Levamisole-associated multifocal inflammatory encephalopathy: clinical and MRI characteristics, and diagnostic algorithm

•LAMIE is an important diagnostic consideration in the demyelinating episode in developing countries.•Only 1/5 of LAMIE patients with specific oligoclonal bands synthesis in CSF convert to multiple sclerosis.•Three MRI LAMIE patterns were described. Levamisole-associated multifocal inflammatory ence...

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Published in:	Multiple sclerosis and related disorders Vol. 69; p. 104418
Main Authors:	Fominykh, V., Averchenkov, D., Volik, A., Popova, E., Bryukhov, V., Nazarov, V., Moshnikova, A., Arzumanian, N., Tatarenko, A., Nechaev, V., Manuylova, O., Lapin, S., Brylev, L., Guekht, A.
Format:	Journal Article
Language:	English
Published:	Netherlands Elsevier B.V 01.01.2023
Subjects:	ADEM Brain - diagnostic imaging Brain - pathology Demyelinating disorders Encephalomyelitis, Acute Disseminated - pathology Female Humans Levamisole Levamisole - adverse effects Levamisole-associated multifocal inflammatory encephalopathy Magnetic Resonance Imaging Male Multiple sclerosis Multiple Sclerosis - diagnostic imaging Multiple Sclerosis - drug therapy Multiple Sclerosis - pathology Neuroinflammation Neurology Retrospective Studies Neuroinflammation Multiple sclerosis Demyelinating disorders Levamisole-associated multifocal inflammatory encephalopathy ADEM Levamisole demyelinating disorders levamisole neuroinflammation multiple sclerosis levamisole-associated multifocal inflammatory encephalopathy
ISSN:	2211-0348, 2211-0356, 2211-0356
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Abstract	•LAMIE is an important diagnostic consideration in the demyelinating episode in developing countries.•Only 1/5 of LAMIE patients with specific oligoclonal bands synthesis in CSF convert to multiple sclerosis.•Three MRI LAMIE patterns were described. Levamisole-associated multifocal inflammatory encephalopathy (LAMIE) is a devastating adverse effect of levamisole (LEV) treatment. In Russia, people often use LEV without a doctor's prescription for anthelmintic prophylaxis. LAMIE often misdiagnosed as the first episode of MS or acute disseminated encephalomyelitis (ADEM). The aim of our study was to describe clinical, laboratory and morphological characteristics of LAMIE, magnetic resonance imaging (MRI) patterns and create an algorithm for the differential diagnosis. This study was a prospective observational study with retrospective analysis of cases. It was performed at two hospitals with ambulatory service for MS. We included 43 patients with LAMIE with follow-up was from 1 year to 5 years. Age was 19-68 y.o. with female predominance. The most typical manifestations of LAMIE were cerebellar, pyramidal and cognitive symptoms, and majority of patients had biphasic course of the disease. Three main types of MRI patterns were described: ADEM-like, MS-like, atypical demyelination. About 40% of patients had CSF specific oligoclonal bands synthesis, but only 20 % of them converted to MS during the period from 1 month until 2 years. The CSF albumin levels and immunoglobulin G index were elevated in LAMIE patients compared to reference values. We described results of brain biopsy in two cases. Therefore LAMIE should be considered in patients with demyelinating or inflammatory conditions with biphasic onset of the disease and variable MRI presentation. [Display omitted]
AbstractList	Highlights•LAMIE is an important diagnostic consideration in the demyelinating episode in developing countries •Only 1/5 of LAMIE patients with specific oligoclonal bands synthesis in CSF convert to multiple sclerosis •Three MRI LAMIE patterns were described Levamisole-associated multifocal inflammatory encephalopathy (LAMIE) is a devastating adverse effect of levamisole (LEV) treatment. In Russia, people often use LEV without a doctor's prescription for anthelmintic prophylaxis. LAMIE often misdiagnosed as the first episode of MS or acute disseminated encephalomyelitis (ADEM). The aim of our study was to describe clinical, laboratory and morphological characteristics of LAMIE, magnetic resonance imaging (MRI) patterns and create an algorithm for the differential diagnosis. This study was a prospective observational study with retrospective analysis of cases. It was performed at two hospitals with ambulatory service for MS. We included 43 patients with LAMIE with follow-up was from 1 year to 5 years. Age was 19-68 y.o. with female predominance. The most typical manifestations of LAMIE were cerebellar, pyramidal and cognitive symptoms, and majority of patients had biphasic course of the disease. Three main types of MRI patterns were described: ADEM-like, MS-like, atypical demyelination. About 40% of patients had CSF specific oligoclonal bands synthesis, but only 20 % of them converted to MS during the period from 1 month until 2 years. The CSF albumin levels and immunoglobulin G index were elevated in LAMIE patients compared to reference values. We described results of brain biopsy in two cases. Therefore LAMIE should be considered in patients with demyelinating or inflammatory conditions with biphasic onset of the disease and variable MRI presentation.Levamisole-associated multifocal inflammatory encephalopathy (LAMIE) is a devastating adverse effect of levamisole (LEV) treatment. In Russia, people often use LEV without a doctor's prescription for anthelmintic prophylaxis. LAMIE often misdiagnosed as the first episode of MS or acute disseminated encephalomyelitis (ADEM). The aim of our study was to describe clinical, laboratory and morphological characteristics of LAMIE, magnetic resonance imaging (MRI) patterns and create an algorithm for the differential diagnosis. This study was a prospective observational study with retrospective analysis of cases. It was performed at two hospitals with ambulatory service for MS. We included 43 patients with LAMIE with follow-up was from 1 year to 5 years. Age was 19-68 y.o. with female predominance. The most typical manifestations of LAMIE were cerebellar, pyramidal and cognitive symptoms, and majority of patients had biphasic course of the disease. Three main types of MRI patterns were described: ADEM-like, MS-like, atypical demyelination. About 40% of patients had CSF specific oligoclonal bands synthesis, but only 20 % of them converted to MS during the period from 1 month until 2 years. The CSF albumin levels and immunoglobulin G index were elevated in LAMIE patients compared to reference values. We described results of brain biopsy in two cases. Therefore LAMIE should be considered in patients with demyelinating or inflammatory conditions with biphasic onset of the disease and variable MRI presentation. •LAMIE is an important diagnostic consideration in the demyelinating episode in developing countries.•Only 1/5 of LAMIE patients with specific oligoclonal bands synthesis in CSF convert to multiple sclerosis.•Three MRI LAMIE patterns were described. Levamisole-associated multifocal inflammatory encephalopathy (LAMIE) is a devastating adverse effect of levamisole (LEV) treatment. In Russia, people often use LEV without a doctor's prescription for anthelmintic prophylaxis. LAMIE often misdiagnosed as the first episode of MS or acute disseminated encephalomyelitis (ADEM). The aim of our study was to describe clinical, laboratory and morphological characteristics of LAMIE, magnetic resonance imaging (MRI) patterns and create an algorithm for the differential diagnosis. This study was a prospective observational study with retrospective analysis of cases. It was performed at two hospitals with ambulatory service for MS. We included 43 patients with LAMIE with follow-up was from 1 year to 5 years. Age was 19-68 y.o. with female predominance. The most typical manifestations of LAMIE were cerebellar, pyramidal and cognitive symptoms, and majority of patients had biphasic course of the disease. Three main types of MRI patterns were described: ADEM-like, MS-like, atypical demyelination. About 40% of patients had CSF specific oligoclonal bands synthesis, but only 20 % of them converted to MS during the period from 1 month until 2 years. The CSF albumin levels and immunoglobulin G index were elevated in LAMIE patients compared to reference values. We described results of brain biopsy in two cases. Therefore LAMIE should be considered in patients with demyelinating or inflammatory conditions with biphasic onset of the disease and variable MRI presentation. [Display omitted] Levamisole-associated multifocal inflammatory encephalopathy (LAMIE) is a devastating adverse effect of levamisole (LEV) treatment. In Russia, people often use LEV without a doctor's prescription for anthelmintic prophylaxis. LAMIE often misdiagnosed as the first episode of MS or acute disseminated encephalomyelitis (ADEM). The aim of our study was to describe clinical, laboratory and morphological characteristics of LAMIE, magnetic resonance imaging (MRI) patterns and create an algorithm for the differential diagnosis. This study was a prospective observational study with retrospective analysis of cases. It was performed at two hospitals with ambulatory service for MS. We included 43 patients with LAMIE with follow-up was from 1 year to 5 years. Age was 19-68 y.o. with female predominance. The most typical manifestations of LAMIE were cerebellar, pyramidal and cognitive symptoms, and majority of patients had biphasic course of the disease. Three main types of MRI patterns were described: ADEM-like, MS-like, atypical demyelination. About 40% of patients had CSF specific oligoclonal bands synthesis, but only 20 % of them converted to MS during the period from 1 month until 2 years. The CSF albumin levels and immunoglobulin G index were elevated in LAMIE patients compared to reference values. We described results of brain biopsy in two cases. Therefore LAMIE should be considered in patients with demyelinating or inflammatory conditions with biphasic onset of the disease and variable MRI presentation.
ArticleNumber	104418
Author	Nechaev, V. Tatarenko, A. Lapin, S. Guekht, A. Brylev, L. Fominykh, V. Volik, A. Moshnikova, A. Popova, E. Bryukhov, V. Averchenkov, D. Nazarov, V. Arzumanian, N. Manuylova, O.
Author_xml	– sequence: 1 givenname: V. orcidid: 0000-0001-5490-2390 surname: Fominykh fullname: Fominykh, V. email: hydrohinon@mail.ru organization: Institute of Higher Nervous Activity and Neurophysiology, Butlerova street 5a, Moscow 117485, Russia – sequence: 2 givenname: D. surname: Averchenkov fullname: Averchenkov, D. organization: Bujanov Moscow City Clinical Hospital, Moscow, Russia – sequence: 3 givenname: A. surname: Volik fullname: Volik, A. organization: Institute of Higher Nervous Activity and Neurophysiology, Butlerova street 5a, Moscow 117485, Russia – sequence: 4 givenname: E. surname: Popova fullname: Popova, E. organization: City Clinical Hospital № 24, Moscow, Russia – sequence: 5 givenname: V. surname: Bryukhov fullname: Bryukhov, V. organization: Research Center of Neurology, Moscow, Russia – sequence: 6 givenname: V. surname: Nazarov fullname: Nazarov, V. organization: Research Center of Neurology, Moscow, Russia – sequence: 7 givenname: A. surname: Moshnikova fullname: Moshnikova, A. organization: Pavlov First Saint Petersburg Medical University, Saint Petersburg, Russia – sequence: 8 givenname: N. surname: Arzumanian fullname: Arzumanian, N. organization: Bujanov Moscow City Clinical Hospital, Moscow, Russia – sequence: 9 givenname: A. surname: Tatarenko fullname: Tatarenko, A. organization: Bujanov Moscow City Clinical Hospital, Moscow, Russia – sequence: 10 givenname: V. surname: Nechaev fullname: Nechaev, V. organization: Bujanov Moscow City Clinical Hospital, Moscow, Russia – sequence: 11 givenname: O. surname: Manuylova fullname: Manuylova, O. organization: Bujanov Moscow City Clinical Hospital, Moscow, Russia – sequence: 12 givenname: S. surname: Lapin fullname: Lapin, S. organization: Pavlov First Saint Petersburg Medical University, Saint Petersburg, Russia – sequence: 13 givenname: L. surname: Brylev fullname: Brylev, L. organization: Institute of Higher Nervous Activity and Neurophysiology, Butlerova street 5a, Moscow 117485, Russia – sequence: 14 givenname: A. surname: Guekht fullname: Guekht, A. organization: Moscow Research and Clinical Center for Neuropsychiatry, Moscow Healthcare Department, Russia
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Keywords	Neuroinflammation Multiple sclerosis Demyelinating disorders Levamisole-associated multifocal inflammatory encephalopathy ADEM Levamisole demyelinating disorders levamisole neuroinflammation multiple sclerosis levamisole-associated multifocal inflammatory encephalopathy
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Snippet	•LAMIE is an important diagnostic consideration in the demyelinating episode in developing countries.•Only 1/5 of LAMIE patients with specific oligoclonal... Highlights•LAMIE is an important diagnostic consideration in the demyelinating episode in developing countries •Only 1/5 of LAMIE patients with specific... Levamisole-associated multifocal inflammatory encephalopathy (LAMIE) is a devastating adverse effect of levamisole (LEV) treatment. In Russia, people often use...
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SubjectTerms	ADEM Brain - diagnostic imaging Brain - pathology Demyelinating disorders Encephalomyelitis, Acute Disseminated - pathology Female Humans Levamisole Levamisole - adverse effects Levamisole-associated multifocal inflammatory encephalopathy Magnetic Resonance Imaging Male Multiple sclerosis Multiple Sclerosis - diagnostic imaging Multiple Sclerosis - drug therapy Multiple Sclerosis - pathology Neuroinflammation Neurology Retrospective Studies
Title	Levamisole-associated multifocal inflammatory encephalopathy: clinical and MRI characteristics, and diagnostic algorithm
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