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Acute disseminated encephalomyelitis complicating dengue infection with neuroimaging mimicking multiple sclerosis: A report of two cases

Viswanathan S, Botross N, Rusli BN, Riad A

Mult Scler Relat Disord, 2016 Nov;10:112-115.
PMID: 27919476 DOI: 10.1016/j.msard.2016.10.001

Acute disseminated encephalomyelitis (ADEM) complicating dengue infection is still exceedingly rare even in endemic countries such as Malaysia. Here we report two such cases, the first in an elderly female patient and the second in a young man. Both presented with encephalopathy, brainstem involvement and worsening upper and lower limb weakness. Initial magnetic resonance imaging (MRI) of the brain was normal in the first case. Serum for dengue Ig M and NS-1 was positive in both cases. Cerebrospinal fluid (CSF) showed pleocytosis in both with Dengue IgM and NS-1 positive in the second case but not done in the first. MRI brain showed changes of perpendicular subcortical palisading white matter, callosal and brainstem disease mimicking multiple sclerosis (MS) in both patients though in the former case there was a lag between the onset of clinical symptoms and MRI changes which was only clarified on reimaging. The temporal evolution and duration of the clinical symptoms, CSF changes and neuroimaging were more suggestive of Dengue ADEM rather than an encephalitis though initially the first case began as dengue encephalitis. Furthermore in dengue encephalitis neuroimaging is usually normal or rarely edema, haemorrhage, brainstem, thalamic or focal lesions are seen. Therefore, early recognition of ADEM as a sequelae of dengue infection with neuroimaging mimicking MS and repeat imaging helped in identifying these two cases. Treatment with intravenous steroids followed by maintenance oral steroids produced good outcome in both patients.

Matched MeSH terms: Encephalomyelitis, Acute Disseminated/cerebrospinal fluid; Encephalomyelitis, Acute Disseminated/complications*; Encephalomyelitis, Acute Disseminated/drug therapy
Fulltext Acute disseminated encephalomyelitis: a report of six cases

Thomas GST, Hussain IHMI

Med J Malaysia, 2004 Aug;59(3):342-51.
PMID: 15727380 MyJurnal

Six children with Acute Disseminated Encephalomyelitis (ADEM) were seen at the Penang Hospital over a two year period (July 1999-June 2001). Diagnosis was based upon typical clinical features and characteristic findings on neuroimaging. Cerebrospinal fluid examination and other investigations were done, where appropriate, to rule out other causes of central nervous system disease. Three children had a prodromal illness. The most common presenting symptoms were fever, seizures, ataxia, focal neurological deficits and labile mood. Two children presented with status epilepticus. All children had an abnormal neurological examination. Brain magnetic resonance imaging revealed hyperintense signals on T2-weighted and FLAIR sequences in the subcortical and deep white matter regions of the frontal, parietal, and temporal lobes, as well as in the thalami, cerebellum and brainstem. One child had multiphasic disseminated encephalomyelitis (three episodes). The child with multiphasic disease had only one treated episode, and has suffered mild disability. Three children were treated with either methylprednisolone or immunoglobulins, and remain well. One child received both treatments but expired as a result of severe gastrointestinal bleeding from the use of methylprednisolone. The child who was not treated has severe disability.

Matched MeSH terms: Encephalomyelitis, Acute Disseminated/complications; Encephalomyelitis, Acute Disseminated/diagnosis*; Encephalomyelitis, Acute Disseminated/therapy
Fulltext Intravenous immunoglobulin in the treatment of acute disseminated encephalomyelitis

Kanaheswari Y, Baizura J, Zulfiqar A

Med J Malaysia, 2004 Mar;59(1):103-7.
PMID: 15535344

We describe a case of acute disseminated encephalomyelitis in a child. This case is unusual in that the illness was characterised by recurrent episodes rather than a monophasic course and that the choice of treatment was intravenous immunoglobulin over corticosteroids. The rapid and remarkable recovery is highlighted and a review of the treatment for this rare condition is discussed.

Matched MeSH terms: Encephalomyelitis, Acute Disseminated/diagnosis; Encephalomyelitis, Acute Disseminated/drug therapy*
Multiphasic disseminated encephalomyelitis followed by optic neuritis in a child with gluten sensitivity

Ching BH, Mohamed AR, Khoo TB, Ismail HI

Mult Scler, 2015 Aug;21(9):1209-11.
PMID: 26199345 DOI: 10.1177/1352458515593404

Multiphasic disseminated encephalomyelitis (MDEM) followed by optic neuritis (ON) has been described as a new entity in recent years. Gluten encephalopathy has also been recognized as a neurological manifestation of celiac disease. Accurate diagnosis of both is important due to the therapeutic implications. We report a girl presenting with recurrent encephalopathic polyfocal demyelinating episodes followed by optic neuritis, and a clinical history suggestive of gluten sensitivity. She had persistently high ESR, neutrophilia, and tested positive for anti-MOG (myelin oligodendrocyte glycoprotein) antibody. She responded well to methylprednisolone in each relapse, and achieved remission for a year after azathioprine was added.

Matched MeSH terms: Encephalomyelitis, Acute Disseminated/complications*; Encephalomyelitis, Acute Disseminated/pathology
Acute disseminated encephalomyelitis in dengue viral infection

Wan Sulaiman WA, Inche Mat LN, Hashim HZ, Hoo FK, Ching SM, Vasudevan R, et al.

J Clin Neurosci, 2017 Sep;43:25-31.
PMID: 28625589 DOI: 10.1016/j.jocn.2017.05.033

Dengue is the most common arboviral disease affecting many countries worldwide. An RNA virus from the flaviviridae family, dengue has four antigenically distinct serotypes (DEN-1-DEN-4). Neurological involvement in dengue can be classified into dengue encephalopathy immune-mediated syndromes, encephalitis, neuromuscular or dengue muscle dysfunction and neuro-ophthalmic involvement. Acute disseminated encephalomyelitis (ADEM) is an immune mediated acute demyelinating disorder of the central nervous system following recent infection or vaccination. This monophasic illness is characterised by multifocal white matter involvement. Many dengue studies and case reports have linked ADEM with dengue virus infection but the association is still not clear. Therefore, this article is to review and discuss concerning ADEM in dengue as an immune-medicated neurological complication; and the management strategy required based on recent literature.

Matched MeSH terms: Encephalomyelitis, Acute Disseminated
Fulltext Acute disseminated encephalomyelitis (ADEM) presenting with seizures secondary to anti-tetanus toxin vaccination

Hamidon BB, Raymond AA

Med J Malaysia, 2003 Dec;58(5):780-2.
PMID: 15190671

Acute disseminated encephalomyelitis (ADEM) is an inflammatory demyelinating disease of the central nervous system (CNS) that is known to occur spontaneously in association with specific and non-specific viral illnesses and after vaccination against various pathogens. Although it is often a self-limited monophasic illness, the fatality rate is estimated to be as high as 20%, and many patients suffer residual neurologic impairment 1. The diagnosis is mainly based on clinical and radiological findings. The clinical presentation varies from merely, an asymptomatic condition to loss of consciousness, seizures, ataxia, optic neuropathy, cranial nerve palsies, and motor dysfunction. MRI of the brain is the single most important diagnostic radiological investigation and can facilitate early diagnosis and prompt treatment. This case report describes a patient with ADEM presenting with only seizures after vaccination with anti-tetanus toxin.

Matched MeSH terms: Encephalomyelitis, Acute Disseminated/etiology*
Fulltext Acute Disseminated Encephalomyelitis (ADEM) Presenting with Bilateral Optic Neuritis

Sapuan S, Basri H

Malays J Med Sci, 2007 Jan;14(1):71-4.
PMID: 22593657 MyJurnal

A 43-year old lady presented with progressive loss of vision in both eyes followed by rapid deterioration of consciousness within the next few days. This was preceded by a viral infection one week before her presentation. At presentation she had evidence of meningism and signs of bilateral upper motor neuron lesions and was managed initially as acute meningoencephalitis with antibiotics. The brain CT was within normal limits but subsequent MRI of the brain revealed multiple foci of hyperintense lesions on T2-weighted and FLAIR images. The cerebrospinal fluid examination revealed lymphocytosis, and normal protein and glucose levels. Cultures of the CSF were negative. She was managed as acute disseminated encephalomyelitis (ADEM) with high-dose of intravenous methlyprednisolone one gram/day for three consecutive days followed by oral prednisolone 60 mg/day. Despite the management she lapsed into coma and succumbed to her illness nine days after admission.

Matched MeSH terms: Encephalomyelitis, Acute Disseminated
Smallpox vaccination: complications and present role

Balasundram R

Med J Malaya, 1972 Dec;27(2):89-94.
PMID: 4145716
Matched MeSH terms: Encephalomyelitis, Acute Disseminated/etiology
Unusual case of cerebral demyelination and bilateral optic neuritis in an infant with suppurative BCG lymphadenitis

Anandakrishnan P, Khoo TB

BMJ Case Rep, 2018 May 30;2018.
PMID: 29848532 DOI: 10.1136/bcr-2018-224496

Cerebral demyelination and optic neuritis are often seen in children with acute disseminated encephalomyelitis following various infections and immunisations. An eight month old girl presented with a left axillary lymph node swelling and an erythematous lace-like rash over her cheeks and trunk. She then developed acute encephalopathy, bilateral nystagmus, right hemiparesis and left facial nerve palsy. Her electroencephalogram showed an encephalopathic process and visual evoked response study were grossly abnormal. Her MRI brain showed hyperintensities in the midbrain, pons and bilateral cerebellar peduncles. She was treated as postinfectious cerebral demyelination with intravenous antibiotics, methylprednisolone and immunoglobulin. Left axillary lymph node excision biopsy and GeneXpert test detected Mycobacterium tuberculosis complex that prompted initiation of antituberculous therapy. Her chest X-ray and cerebrospinal fluid examinations for tuberculosis were normal. She showed significant recovery after 2 weeks. This case illustrates a rare presentation of cerebral demyelination and bilateral optic neuritis following suppurative BCG lymphadenitis.

Matched MeSH terms: Encephalomyelitis, Acute Disseminated/chemically induced*
Development of the clinical assessment scale in autoimmune encephalitis

Lim JA, Lee ST, Moon J, Jun JS, Kim TJ, Shin YW, et al.

Ann Neurol, 2019 03;85(3):352-358.
PMID: 30675918 DOI: 10.1002/ana.25421

OBJECTIVE: There is no scale for rating the severity of autoimmune encephalitis (AE). In this study, we aimed to develop a novel scale for rating severity in patients with diverse AE syndromes and to verify the reliability and validity of the developed scale.
METHODS: The key items were generated by a panel of experts and selected according to content validity ratios. The developed scale was initially applied to 50 patients with AE (development cohort) to evaluate its acceptability, reproducibility, internal consistency, and construct validity. Then, the scale was applied to another independent cohort (validation cohort, n = 38).
RESULTS: A new scale consisting of 9 items (seizure, memory dysfunction, psychiatric symptoms, consciousness, language problems, dyskinesia/dystonia, gait instability and ataxia, brainstem dysfunction, and weakness) was developed. Each item was assigned a value of up to 3 points. The total score could therefore range from 0 to 27. We named the scale the Clinical Assessment Scale in Autoimmune Encephalitis (CASE). The new scale showed excellent interobserver (intraclass correlation coefficient [ICC] = 0.97) and intraobserver (ICC = 0.96) reliability for total scores, was highly correlated with modified Rankin scale (r = 0.86, p

Matched MeSH terms: Encephalomyelitis, Acute Disseminated/complications; Encephalomyelitis, Acute Disseminated/physiopathology; Encephalomyelitis, Acute Disseminated/psychology