|Year : 2022 | Volume
| Issue : 5 | Page : 12-14
An interesting case of Parkinsonism in the young
Akula Sushmitha, Kamani Naresh Babu, Ashwitha Chawan, Y Sathyanarayana Raju
Department of General Medicine, Nizam's Institute of Medical Sciences, Hyderabad, Telangana, India
|Date of Submission||27-Aug-2020|
|Date of Decision||19-Oct-2020|
|Date of Acceptance||17-Jan-2021|
|Date of Web Publication||30-Aug-2022|
Y Sathyanarayana Raju
Professor, Department of General Medicine, Nizam's Institute of Medical Sciences, Panjagutta, Hyderabad 500 082, Telangana
Source of Support: None, Conflict of Interest: None
A 16-year-old female with no prior comorbidities came with chief complaints of fever and headache for 4 days associated with altered sensorium for the past 15 days in the form of decreased responsiveness. On general physical examination, Glasgow Coma Scale score was 6 with E1V1M4. Vitals were stable. Central nervous system examination revealed that bilateral pupils were sluggishly reactive to light. All extremities were rigid. A provisional diagnosis of viral encephalitis was made and was empirically started on acyclovir, doxycycline, ceftriaxone and vancomycin. Cerebrospinal fluid (CSF) showed lymphocytic pleocytosis, elevated proteins and normal glucose. CSF pan-neurotropic virus panel was negative. Serological testing for immunoglobulin M (IgM) against Japanese encephalitis virus positive. Magnetic resonance imaging of the brain showed symmetrical T2 hyperintense areas in the bilateral basal ganglia, thalamus and splenium of corpus callosum. The patient was started on oral levodopa plus carbidopa; sensorium became better and Parkinsonian features improved gradually.
Keywords: Basal ganglia, Japanese encephalitis, levodopa, Parkinsonism, rigidity
|How to cite this article:|
Sushmitha A, Babu KN, Chawan A, Raju Y S. An interesting case of Parkinsonism in the young. J Clin Sci Res 2022;11, Suppl S1:12-4
| Introduction|| |
Japanese encephalitis (JE) virus (JEV), a mosquito-borne flavivirus, is the most common cause of viral encephalitis in Asia. After an incubation period of 5–15 days, initial symptoms are usually non-specific and include fever, diarrhoea and rigors followed by headache, vomiting and generalised weakness., A very distinctive clinical presentation of JEV is a Parkinsonian syndrome resulting from extrapyramidal involvement; the findings include dull, flat, mask-like faces with unblinking eyes, tremor and cogwheel rigidity.
We present this case to highlight that parkinsonism can be a clinical manifestation in JEV infection if basal ganglia is involved.
| Case Report|| |
A 16-year-old female, student with no prior comorbidities, came with chief complaints of fever for 4 days, without chills, not associated with myalgias, arthralgias, rash, bleeding manifestations, associated with holocephalic headache dull-aching type, associated with altered sensorium for 15 days in the form of drowsy, not obeying commands and decreased responsiveness. On general physical examination, the patient was not responding to deep stimulus; Doll's eye movement was present. Central nervous system examination revealed Glasgow Coma score of 6 with E1V1M4. Bilateral pupils were sluggishly reactive to light. Both upper and lower limbs were rigid. Reflexes were normal. Terminal neck stiffness and Kernig's sign were negative. Other system examination was normal. A provisional diagnosis of viral encephalitis was made, and the patient was empirically started on acyclovir, doxycycline, ceftriaxone and vancomycin after collecting sample for blood culture. On laboratory testing the following were noted: haemoglobin 11.8 g/dL, total leucocyte count 12,900 cells/mm3 and platelet count 1.8 lakh/mm3. Liver and renal function tests were normal. Urinalysis was normal. Serological testing for human immunodeficiency virus, hepatitis B and C viruses was negative.
Smear and strip test for malarial parasite was negative. Dengue and scrub typhus immunoglobulin M (IgM) serological testing were negative. Antinuclear antibody was negative. Magnetic resonance imaging (MRI) of the brain plain and contrast showed symmetrical T2 hyperintense areas in the bilateral basal ganglia, thalamus and splenium of corpus callosum [Figure 1]. Lumbar puncture was done. Opening pressure of cerebrospinal fluid (CSF) was 8 cm H2O. CSF analysis showed glucose 58 mg/dL (serum glucose 98 mg/dL) and protein 120 mg/dL with white blood cells of 110/mm3 with 90% lymphocytes and 10% polymorphs. CSF smear for acid–fast bacilli was negative. CSF GeneXpert testing for Mycobacterium tuberculosis was negative. CSF adenosine deaminase was 5 U/L. CSF culture was sterile. CSF pan-neurotropic virus panel was negative. Serological testing for JE serum IgM (National Institute of Virology, Pune, kit) was positive. Electroencephalogram showed generalised slowing, possibility of encephalopathy. The patient was continued on acyclovir and rest other antibiotics were discontinued. The patient was started on oral levodopa plus carbidopa 100/25 half tablet thrice-daily. The patient's sensorium improved gradually over 2 weeks and was discharged in stable condition. At the time of discharge, bradykinesia and rigidity were present in all extremities and advised to follow-up on outpatient basis.
|Figure 1: Magnetic resonance imaging T2-weighted image showing hyperintense areas (arrows) in the bilateral basal ganglia|
Click here to view
After 1 month of treatment with levodopa and carbidopa, parkinsonism improved. Levodopa was gradually tapered.
| Discussion|| |
JE, a vector-borne viral disease, is endemic to large parts of Asia and the Pacific regions. JE is a major public health challenge due to its high-epidemic potential, high case–fatality and neuropsychiatric sequelae among survivors. JE was first recognised in Japan in 1924. In India, the first human case was reported from North Arcot district of Tamil Nadu in 1955. Until 1973, the disease was confined to southern parts of India, with low prevalence; subsequently, the disease spread to various other parts of India. The first outbreak of JE was recorded in 1973 from Burdwan and Bankura districts of West Bengal. In 1978, suspected outbreaks of JE were reported from 18 states, and 24 states and union territories have reported suspected JE cases till recent past.
The most common presenting neurological symptoms recorded were altered sensorium, seizures, headache and signs of meningeal irritation. A proportion of patients with JE have an acute flaccid paralysis that is easily mistaken for poliomyelitis.
JE presents as wide spectrum of movement disorders including hypokinesia tremors, rigidity and dystonia. A transient form of Parkinsonian syndrome characterised by varying severity of rigidity, hypokinesia, masking of face, tremors and hypophonia was observed in acute phase of illness after 1–4 weeks after disease onset. Reports of parkinsonism as a long-term sequelae of JE was also present in patients 3–5 years after acute JE infection with associated lesions in the substantia nigra observed on MRI. Brain computed tomography and MRI studies often reveal lesions in the thalamus, basal ganglia and midbrain. JE is diagnosed serologically by detection of JEV-specific immunoglobulin IgM antibodies in CSF or serum by an enzyme linked immunosorbent assay. Virus isolation or detection of viral ribonucleic acid (RNA) with nucleic acid amplification test (NAAT) can provide a definitive diagnosis, but positive results from CSF or blood are rare., Because humans have low levels of transient viremia and high levels of neutralising antibodies by the time distinctive clinical symptoms are recognised, virus isolation, and NAAT are insensitive for detection of JE viral in blood or CSF.
There is no specific antiviral treatment for JE. Treatment consists of supportive care with emphasis on control of intracranial pressure, maintenance of adequate cerebral perfusion pressure, seizures control and prevention of secondary complications. Some patients with JE may have lesions in the basal ganglia and presents with parkinsonism features as seen in our case which responded to levodopa.
Financial support and sponsorship
Conflicts of interest
Y Sathyanarayana Raju is an Editor of Journal of Clinical and Scientific Research. The article was subject to the journal's standard procedures, with peer review handled independently of these faculty and their research groups.
| References|| |
Halstead SB, Jacobson J. Japanese encephalitis. Adv Virus Res 2003;61:103-38.
Halstead SB, Jacobson J, Dubischar-Kastner K. Japanese Encephalitis Vaccines. Vaccines. Plotkin, SA, Orenstein, WA, Offit, PA, editors. 6th
ed. 2008. p. 312-51.
Solomon T, Dung NM, Kneen R, Gainsborough M, Vaughn DW, Khanh VT. Japanese encephalitis. J Neurol Neurosurg Psychiatry 2000;68:405-15.
Solomon T. Control of Japanese encephalitis–within our grasp? N Engl J Med 2006;355:869-71.
Webb JK, Pereira S. Clinical diagnosis of an arthropod borne type of virus encephalitis in children of north Arcot district, Madras state, India. Indian J Medi Sci 1956;10:573-81.
Chatterjee P. Japanese encephalitis outbreak in India. Lancet Neurol 2005;4:700.
Solomon T, Kneen R, Dung NM, Khanh VC, Thuy TT, Ha DQ, et al
. Poliomyelitis-like illness due to Japanese encephalitis virus. Lancet 1998;351:1094-7.
Misra UK, Kalita J. Prognosis of Japanese encephalitis patients with dystonia compared to those with parkinsonian features only. Postgrad Med J 2002;78:238-41.
Murgod UA, Muthane UB, Ravi V, Radhesh S, Desai A. Persistent movement disorders following Japanese encephalitis. Neurology 2001;57:2313-5.
Lin CY, Changlia SP, Huang CK, Lin MS, Yeh CH. Positive Tc-99m TRODAT findings in Japanese encephalitis. Clin Nucl Med 2007;32:484-5.
Solomon T, Dung NM, Kneen R, Thao le TT, Gainsborough M, Nisalak A, et al
. Seizures and raised intracranial pressure in Vietnamese patients with Japanese encephalitis. Brain 2002;125:1084-93.
Swami R, Ratho RK, Mishra B, Singh MP. Usefulness of RT-PCR for the diagnosis of Japanese encephalitis in clinical samples. Scand J Infect Dis 2008;40:815-20.
Burke DS, Lorsomrudee W, Leake CJ, Hoke CH, Nisalak A, Chongswasdi V, et al
. Fatal outcome in Japanese encephalitis. Am J Trop Med Hyg 1985;34:1203-10.
Tiroumourougane SV, Raghava P, Srinivasan S. Japanese viral encephalitis. Postgrad Med J 2002;78:205-15.