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| TOPIC OF THE ISSUE: LETTER TO EDITOR |
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| Year : 2010 | Volume
: 58
| Issue : 4 | Page : 599-601 |
Acute disseminated encephalomyelitis following dengue hemorrhagic fever
Challa Sundaram1, Shantveer G Uppin1, KV Dakshinamurthy2, Rupam Borgahain3
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 Acceptance | 28-Jul-2010 |
| Date of Web Publication | 24-Aug-2010 |
Correspondence Address:
Challa Sundaram
Department of Pathology, Nizam's Institute of Medical Sciences, Punjagutta, Hyderabad
India
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/0028-3886.68666
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 |
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.
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[Figure 1], [Figure 2]
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