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TOPIC OF THE ISSUE: LETTER TO EDITOR
Year : 2010  |  Volume : 58  |  Issue : 4  |  Page : 599-601

Acute disseminated encephalomyelitis following dengue hemorrhagic fever


1 Department of Pathology, Nizam's Institute of Medical Sciences, Punjagutta, Hyderabad, India
2 Department of Nephrology, Nizam's Institute of Medical Sciences, Punjagutta, Hyderabad, India
3 Department of Neurology, Nizam's Institute of Medical Sciences, Punjagutta, Hyderabad, India

Date of Acceptance28-Jul-2010
Date of Web Publication24-Aug-2010

Correspondence Address:
Challa Sundaram
Department of Pathology, Nizam's Institute of Medical Sciences, Punjagutta, Hyderabad
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.68666

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How to cite this article:
Sundaram C, Uppin SG, Dakshinamurthy K V, Borgahain R. Acute disseminated encephalomyelitis following dengue hemorrhagic fever. Neurol India 2010;58:599-601

How to cite this URL:
Sundaram C, Uppin SG, Dakshinamurthy K V, Borgahain R. Acute disseminated encephalomyelitis following dengue hemorrhagic fever. Neurol India [serial online] 2010 [cited 2019 May 21];58:599-601. Available from: http://www.neurologyindia.com/text.asp?2010/58/4/599/68666


Sir,

Dengue infection is caused by a flavivirus, and the nervous system involvement is seen with the serotypes 2 and 3. Neurologic manifestations occur in 4% to 5% of patients, [1] and the manifestations include encephalopathy, encephalitis, Guillain-Barre syndrome (GBS), myelitis, meningitis, acute disseminated encephalomyelitis (ADEM), facial and ulnar mononeuropathy and stroke, both ischemic and hemorrhagic. [2],[3],[4],[5],[6],[7],[8],[9],[10],[11],[12],[13] ADEM following dengue infections is very infrequent, and only very few cases have been documented. [8],[13] We report an autopsy study of a patient with dengue infection and ADEM.

A 27-year-old male presented with high-grade fever associated with chills and rigors of 5 days' duration; headache, vomiting, of 3 days' duration; and altered sensorium of 1 day's duration. He had history of mild hematochezia. On examination, his vitals were stable, and he had mild hepatomegaly. Central nervous system (CNS) examination showed obtunded level of sensorium and mild terminal neck stiffness. Investigations revealed hemoglobin, 16.2 g/dL; total leukocyte count, 14,500/μL; platelet count, 20,000/μL; blood urea, 84 mg/dL; serum creatinine, 4.3 mg/dL; Serum glutamate oxaloacetic transaminase (SGOT), 778 IU/L; Serum glutamate pyruvic transaminase (SGPT), 1043 IU/L; and prothrombin time, activated thromboplastin time were prolonged. Serum IgG and IgM dengue antibodies were positive. Antibodies for leptospira were negative. Peripheral smear for malarial parasite was negative. Magnetic resonance imaging (MRI) of brain showed hypointense lesions on T1-weighted images involving bilateral hippocampi, thalami, cerebellar hemispheres and posterior part of the pons. These lesions were hyperintense on T2-weighted images. There was no contrast enhancement.

During hospital stay, the patient developed myalgia, jaundice, conjunctival hemorrhage, hematuria, oliguria, shortness of breath and became stuporous. Patient further deteriorated and developed adult respiratory distress syndrome (ARDS), acute kidney injury and metabolic acidosis. Chest radiograph at this stage showed bilateral fluffy shadows. He was put on mechanical ventilator and managed on the lines of ARDS. For acidosis and acute kidney injury, he was started on peritoneal dialysis. Patient rapidly deteriorated and succumbed to his illness.

A complete autopsy was performed. At autopsy, the brain was edematous; and on coronal sections, there were bilaterally symmetrical gray-white granular areas with foci of hemorrhage involving bilateral hippocampi, thalami, cerebellar hemispheres and brainstem [Figure 1] On histological examination, the lesions were clearly demarcated from the adjacent brain parenchyma and showed perivenous demyelination, macrophage infiltration and perivascular lymhomononuclear infiltrates. There were few reactive gemistocytes. Luxol-fast blue stain for myelin showed loss of myelin, and immunohistochemistry for neurofilament (Dako, USA; 1:50) showed preservation of axons [Figure 2]. There were foci of hemorrhage around blood vessels. Sections from the lungs showed hyaline membrane disease, and section from both the kidneys showed acute tubular necrosis. Sections from liver and spleen showed congestion. The final pathological diagnosis was ADEM with multi-organ failure.
Figure 1 : Coronal sections of brain showed gray-white granular areas with foci of hemorrhage involving both hippocampi, thalami, brainstem and both cerebellar hemispheres

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Figure 2 : (a and b) Histopathology from the lesions showed perivenous demyelination, macrophage collection and hemorrhages (H and E, ×200). (c and d): Perivenous demyelination (Luxol fast blue - cresyl violet (LFBCV) ×200)

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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. [3],[4],[5],[12] Our patient had thrombocytopenia, hemoconcentration, coagulopathy and multi-organ failure. In addition, he had bilaterally symmetrical involvement of hippocampi, thalami, cerebellar hemispheres and brainstem. Histologically, these lesions showed demyelination with foci of hemorrhages. Earlier reports have documented lesions in similar locations on computed tomography and MRI of the brain with partial neurologic recovery. [11],[13] The lesions were considered to be of demyelinating pathology. Ours is the first documented case of ADEM following dengue fever. Our patient had dengue hemorrhagic fever, and the focal hemorrhages seen in the lesions may be related to the thrombocytopenia.

 
 » 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  [PUBMED]  [FULLTEXT]  
2.Lum LC, Lam SK, Choy YS, George R, Harun F. Dengue encephalitis: a true entity? Am Trop Med Hyg 1996;54:256-9.  Back to cited text no. 2      
3.Solomon T, Dung NM, Vaughn DW, Kneen R, Thao LTT, Raengsakulrach B, et al. Neurological manifestations of dengue infection. Lancet 2000;355:1053-9.  Back to cited text no. 3      
4.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. 4      
5.Miagostovich MP, Ramos RG, Nicol AF, Nogueira RM, Cuzzi-Maya T, Oliveira AV, et al. Retrospective study on dengue fatal cases. Clin Neuropathol 1997;16:204-8.  Back to cited text no. 5  [PUBMED]    
6.Pancharoen C, Thisyakorn U. Neurological manifestations in dengue patients. Southeast Asian J Trop Med Public Health 2001;32:341-5.  Back to cited text no. 6  [PUBMED]    
7.Thisyakorn U, Thisyakorn C, Limpitiku W, Nisalak A. Dengue infection with central nervous system manifestations. Southeast Asian J Trop Med Public Health 1999;30:504-6.  Back to cited text no. 7      
8.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. 8  [PUBMED]  [FULLTEXT]  
9.Soares CN, Faria LC, Peralta JM, Freitas MRG, Puccioni-Sohler M. Dengue infection: neurological manifestations and cerebrospinal fluid (CSF) analysis. J Neurol Sci 2006;249:19-24.  Back to cited text no. 9      
10.Miranda de Sousa A, Puccioni-Sohler M, Dias Borges A, Fernandes Adorno L, Papais Alvarenga M, Papais Alvarenga RM. Post-dengue neuromyelitis optica: case report of a Japanese-desendent Brazilian child. J Infect Chemother 2006;12:396-8.  Back to cited text no. 10  [PUBMED]  [FULLTEXT]  
11.Chimelli L, Hahn MD, Netto MB, Ramos RG, Dias M G ray F. Dengue: neuropathological findings in 5 fatal cases from Brazil. Clin Neuropathol 1990;9:157-62.  Back to cited text no. 11      
12.Yeo PS, Pinheiro L, Tong P, Lim PL, Sitoyy YY. Hippocampal involvement in dengue fever. Singapore Med J 2005;46:647-50.  Back to cited text no. 12      
13.Kumble R, Jayakumar N, Perivvamba, Kovoor J, Ravishankar S, Kolar BS. Bilateral thalamic involvement in dengue infection. Neuro Int 2007;55:418-9.  Back to cited text no. 13      


    Figures

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