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   Table of Contents      
CASE REPORT
Year : 2016  |  Volume : 2  |  Issue : 1  |  Page : 54-56

Dengue presenting as hemorrhagic acute disseminated encephalomyelitis


1 Department of Medicine, Maulana Azad Medical College and LNH, New Delhi, India
2 Department of Medicine, AIIMS, New Delhi, India

Date of Web Publication25-Jan-2016

Correspondence Address:
Prayas Sethi
Department of Medicine, AIIMS, New Delhi
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2394-7438.174840

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  Abstract 

Dengue fever over the recent years has gained attention of not only the health care professionals and researchers, but also public at large and it is a major seasonal health care problem in the tropics and the developing countries. Mortality due to complications such as hemorrhage and shock is well-known and is a major concern for the physician. Neurological manifesations include myelitis and encephalitis have started to gain attention as they present with high mortality and morbidity. We report a case of dengue fever with predominant neurological manifestations of acute disseminated encephalomyelitis (ADEM). The patient presented with fever, vomiting, and altered sensorium of recent onset. On evaluation, the patient was positive for dengue serology and magnetic resonance imaging picture suggested ADEM.

Keywords: Dengue fever, disease, nervous system


How to cite this article:
Dewan R, Anuradha S, Sethi P, Ish P. Dengue presenting as hemorrhagic acute disseminated encephalomyelitis. MAMC J Med Sci 2016;2:54-6

How to cite this URL:
Dewan R, Anuradha S, Sethi P, Ish P. Dengue presenting as hemorrhagic acute disseminated encephalomyelitis. MAMC J Med Sci [serial online] 2016 [cited 2019 Oct 17];2:54-6. Available from: http://www.mamcjms.in/text.asp?2016/2/1/54/174840


  Introduction Top


Dengue infection is caused by a flavivirus and the nervous system involvement is seen in infection with serotypes 2 and 3. Neurologic manifestations occur in 4–5% of patients and include encephalopathy, encephalitis, Guillain-Barre syndrome, myelitis, meningitis, acute disseminated encephalomyelitis (ADEM), facial and ulnar mononeuropathy, and stroke, both ischemic and hemorrhagic.[1],[2],[3],[4] ADEM following dengue infections is very infrequent and very few cases have been documented. We present a case of ADEM with hemorrhage.


  Case Report Top


A 17-year-old male presented with the complaints of fever and vomiting of 3 days duration and altered sensorium for 1 day. Fever was high grade, continuous, and associated with rash all over the body. On examination, the patient was hyperventilating and unresponsive, Glasgow Coma Scale 7 (E2V2M3). His pulse was 92 beats per minute, radial artery, blood pressure was 100/60, and a generalized maculopapular rash was present. On neurological examination, patient's left lower limb was externally rotated with decreased movement of left half of body on painful stimuli and bilateral extensor plantar response. Pupillary reflexes were normal. Rest of the systemic examination was unremarkable. Investigations revealed hemoglobin: 15.4 g/dl, hematocrit: 46%, white cell count: 14,600/mm3, with polymorphs 53% and lymphocytes 46%, platelet count: 40,000/mm3, blood urea of 26 mg/dl, serum creatinine: 0.6 mg/dl, alanine aminotransferase: 38 U/L, and aspartate aminotransferase: 95 U/L. Cerebrospinal fluid examination showed no cells and elevated protein (228 mg/dl) and normal glucose (75 mg/dl; blood glucose was 110 mg/dl). NS1 antigen and IgM antibody were positive for dengue.

Noncontrast computed tomography head revealed subtle foci of hyperdensity with surrounding hypodensities seen in right ganglio-thalamic complex causing ipsilateral ventricular compression. The patient was put on decongestive measures to reduce intracranial tension. Magnetic resonance imaging (MRI) brain [Figure 1],[Figure 2],[Figure 3] was suggestive of large area of T1- and T2-hypo/fluid attenuated inversion recovery hyperintensity in right basal ganglia, thalamus, cerebral peduncle with hemorrhagic foci, with similar areas in bilateral frontal, right temporal, and bilateral parietal white matter predominantly in subcortical location suggestive of hemorrhagic ADEM. Despite adequate supportive care and management, the patient's condition did not improve and he succumbed to his illness on the next day.
Figure 1: Axial T2-weighted magnetic resonance image shows ill-defined hyper intense signal in right ganglia-thalamic complex predominantly involving the lentiform and caudate nucleus with associated mass effect as evident by effacement of ipsilateral lateral ventricle and midline shift toward left side

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Figure 2: Coronal fluid attenuated inversion recovery images show hyper intensity in right basal ganglia and minimal extension into the right cerebral peduncle

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Figure 3: Axial flash image shows multiple foci of blooming in the right ganglio-thalamic region suggestive of hemorrhagic foci

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  Discussion Top


ADEM is an immune-mediated disease of the brain. It usually occurs following a viral infection, but may appear following vaccination, bacterial, or parasitic infection.[5] The annual incidence of ADEM is reported to be 0.4–0.8/100,000 and the disease more commonly affects children and young adults, probably related to the high frequency of exanthematous and other infections and vaccination in this age group.[6],[7],[8],[9],[10] In dengue infection, the pathophysiology of neurologic manifestations may be related to direct viral invasion; systemic complications related to dengue infection; or immune-mediated, autoimmune reaction secondary to dengue infection.[11],[12],[13]

Imaging studies in ADEM following dengue are few with MRI showing white matter lesions in the centrum semiovale, corona radiata, thalamus, extensive involvement of the frontal, parietal, and temporal white matter as well as the corpus callosum. White matter lesions also involved the brainstem, bilateral thalami, and cerebellum. Demyelinating lesions with or without foci of hemorrhage on MRI are probably pathognomonic of ADEM following dengue infection.[14] The pathological hallmark of ADEM is perivenular inflammation with demyelination of neurons usually in the white matter, while if hemorrhage occurs due to necrotizing vasculitis of venules, it results in acute hemorrhagic leukoencephalitis which has a very fulminant course and high mortality as seen in our patient.[15]

The alarmingly rising trend in dengue fever and dengue hemorrhagic fevers with increased morbidity and mortality prompted us to report this uncommon yet fatal outcome. A high index of suspicion must be kept for early recognition of these potential complications, especially if patient presents with associated neurological symptoms.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Puccioni-Sohler M, Soares CN, Papaiz-Alvarenga R, Castro MJ, Faria LC, Peralta JM. Neurologic dengue manifestations associated with intrathecal specific immune response. Neurology 2009;73:1413-7.  Back to cited text no. 1
    
2.
Solomon T, Dung NM, Vaughn DW, Kneen R, Thao LT, Raengsakulrach B, et al. Neurological manifestations of dengue infection. Lancet 2000;355:1053-9.  Back to cited text no. 2
    
3.
Misra UK, Kalita J, Syam UK, Dhole TN. Neurological manifestations of dengue virus infection. J Neurol Sci 2006;244:117-22.  Back to cited text no. 3
    
4.
Yamamoto Y, Takasaki T, Yamada K, Kimura M, Washizaki K, Yoshikawa K, et al. Acute disseminated encephalomyelitis following dengue fever. J Infect Chemother 2002;8:175-7.  Back to cited text no. 4
    
5.
Alvord BC Jr. Demyelinating diseases. In: Vinken PJ, Bruyen GW, editors. Handbook of Clinical Neurology. Vol. 3. Amsterdam: Elsevier Publisher BV; 1985. p. 467-502.  Back to cited text no. 5
    
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Ascherio A, Munger K. Epidemiology of multiple sclerosis: From risk factors to prevention. Semin Neurol 2008;28:17-28.  Back to cited text no. 6
    
7.
Panicker JN. Acute Disseminated Encephalomyelitis: Clinical Profile and Predictors of Outcome (Dissertation). Bangalore: National Institute of Mental Health and Neurosciences, NIMHANS (Deemed University); 2004.  Back to cited text no. 7
    
8.
Menge T, Hemmer B, Nessler S, Wiendl H, Neuhaus O, Hartung HP, et al. Acute disseminated encephalomyelitis: An update. Arch Neurol 2005;62:1673-80.  Back to cited text no. 8
    
9.
Anlar B, Basaran C, Kose G, Guven A, Haspolat S, Yakut A, et al. Acute disseminated encephalomyelitis in children: Outcome and prognosis. Neuropediatrics 2003;34:194-9.  Back to cited text no. 9
    
10.
Tenembaum S, Chamoles N, Fejerman N. Acute disseminated encephalomyelitis: A long-term follow-up study of 84 pediatric patients. Neurology 2002;59:1224-31.  Back to cited text no. 10
    
11.
Gera C, George U. Acute disseminating encephalomyelitis with hemorrhage following dengue. Neurol India 2010;58:595-6.  Back to cited text no. 11
[PUBMED]  Medknow Journal  
12.
Chowdhury RN, Siddiqui MR, Mahbub MS, Hasan OSI, Talukder A, Nabi S, et al. Dengue fever as a cause of acute disseminated encephalomyelitis (ADEM). J Med 2011;12:185-7.  Back to cited text no. 12
    
13.
Sundaram C, Uppin SG, Dakshinamurthy KV, Borgahain R. Acute disseminated encephalomyelitis following dengue hemorrhagic fever. Neurol India 2010;58:599-601.  Back to cited text no. 13
[PUBMED]  Medknow Journal  
14.
Murthy JM. Acute disseminated encephalomyelitis. Neurol India 2002;50:238-43.  Back to cited text no. 14
[PUBMED]  Medknow Journal  
15.
Meilof JF, Hijdra A, Vermeulen M. Successful recovery after high-dose intravenous methylprednisolone in acute hemorrhagic leukoencephalitis. J Neurol 2001;248:898-9.  Back to cited text no. 15
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]


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