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LETTER TO EDITOR
Year : 2012  |  Volume : 60  |  Issue : 5  |  Page : 526-527

Choreoathetosis in herpes simplex encephalitis relapse with bilateral thalamic gliotic lesions on magnetic resonance imaging


Department of Radiology, St. Johns Medical College Hospital, Sarjapur Road, Bangalore, India

Date of Web Publication3-Nov-2012

Correspondence Address:
Anisha S Tandon
Department of Radiology, St. Johns Medical College Hospital, Sarjapur Road, Bangalore
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.103208

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How to cite this article:
Tandon AS. Choreoathetosis in herpes simplex encephalitis relapse with bilateral thalamic gliotic lesions on magnetic resonance imaging. Neurol India 2012;60:526-7

How to cite this URL:
Tandon AS. Choreoathetosis in herpes simplex encephalitis relapse with bilateral thalamic gliotic lesions on magnetic resonance imaging. Neurol India [serial online] 2012 [cited 2019 Sep 23];60:526-7. Available from: http://www.neurologyindia.com/text.asp?2012/60/5/526/103208


Sir,

An 11-month-old male child presented to the emergency department with high fever of 101Ί F, altered sensorium and generalized tonic-clonic seizures of 2 days' duration, with no focal neurological deficits. Magnetic resonance imaging (MRI) revealed cortical and sub-cortical areas of T1 and T2 prolongation in the gray and white matter of bilateral parietal paramedian regions and bilateral cingulate gyri in the anterior cerebral artery territories with restricted diffusion [Figure 1]a,b. Focal lesions with restricted diffusion were also present in bilateral medial thalami [Figure 1]c. There was no significant meningeal enhancement. Cerebrospinal fluid examination (CSF) examination revealed protein: 36 mg/dL; glucose: 90 mg/dL; white blood cell count: 70 cells/mm 3 (2 neutrophils, 38 lymphocytes, 30 macrophages); and red blood cell count: 6 cells/mm 3 . CSF qualitative HSV Polymerase Chain Reaction (PCR) results were positive and sub-typing revealed HSV 1 virus. Based on these findings, diagnosis of herpes simplex virus (HSV) encephalitis was made and he was started on intravenous acyclovir therapy 20 mg/ kg/ day every 8 h for 10 days. He clinically improved and was discharged. Three weeks later, he presented with choreoathetosis. Repeat MRI revealed encephalomalacic transformation of bilateral parietal and cingulate gyrus regions with bilateral medial thalamic gliosis [Figure 2]a and b. Diffuse T2 hyperintense signal was present in bilateral fronto-parietal deep white matter with no restriction on diffusion images [Figure 2]c. Repeat CSF analysis revealed protein: 100 mg/dL; glucose: 35 mg/ dL; white blood cell count: 62 cells/mm 3 (0 neutrophils, 56 lymphocytes, 5 macrophages, 1 eosinophil); and red blood cell count: 3 cells/mm 3 . Repeat CSF qualitative HSV PCR was negative. In spite of a negative HSV PCR, he was initially started on intravenous acyclovir. Later a diagnosis of post-infectious immune-mediated encephalitis rather than re-infection was considered based on the imaging and CSF findings. Steroid therapy was started and acyclovir was discontinued after 48 h. Initially, the patient received intravenous dexamethasone 0.5 mg/kg for 24 h followed by a two-week course of prednisolone 2 mg/kg orally. He was symptom-free within seven days of starting steroid therapy and recovered completely in two weeks.
Figure 1: (a) Axial diffusion-weighted MRI shows restricted diffusion in bilateral medial thalami (white arrowheads) and in bilateral parieto-occipital cortex and underlying white matter (white arrows), (b) Axial diffusion-weighted MRI shows restricted diffusion in bilateral parieto-occipital cortex and underlying white matter (white arrows), (c) Axial T2W MRI shows areas of T2 prolongation in bilateral parieto-occipital cortex and white matter (white arrows)

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Figure 2: (a) Follow-up MRI after 3 weeks, axial T1W image shows hypointense focal lesions in bilateral thalami (white arrowheads) and in bilateral medial occipital lobes (white arrows), (b) Follow-up MRI after 3 weeks, axial T1W image shows hypointense signal suggesting gliotic transformation in bilateral parietal cortices (white arrows), (c) Follow-up MRI after 3 weeks, axial T1W image shows hypointense signal suggesting gliotic transformation in bilateral parietal cortices (white arrows)

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The presenting features of HSV encephalitis usually include fever, seizures, drowsiness, headache and focal neurological deficits. [1] Relapses of HSV encephalitis have varying symptoms, movement disorder, particularly choreoathetosis has been mentioned as a symptom of relapse. [1],[2] Hargrave and Webb [3] studied 20 patients of HSV encephalitis with relapse and all of them had choreoathetosis. Imaging (computed tomography (CT) and/or MRI was done both in the acute phase and during relapse in 16 patients and in all of them basal ganglia and thalamus were normal on imaging. De Tiθge et al., [4] analyzed 42 pediatric patients of HSV encephalitis with relapse and described two distinct clinical syndromes: (1) Post-infectious immune-mediated syndrome characterized by choreoathetosis and (2) re-infection characterized by fever with new neurologic symptoms and no movement disorder. They also suggested that the immune-mediated syndrome does not show new lesions on imaging, whereas re-infection may present with new lesions. Of note, they reiterated that since structural and functional abnormalities have not been documented on imaging, the pathogenesis of movement disorders is unclear. [4] Kullnat et al., [5] described a single case of HSV encephalitis with relapse with choreoathetosis. The clinical features in this case were consistent with an immune-mediated syndrome, but the imaging showed new thalamic lesions. In our patient, the initial imaging revealed bilateral thalamic lesions and imaging following relapse showed gliosis in the medial thalami. Re-infection has been described to have a positive CSF PCR for herpes simplex virus and usually presents with new lesions on imaging. On the other hand, immune-mediated relapse will be CSF HSV PCR-negative with no new lesions on imaging. [6] The distinction between active re-infection and post-infectious inflammatory encephalopathy is crucial, because the former would be managed with a repeat course of acyclovir, and the latter with corticosteroids and, possibly, intravenous immunoglobulin or plasmapheresis. [6] The presence of gliotic lesions in the medial thalami in our patient ruled out re-infection and aided in the management with corticosteroids.

 
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1.Mustafa AM, Salih, Heba Y, El Khashab, Hamdy H, Hassan, et al. A study on herpes simplex encephalitis in 18 children, including 3 relapses. Open Pediatr Med J 2009;3:48-57.  Back to cited text no. 1
    
2.Buff BL Jr, Mathews VP, Elster AD. Bacterial and parenchymal infections of the brain. Top Magn Reson Imaging 1994;6:11-21.  Back to cited text no. 2
    
3.Hargrave DR, Webb DW. Movement disorders in association with herpes simplex virus encephalitis in children: A review. Dev Med Child Neurol 1998;40:640-2.  Back to cited text no. 3
    
4.De Tiège X, Rozenberg F, Des Portes V, Lobut JB, Lebon P, Ponsot G, et al. Herpes simplex encephalitis in children: Differentiation of two neurological entities. Neurology 2003;61:241-3.  Back to cited text no. 4
    
5.Kullnat MW, Morse PR. Choreoathetosis after herpes simplex encephalitis with basal ganglia involvement on MRI. Pediatrics 2008;121:e1003-7.  Back to cited text no. 5
    
6.Ono Y, Manabe Y, Nishimura H, Kono S, Narai H, Omori N, et al. Unusual progression of herpes simplex encephalitis with basal ganglia and extensive white matter involvement. Neurol Int 2009;1:e9, 32-34.  Back to cited text no. 6
    


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