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      Co-occurrence of Marchiafava-Bignami Disease and Alcoholic Polyneuropathy in Chronic Alcoholic Patient Who Had Past History of Wernicke Encephalopathy: a Case Report

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          Abstract

          Marchiafava-Bignami disease (MBD), Wernicke encephalopathy (WE) and alcoholic polyneuropathy (AP) are distinct diseases and all have strong relationship with chronic alcoholism. A 70-year-old male who had altered mentality and ataxia of both lower limbs and had past history of WE 3 years previously admitted with 6 months history of impaired walking. He also had a symptom of altered sensorium by impaired consciousness for 2 days. In brain magnetic resonance imaging, the body, splenium of corpus callosum and bilateral frontal cortex were involved. The patient was diagnosed with MBD on the basis of the clinical features and the brain imaging findings. The electrodiagnostic findings implied demyelinating neuropathy in all extremities. He failed to recover his mentality and the function of the limbs remained poor finishing several treatment options including medications and physical therapy. The poor prognosis of this patient is thought to be associated with cortical involvement of MBD. We reported this very rare case who was affected by 3 distinct diseases of MBD, AP, and WE as complications of chronic alcohol abuse. Moreover, the case was relevant to a rare clinical presentation of MBD with cortical involvement which was associated with poor prognosis.

          Highlights

          • • Three distinct neurologic diseases occurred in one chronic alcoholic patient.

          • • MBD which present poor prognosis is associated with cortical involvement.

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          Most cited references12

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          Alcohol-related peripheral neuropathy: a systematic review and meta-analysis

          The primary aim of this systematic review was to establish the prevalence, character, and risk factors of peripheral neuropathy amongst chronic alcohol abusers and to identify the most appropriate management strategies. In this review, possible pathogenetic mechanisms are also discussed. A systematic, computer-based search was conducted using the PubMed database. Data regarding the above parameters were extracted. 87 articles were included in this review, 29 case–control studies, 52 prospective/retrospective cohort studies and 2 randomised control trials, 1 cross sectional study, and 3 population-based studies. The prevalence of peripheral neuropathy amongst chronic alcohol abusers is 46.3% (CI 35.7– 57.3%) when confirmed via nerve conduction studies. Alcohol-related peripheral neuropathy generally presents as a progressive, predominantly sensory axonal length-dependent neuropathy. The most important risk factor for alcohol-related peripheral neuropathy is the total lifetime dose of ethanol, although other risk factors have been identified including genetic, male gender, and type of alcohol consumed. At present, it is unclear what the pathogenetic mechanisms for the development of neuropathy amongst those who chronically abuse alcohol are, and therefore, it is unknown whether it is attributed to the direct toxic effects of ethanol or another currently unidentified factor. There is presently sparse data to support a particular management strategy in alcohol-related peripheral neuropathy, but the limited data available appears to support the use of vitamin supplementation, particularly of B-vitamin regimens inclusive of thiamine.
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            Neuroimaging findings in acute Wernicke's encephalopathy: review of the literature.

            Wernicke's encephalopathy is an acute neurological syndrome resulting from thiamine (vitamin B1) deficiency. Early recognition is important because timely thiamine supplementation can reverse the clinical features of the disease. The aim of this article is to provide an update on the typical and atypical neuroimaging findings of the acute phase of the disease. Wernicke's encephalopathy is characterized by a quite distinct pattern of MR alterations, which include symmetrical alterations in the thalami, mamillary bodies, tectal plate, and periaqueductal area, but atypical alterations may also been seen. A thorough knowledge of the neuroimaging findings of Wernicke's encephalopathy will assist in arriving at an early diagnosis, thus reducing the morbidity and mortality associated with this disease.
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              Acute Marchiafava-Bignami disease with extensive diffusion restriction and early recovery: case report and review of the literature.

              Marchiafava-Bignami disease (MBD) is a neurological disorder that has been found to be associated with chronic alcoholism and malnutrition. MBD classically results in acute edema and demyelination of the corpus callosum. Edema of the complete corpus callosum has been described to be an unfavorable prognostic factor. We present an acute onset of MBD with diffusion restriction of the complete corpus callosum and symmetric bilateral extension into the semioval center, that almost completely resolved clinically as well as in MRI only 3 days later. With early detection and treatment, the prognosis of MBD may be good even in cases with severe diffusion restriction of the complete corpus callosum.
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                Author and article information

                Journal
                Brain Neurorehabil
                Brain Neurorehabil
                BN
                Brain & NeuroRehabilitation
                Korean Society for Neurorehabilitation
                1976-8753
                2383-9910
                July 2021
                30 June 2021
                : 14
                : 2
                : e19
                Affiliations
                Department of Physical Medicine and Rehabilitation, Inje University Busan Paik Hospital, College of Medicine, Inje University, Busan, Korea.
                Author notes
                Correspondence to Mi Ja Eom. Department of Physical Medicine and Rehabilitation, Inje University Busan Paik Hospital, College of Medicine, Inje University, 75 Bokji-ro, Busanjin-gu, Busan 47392, Korea. reheom@ 123456inje.ac.kr
                Author information
                https://orcid.org/0000-0003-4595-0205
                https://orcid.org/0000-0003-3651-9755
                https://orcid.org/0000-0002-3652-045X
                Article
                10.12786/bn.2021.14.e19
                9879502
                13aa1a5a-ab36-43e9-8733-2f3e383c8685
                Copyright © 2021. Korean Society for Neurorehabilitation

                This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( https://creativecommons.org/licenses/by-nc/4.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                : 11 May 2021
                : 21 June 2021
                : 28 June 2021
                Categories
                Case Report

                marchiafava-bignami disease,wernicke encephalopathy,alcoholic neuropathy,magnetic resonance imaging,nerve conduction study

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