Epilepsy & Behavior 20 (2011) 395–397

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Case Report

Epilepsia partialis continua as a manifestation of dengue encephalitis

Rajesh Verma a,⁎, Aravinthan Varatharaj b

a Department of Neurology, Chhatrapati Sahuji Maharaj Medical University, Lucknow, UP, Indiab Department of Neuropathology, Pembroke College, Oxford, UK

⁎ Corresponding author. Department of NeurologyMedical University, Lucknow, UP, India. Fax: +91 522 2

E-mail address: drrajeshverma32@yahoo.com (R. Ve

1525-5050/$ – see front matter © 2010 Elsevier Inc. Aldoi:10.1016/j.yebeh.2010.12.003

a b s t r a c t

a r t i c l e i n f o

Article history:Received 28 October 2010Revised 30 November 2010Accepted 1 December 2010Available online 7 January 2011

Keywords:Dengue feverEpilepsia partialis continuaEncephalitis

Dengue fever is becoming a great public health problem leading to significant morbidity and mortality,particularly in developing countries. In the past few years, various neurological complications have beenreported globally. Dengue encephalitis as a clinical presentation of dengue fever is now gaining recognition inthe scientific community. It is characterized by fever, headache, reduced consciousness, and generalizedseizures. Epilepsia partialis continua, a type of focal status epilepticus, is caused by multiple clinical disorders,including viral encephalitis. We describe a young woman who manifested epilepsia partialis continua causedby dengue encephalitis.

, Chhatrapati Sahuji Maharaj257090.rma).

l rights reserved.

© 2010 Elsevier Inc. All rights reserved.

1. Introduction

Dengue viral infections pose a significant and increasing problemto global health [1]. Neurological manifestations of dengue infectionare well reported and include typical viral encephalitis with fever,headache, and reduced consciousness. Seizures are common and aretypically generalized [2]. Described here is a case of dengue encepha-litis in Lucknow, India, in which a manifestation of the illness wasepilepsia partialis continua. To the best of our knowledge such anassociation has not been previously reported.

2. Case report

A 20-year-old right-handed woman presented with a 4-weekhistory of fever, headache, and reduced consciousness. The onset ofthe febrile illness was accompanied by severe myalgia and retro-orbital pain. Relatives also stated that the patient had displayedstiffening and paucity of movement on the left side of the body for thelast 15 days. There was no history of preceding rash, icterus,arthralgia, coryza, or cough, and no suggestion of trauma, metabolicdisorders, or tuberculosis. There was no past medical history of note.

Observations on admissionwere a blood pressure of 116/78 mmHg,pulse of 92/minute, and temperature of 38.9 °C. Consciousness wasreduced and the Glasgow coma score was E1, V2, M5. Cardiovascular,respiratory, and abdominal examinations were unremarkable. Cranialnerve examination revealed an upper motor neuron type of facial palsyon the left side. The rest of the cranial nerve evaluation wasunremarkable, with pupils equal and reactive to light. Upper and

lower limb examination revealed lead-pipe rigidity on the left side andspasticity on the right side. Deep tendon reflexeswere brisk throughout.Plantars were equivocal. During the course of the illness, the patientdeveloped persistent clonic flexion jerks of the fingers of the left handlasting hours and intermittently interrupted bygeneralized tonic–clonicseizures. These abnormal movements were diagnosed clinically asepilepsia partialis continua (EPC).

Laboratory investigations including full blood count, urea, creat-inine, and electrolytes were all within normal limits, apart from amildanemia (hemoglobin 10 g/dL, normal 11.5–16 g/dL). Liver and thyroidfunction tests were unremarkable, and serum creatine kinase wasnormal. Serological assays for HIV, hepatitis B, hepatitis C, cytomeg-alovirus, and Epstein–Barr virus were normal. Serum copper andceruloplasmin were within the normal ranges, and serum autoanti-bodies including antinuclear (ANA), anticardiolipin (ACL), and anti-neutrophil cytoplasmic (ANCA) antibodies were negative. Cerebro-spinal fluid (CSF) analysis yielded 20 cells/μL, all lymphocytes (normalb5 cells), protein 0.26 g/L (0.1–0.45 g/L), and slightly low glucose2.05 mmol/L (normally more than 50% of serum glucose). CSF exami-nation showed no organisms with Gram's stain, India ink preparation,or stains for acid-fast bacilli, and polymerase chain reaction (PCR)assay of the CSF was negative for both herpes simplex and Japaneseencephalitis viruses. Results for IgM antibody titer for Japaneseencephalitis in CSF were negative. Anti-dengue immunoglobulinM (IgM) in paired serum samples taken 1 week apart was positiveand showed rising titers. CSF anti-dengue IgM was also positive.Electroencephalography in an anteroposterior bipolar montageshowed focal spikes and phase reversal in the right temporal area(Fig. 1). Contrast-enhanced magnetic resonance images of the brainwere normal (Fig. 2).

A diagnosis of dengue encephalitis was made and the patient wasmanaged supportively in an intensive care setting. Seizure activity did

Fig. 1. Electroencephalogram of the patient showing focal spikes (thin arrow) and phase reversal in the right temporal area (thick arrow).

396 R. Verma, A. Varatharaj / Epilepsy & Behavior 20 (2011) 395–397

not respond to intravenous lorazepam, phenytoin, or phenobarbital,but was suppressed with intravenous midazolam. Unfortunately, thepatient died during the course of treatment.

Fig. 2. Image obtained with T2-weighted MRI of the cranium showing normal results.

3. Discussion

Epilepsia partialis continua is a rare focal motor form of statusepilepticus [3].. The clinical picture comprises spontaneous muscletwitching and clonic movements confined to a particular part of thebody, whereas the neurophysiological correlate is prolonged seizureactivity in the corresponding area of the contralateral primary motorcortex (although subcortical structures may also be involved). Thehand is most commonly affected because of its relatively large corticalrepresentation. Typically there is also a hemiparesis on the side of theaffected part, reflecting the cortical origin of the phenomenon [4]. EPCmay be difficult to distinguish from tremor, although in our patientthe abnormal movements did not have the rhythmical, oscillatoryquality of tremor. In addition, the periodic conversion to secondarilygeneralized seizures and the persistent epileptiform activity identifiedin the right temporal region were suggestive of a diagnosis of EPC.

Numerous causes of EPC have been described, including vascular,neoplastic, and metabolic pathologies. Encephalitis is a commoncause, and Rasmussen's encephalitis is the prototypical cause ofchildhood EPC [5]. Viral encephalitis is also a well-recognized cause[6]. In our patient a wide range of differential diagnoses were con-sidered and systematically excluded. The history, examination, andimaging findings were inconsistent with Rasmussen's encephalitis,vascular disorders, or intracranial neoplasia. Screening for a range of

397R. Verma, A. Varatharaj / Epilepsy & Behavior 20 (2011) 395–397

common central nervous system (CNS) pathogens, including herpessimplex virus, Japanese encephalitis virus, and Cryptococcus, wasnegative, as were tests for various metabolic encephalopathies (e.g.,hepatic encephalopathy, Wilson's disease).

In our patient the clinical features of fever, headache, reducedconsciousness, and seizures suggested encephalitis. The rising serumantibody titers confirmed dengue infection, and the CSF lymphocytecount and anti-dengue IgM positivity suggested dengue viral infil-tration of the CNS. Exclusion of liver failure, shock, electrolyteabnormalities, and intracranial hemorrhage led us to conclude thatthe neurological manifestations were not attributable to thesecomplications of severe dengue infection, but instead were causedby true dengue encephalitis.

Seizures are a typical feature of viral encephalitis and occur in47% of dengue encephalitis cases (previously published data from34 cases). In the vast majority of these cases, seizures were gener-alized [2,7]. EPC is a rare type of seizure activity and has beenassociatedwith other viral encephalitides, but has not previously beenreported in dengue encephalitis. Many viral encephalitides display atropism for particular brain structures, and though there are few datato suggest such specificity in dengue encephalitis, many casesreported in the literature feature focal involvement on brain imaging.In our patient, neurological examination and brain imaging were notlocalizing, although the presence of EPC raises the possibility of focalpathology in the primary motor area.

One differential diagnosis deserves special mention: Could this beJapanese encephalitis (JE)? The history suggested bradykinesia in theleft hemibody, and examination revealed lead-pipe rigidity, bothsuggestive of the basal ganglia involvement that is typical of JE [8].Though EPC has not been reported in JE, we considered either thata new association was possible or that we had misdiagnosed EPCwhen in fact the abnormal jerks were another movement disorder.However, for the reasons stated earlier we were confident of thediagnoses of EPC and dengue encephalitis. In excluding JE we placedconsiderable weight on the negative CSF IgM and PCR results and the

fact that the positive serum and CSF dengue antibodies provided aplausible alternative diagnosis.

4. Conclusions

Across theworld, 2.5 billion people live in areas in which dengue isendemic. Encephalitis is a rare but important complication of dengueinfection. Regardless of the presence or absence of classic featuressuch as myalgia and retro-orbital pain, in endemic areas, it is worth-while to consider dengue in any patient presenting with an encepha-litic picture. Fever, headache, and reduced consciousness are the corefeatures, although a wide range of neurological manifestations havebeen reported in the literature. Seizures are common and typicallygeneralized. This case report adds EPC to the list of neurologicalmanifestations of dengue encephalitis.

References

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