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Table of Contents    
LETTER TO EDITOR
Year : 2014  |  Volume : 62  |  Issue : 4  |  Page : 439-441

Lingual epilepsia partialis continua as the presenting manifestation of herpes simplex encephalitis: Uncommon presentation of a common disease


Department of Neurology, KG Hospital and Post Graduate Medical Institute, Coimbatore, Tamil Naidu, India

Date of Web Publication19-Sep-2014

Correspondence Address:
Rajesh Shankar Iyer
Department of Neurology, KG Hospital and Post Graduate Medical Institute, Coimbatore, Tamil Naidu
India
Rajesh Shankar Iyer
Department of Neurology, KG Hospital and Post Graduate Medical Institute, Coimbatore, Tamil Naidu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.141230

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How to cite this article:
Iyer RS, Ramalingam Ramakrishnan TC, Iyer RS, Ramalingam Ramakrishnan TC. Lingual epilepsia partialis continua as the presenting manifestation of herpes simplex encephalitis: Uncommon presentation of a common disease . Neurol India 2014;62:439-41

How to cite this URL:
Iyer RS, Ramalingam Ramakrishnan TC, Iyer RS, Ramalingam Ramakrishnan TC. Lingual epilepsia partialis continua as the presenting manifestation of herpes simplex encephalitis: Uncommon presentation of a common disease . Neurol India [serial online] 2014 [cited 2020 Oct 31];62:439-41. Available from: https://www.neurologyindia.com/text.asp?2014/62/4/439/141230


Sir,

Clinical presentation of herpex simplex encephalitis (HSE) can sometimes be non-specific and challenging to the clinician. Here we describe a boy with HSE who presented with epilepsia partialis continua (EPC) of the tongue.

A 5-year-old boy presented with a two-day history of involuntary tongue movements. This was associated with mild dysarthtria and swallowing difficulties. Clinical examination revealed EPC of the tongue and bifacial jerks [Video 1]. Magnetic resonance imaging (MRI) of brain and EEG were normal. Cerebrospinal fluid (CSF) done on the third day of illness was normal. The EPC did not respond to parenteral lorazepam, phenytoin, or levetiracetam. Meanwhile, he developed intermittent drooling and swallowing difficulties. Considering the possibility of viral encephalitis, the child was given acyclovir injection empirically. Repeat MRI after 5 days showed hyperintense lesions in frontal operculum bilaterally [Figure 1]a-c, and EEG showed nonspecific slowing [Figure 1]d. A repeat CSF study showed 15 lymphocytes per ml, protein of 45 mg/dL, and normal glucose. Herpes simplex virus (HSV) DNA detection in the CSF by polymerase chain reaction (PCR) was positive. The child was continued on acyclovir treatment. EPC stopped after 3 weeks of illness. He recovered well without any neurological deficit.
Figure 1: FLAIR axial MR imaging of the brain shows bilateral frontal opercular hyperintense lesions (a). Contrast-enhanced coronal T1-weighted image showing mild enhancement on the left side (b). Susceptibility weighted image (c) shows hemorrhage within the left frontal hyperintense lesion. EEG (d) showing nonspecifi c delta slowing predominantly involving the posterior head region (bipolar montage, sensitivity of 20 ƒÊV/mm)

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EPC is a rare type of focal status epilepticus. It may be caused by vascular disorders (24-28%), encephalitides (immune-mediated or infectious 15-19%), neoplasms (5-16%), or metabolic and toxic causes (6-14%). [1],[2],[3] In approximately 19% of cases, the etiology is unknown. The most common cause of EPC in children is Rasmussen's encephalitis, while in adults it is cerebrovascular disease or neoplasms. Isolated EPC of the tongue (lingual EPC) is extremely uncommon, and to our knowledge, has not been reported in HSE. Lingual EPC, however, has been reported in Rasmussen's encephalitis. [4],[5]

Our patient probably suffers from the anterior opercular syndrome (AOS) characterized by paralysis of the masticatory, facial, and pharyngeal muscles resulting in dysarthria, dysphagia, and drooling. Bilateral frontal and parietal opercular lesions, as seen in our case, can cause this syndrome. AOS can occur in patients with HSE. [6] It can also be the initial presentation of HSE. [7],[8] EPC of the facial muscle is described appearing during the course of the disease. [7] However, lingual EPC occurring in AOS, more so as the presenting manifestation of HSE, is extremely uncommon. HSE has protean presentations, some being non-specific and preventing early diagnosis and treatment. Even the initial investigations including MRI, EEG, and CSF may not contribute to the diagnosis, as happened in our case. Negative CSF HSV PCR results can also occur if done very early in disease development, with subsequent tests becoming positive. [9] Early recognition of lingual EPC as a possible presenting feature of HSE may help the clinician to evaluate further despite normal initial diagnostic studies and empirically treat for HSE. This would expedite the diagnosis and improve the prognosis of the patient. [10]

 
  References Top

1.Thomas JE, Reagan TJ, Klass DW. Epilepsia partialis continua. A review of 32 cases. Arch Neurol 1977;34:266-75.  Back to cited text no. 1
    
2.Cockerell OC, Rothwell J, Thompson PD, Marsden CD, Shorvon SD. Clinical and physiological features of epilepsia partialis continua. Cases ascertained in the UK. Brain 1996;119:393-407.  Back to cited text no. 2
    
3.Sinha S, Satishchandra P. Epilepsia Partialis Continua over last 14 years: Experience from a tertiary care center from south India. Epilepsy Res 2007;74:55-9.  Back to cited text no. 3
    
4.Nayak D, Abraham M, Kesavadas C, Radhakrishnan K. Lingual epilepsia partialis continua in Rasmussen's encephalitis. Epileptic Disord 2006;8:114-7  Back to cited text no. 4
    
5.Vukadinovic Z, Hole MK, Markand ON, Matt BH, Sokol DK. Lingual epilepsia partialis continua in a girl. Epileptic Disord 2007;9:323-6.  Back to cited text no. 5
    
6.McGrath NM, Anderson NE, Hope JK, Croxson MC, Powell KF. Anterior opercular syndrome, caused by herpes simplex encephalitis. Neurology 1997;49:494-7.  Back to cited text no. 6
    
7.Almekhlafi MA, Couillard PL, Patry DG, Jetté N. Herpes encephalitis presenting with an opercular syndrome and epilepsia partialis continua. Neurologist 2010;16:208-10.  Back to cited text no. 7
    
8.De Kleermaeker FG, Bouwmans AE, Nicolai J, Klinkenberg S. Anterior opercular syndrome as a first presentation of herpes simplex encephalitis. J Child Neurol 2014;29:560-3.  Back to cited text no. 8
    
9.Adler AC, Kadimi S, Apaloo C, Marcu C. Herpes simplex encephalitis with two false-negative cerebrospinal fluid PCR tests and review of negative PCR results in the clinical setting. Case Rep Neurol 2011;3:172-8.  Back to cited text no. 9
    
10.Sili U, Kaya A, Mert A; HSV Encephalitis Study Group. Herpes simplex virus encephalitis: Clinical manifestations, diagnosis and outcome in 106 adult patients. J Clin Virol 2014;60:112-8.  Back to cited text no. 10
    


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