not just a heatwave! Neuroleptic malignant-like syndrome in a patient with Parkinson's disease. Age Ageing. 2006;35(6):640–1. https://doi.org/10.1093/ageing/afl096.CrossRefPubMed

25.

Hashimoto T, Tokuda T, Hanyu N, Tabata K, Yanagisawa N. Withdrawal of levodopa and other risk factors for malignant syndrome in Parkinson's disease. J Parkinsonism Relat Disord. 2003:S25–30. https://doi.org/10.1016/s1353-8020(02)00119-0.

26.

Sechi G, Manca S, Deiana GA, Corda DG, Pisu A, Rosati G. Acute hyponatremia and neuroleptic malignant syndrome in Parkinson's disease. Prog Neuropsychopharmacol Biol Psychiatry. 1996;20(3):533–42. https://doi.org/10.1016/0278-5846(96)00015-2.CrossRefPubMed

27.

Sone H, Okuda Y, Bannai C, Suzuki S, Yamaoka T, Asakura Y, et al. Syndrome of inappropriate secretion of antidiuretic hormone (SIADH) and Gerhardt syndrome associated with shy-Drager syndrome. Intern Med. 1994;33(12):773–8. https://doi.org/10.2169/internalmedicine.33.773.CrossRefPubMed

28.

Samukawa M, Hirano M, Sakamoto H, Kitada M, Kusunoki S, Nakamura Y. Risks of inappropriate secretion of antidiuretic hormone in multiple system atrophy. Mov Disord. 2011;26(14):2572–3. https://doi.org/10.1002/mds.23904.CrossRefPubMed

29.

Nisijima K, Shioda K. A rare case of neuroleptic malignant syndrome without elevated serum creatine kinase. Neuropsychiatr Dis Treat. 2014;10:403–7. https://doi.org/10.2147/ndt.S58677.CrossRefPubMedPubMedCentral

30.

Nisijima K. Elevated creatine kinase does not necessarily correspond temporally with onset of muscle rigidity in neuroleptic malignant syndrome: a report of two cases. Neuropsychiatr Dis Treat. 2012;8:615–8. https://doi.org/10.2147/ndt.S38638.CrossRefPubMedPubMedCentral

31.

Rehman T, deBoisblanc BP. Persistent fever in the ICU. Chest. 2014;145(1):158–65. https://doi.org/10.1378/chest.12-2843.CrossRefPubMed

32.

Onofrj M, Bonanni L, Cossu G, Manca D, Stocchi F, Thomas A. Emergencies in parkinsonism: akinetic crisis, life-threatening dyskinesias, and polyneuropathy during L-Dopa gel treatment. Parkinsonism Relat Disord. 2009:S233–6. https://doi.org/10.1016/s1353-8020(09)70821-1.

33.

Wang HY, Li T, Li XL, Zhang XX, Yan ZR, Xu Y. Anti-N-methyl-D-aspartate receptor encephalitis mimics neuroleptic malignant syndrome: case report and literature review. Neuropsychiatr Dis Treat. 2019;15:773–8. https://doi.org/10.2147/ndt.S195706.CrossRefPubMedPubMedCentral

34.

Fiore S, Persichino L, Anticoli S, De Pandis MF. A neuroleptic malignant-like syndrome (NMLS) in a patient with Parkinson's disease resolved with rotigotine: a case report. Acta biomed Atenei Parmensis. 2014;85(3):281–4.

35.

Man SP. An uncommon adverse effect of levodopa withdrawal in a patient taking antipsychotic medication: neuroleptic malignant-like syndrome. Hong Kong Med J. 2011;17(1):74–6.PubMed

36.

Auffret M, Drapier S, Vérin M. New tricks for an old dog: a repurposing approach of apomorphine. Eur J Pharmacol. 2019;843:66–79. https://doi.org/10.1016/j.ejphar.2018.10.052.CrossRefPubMed

37.

Kishimoto S, Nakamura K, Arai T, Yukimasa O, Fukami N. Postoperative neuroleptic malignant syndrome-like symptoms improved with intravenous diazepam: a case report. J Anesth. 2013;27(5):768–70. https://doi.org/10.1007/s00540-013-1602-z.CrossRefPubMedPubMedCentral

38.

Azuma H, Negi A, Hattori M, Masayasu T, Akechi T, Furukawa TA. Neuroleptic malignant syndrome-like state in an epileptic patient with organic brain comorbidity treated with zonisamide and carbamazepine. Epilepsia. 2007;48(10):1999–2001. https://doi.org/10.1111/j.1528-1167.2007.01165_2.x.CrossRefPubMed

39.

Shin HW, Youn YC. Neuroleptic malignant syndrome induced by phenytoin in a patient with drug-induced parkinsonism. Neurol Sci. 2014;35(10):1641–3. https://doi.org/10.1007/s10072-014-1826-1.CrossRefPubMed

Title: Neuroleptic malignant-like syndrome associated multiple system atrophy: report on three cases
Authors: Yan Lin
Lin Ma
Nan Zhang
Ruihua Li
Wenjing Jiang
Publication date: 01-12-2022
Publisher: BioMed Central
Keyword: Levodopa
Published in: BMC Neurology / Issue 1/2022
Electronic ISSN: 1471-2377
DOI: https://doi.org/10.1186/s12883-022-02583-8

At a glance: The ONWARDS insulin icodec trials

Springer Medicine

Neuroleptic malignant-like syndrome associated multiple system atrophy: report on three cases

Abstract

Background

Case presentation

Conclusions

At a glance: The ONWARDS insulin icodec trials

Springer Medicine

Abstract

Background

Case presentation

Conclusions

Please log in to get access to this content

Other articles of this Issue 1/2022

Early predictors of new-onset immune-related seizures: a preliminary study

Collateral status, hyperglycemia, and functional outcome after acute ischemic stroke

Barriers to quality of life in patients with multiple sclerosis: a qualitative study

Informing the development of an outcome set and banks of items to measure mobility among individuals with acquired brain injury using natural language processing

Evaluating the impact of adjunctive istradefylline on the cumulative dose of levodopa-containing medications in Parkinson’s disease: study protocol for the ISTRA ADJUST PD randomized, controlled study

Is diabetic striatopathy the culprit of seizures in a patient with ketotic hyperglycemia-induced hemichorea–hemiballismus?