Artigo Acesso aberto Produção Nacional

Central nervous system demyelination following isolated levamisole use: Case report and systematic review

2022; Elsevier BV; Volume: 2; Linguagem: Inglês

10.1016/j.nerep.2022.100058

ISSN

2667-257X

Autores

Luan CÔRTES, Silas SANTANA, Thiago Gonçalves Fukuda, Aroldo Bacellar,

Tópico(s)

Cerebrospinal fluid and hydrocephalus

Resumo

In most reported cases of central nervous system (CNS) demyelination following levamisole use, patients were exposed to at least one more known neurotoxic drug (contaminated cocaine or colon cancer combination therapy with fluorouracil). No reviews of CNS demyelination following isolated levamisole use have been published to date. This study aimed to assess clinical, diagnostic, and therapeutic features of CNS demyelination following isolated levamisole use and report an original case. A sensitive search strategy was used to search MEDLINE, EMBASE, and LILACS (inception to February 2021) databases for English, Spanish, and Portuguese articles. Publications reporting on cases of CNS demyelination following isolated levamisole use in patients without previously diagnosed demyelinating diseases were included. A standardized approach was employed to extract predefined data, including demographics, details on levamisole exposure, clinical features, diagnostic workup, treatment, and outcomes. Descriptive statistics was used to summarize data. Eleven articles reporting on 61 patients met inclusion criteria and were reviewed together with the present report for a total of 62 cases. Patient mean age was 45.1 ± 13.3 years, and two thirds of them were women. Patients were exposed to variable total doses of levamisole (50 to 13,500 mg) mainly for the treatment of recurrent aphthous ulcers and intestinal parasitic infections. One to 144 days after exposure, patients developed a subacute, progressive neurological syndrome characterized by encephalopathy, including cognitive impairment in 22 (40.7%) and an altered level of consciousness in 9 of them (16.7%), accompanied by focal neurological deficits, chiefly muscle weakness in 25 (46.3%), language disorder in 23 (42.6%), and ataxia in 18 cases (33.3%). In all patients, brain magnetic resonance imaging (MRI) showed multiple, hyperintense lesions on T2-weighted and fluid-attenuated inversion recovery images characteristic of demyelination in the white matter, most of them periventricular and subcortical; in 17 (31.5%) and 27 cases (50.9%), MRI showed infratentorial and basal ganglia involvement, respectively; and in 26 cases (72.2%), enhancing lesions were detected. Cerebrospinal fluid analysis showed pleocytosis (8 to 22 cells/mm³), elevated protein levels (49 to 81 mg/dL), and oligoclonal bands in 11 (34.4%), 9 (28.1%), and 2 (11.1%) patients, respectively. Improvement was seen in 59 cases (96.7%) after levamisole discontinuation, and 60 patients (96.8%) received high-dose corticosteroids. Missing data for many included patients and language restrictions were the key limitations. Neurologists should be aware of the clinical features of CNS demyelination following isolated levamisole use to avoid diagnostic errors.

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