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CASE REPORT |
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Year : 2014 | Volume
: 7
| Issue : 6 | Page : 766-768 |
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Case report of patient with orolingual tremors as post Japanese encephalitis sequelae
Anirudda Deshpande1, Supriya Khardenavis2, Aakash Shetty1
1 Department of Neurology, Kasturba Medical College, Manipal, Karnataka, India 2 Department of Ophthalmology, JJM Medical College, Davanagere, Karnataka, India
Date of Web Publication | 18-Nov-2014 |
Correspondence Address: Anirudda Deshpande Department of Neurology, Kasturba Medical College, Manipal - 576 104, Karnataka India
Source of Support: None, Conflict of Interest: None | Check |
DOI: 10.4103/0975-2870.144874
Japanese encephalitis (JE), mosquito-born flavivirus, is a common human viral encephalitis in the world occurring in epidemics throughout Asia. Among the various forms of movement disorders associated with JE, generalized/hemi-dystonia and Parkinsonism are well-documented movement disorders in patients, post JE. As per the available literature, patients with a movement disorder as long-term sequelae have definite lesions on magnetic resonance imaging. Patients present with muteness, generalized dystonia and are associated with lesions in both thalami. Our patient with characteristic orolingual tremors had bilateral thalamic hyperintensities, as is characteristically seen in JE. To the best of our knowledge, orolingual tremors as a manifestation in patient with post JE sequelae, is extremely rare and yet to be reported. However, there is no report of orolingual tremor as post JE sequelae to the best of our knowledge. Keywords: Japanese encephalitis (JE); orolingual tremors; post JE movement disorders
How to cite this article: Deshpande A, Khardenavis S, Shetty A. Case report of patient with orolingual tremors as post Japanese encephalitis sequelae. Med J DY Patil Univ 2014;7:766-8 |
Introduction | | |
Although movement disorders such as generalized/hemi-dystonias, Parkinsonism More Details and other forms of akinetic rigid syndromes are reported in the literature, it is uncommon to find orolingual tremors in patients who have recovered from Japanese encephalitis (JE).
Abnormal magnetic resonance imaging (MRI) brain and its correlation with patients having movement disorders as post-JE sequelae seem to demonstrate bilateral thalamic hyper intensities on T2W/fluid attenuated inversion recovery (FLAIR). Interestingly, our patient also has similar MRI brain finding.
The rarity of such peculiar movement disorder as a manifestation of post JE sequelae has prompted us to report this case with clinical video, MRI of the brain. To the best of our knowledge, this is the first case report mentioning orolingual tremors as post JE sequelae.
Case Report | | |
The present case report is about a 65-year-old female, farmer by occupation, presented with history of fever, altered sensorium and vomiting of 2 days duration. Neurological examination revealed Glasgow Coma Scale score of 12 on admission. The patient had signs of meningeal irritation including Kernig's and neck stiffness. She also had bilateral extensor plantars. No other significant contributory history. Other conditions giving rise febrile encephalopathy such as dengue encephalopathy, cerebral malaria, aseptic meningoencephalitis (viral or bacterial) were considered; however, further investigations in the form of JE immunoglobin M enzyme linked immunosorbent assay (ELISA) which demonstrated significant high titers, MRI brain showing characteristic bilateral thalamic hyper-intensities in T2W, FLAIR, [Figure 1] and [Figure 2]. Cerebrospinal fluid (CSF) analysis showed lymphocytosis with normal sugars, confirming the diagnosis of JE. Although, there were no clinically obvious seizures noticed electro encephalogram was done which showed bi-hemispherical generalized delta activity, which are considered non-epileptiform in nature. Patient was intubated and ventilated to secure the airway and prevent aspiration. There was no hypoxia or drop in oxygen saturation at any point of time. Supportive therapy including intravenous fluids, anti-edema measures (mannitol, furosemide) was started. She was gradually weaned off a week after her admission.
She was started on levodopa carbidopa combination, trihexyphenidyl for her orolingual tremors and parkinsonian features with which she was symptomatically better. Patient recovered from JE and was discharged 2 weeks after admission on multivitamin tablets.
Patient developed orolingual tremors, parkinsonian symptoms 3 months after discharge.
Discussion | | |
In orolingual tremors, the parts of the orolingual region potentially affected by tremor in isolation, or in various combinations include the jaw, tongue, face and pharynx. The tremor may occur either at "rest" or with activation of the orolingual structures.
It may be postulated that loss of dopaminergic neurons was possibly considerable, the surviving dopaminergic nigral neurons being unable to compensate sufficiently and is postulated to give rise to orolingual tremors.
Orolingual tremors associated with dystonia, parkinsonian orolingual tremor, palatal tremor with orolingual tremor, drug induced orolingual tremor and psychogenic orolingual tremors are some of the other causes for such a presentation. However, since, there was presence of preceding JE, a known cause of extrapyramidal manifestations they did not satisfy the exclusion criteria as per Silverdale et al. [1]
Murgod et al. noted that patients with a movement disorder as long-term sequelae have definite lesions on MRI. Both the children mentioned in their study, were mute and had generalized dystonia associated with lesions in both thalami. One child had a lesion in the midbrain, whereas the other child had a lesion in the pons. The two patients with parkinsonism had lesions confined to the substantia nigra with a normal striatum.
Among the survivors of JE, 60% had no residual neurologic deficits; movement disorders, present in 26.7% of patients, were the most common neurologic sequelae. The four patients with persistent movement disorders were followed for a mean period of 4.7 years. Two patients with parkinsonism had associated lesions in the substantia nigra. [2]
Among the various sensitive investigational modalities to diagnose JE (apart from MRI brain) the commercial ELISA for the diagnosis of JE virus infections showed a sensitivity of 88% with sera and 81% with CSF. [3]
The clinical Videos 1-3 of the patient demonstrates rhythmical, jaw, oral, lingual tremor with parkinsonian features in the form of bradykinesia, asymmetrical decreased arm swing of the left upper limb and bilateral upper limb distal dystonic posturing.
[Figure 1],[Figure 2] and [Figure 3] show MRI Brain with T2W, DWI and FLAIR sequences, respectively suggestive of bilateral symmetrical thalamic hyper intensities. | Figure 1: T2W magnetic resonance imaging brain sequence showing bilateral, symmetrical hyperintensities in thalami, seen commonly in Japanese encephalitis
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| Figure 2: Diffusion weighted magnetic resonance imaging sequence showing bilateral, symmetrical hyperintensities in thalami, seen commonly in Japanese encephalitis
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| Figure 3: Fluid attenuated inversion recovery (FLAIR) magnetic resonance imaging sequence showing bilateral, symmetrical hyperintensities in thalami, seen commonly in Japanese encephalitis
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Acknowledgement | | |
Dr. Suryanarayan Rao, Professor and Head, Department of Neurology, Kasturba Medical College, Manipal - 576 104, Karnataka, India.
References | | |
1. | Silverdale MA, Schneider SA, Bhatia KP, Lang AE. The spectrum of orolingual tremor - A proposed classification system. Mov Disord 2008;23:159-67. |
2. | Murgod UA, Muthane UB, Ravi V, Radhesh S, Desai A. Persistent movement disorders following Japanese encephalitis. Neurology 2001;57:2313-5. |
3. | Cuzzubbo AJ, Endy TP, Vaughn DW, Solomon T, Nisalak A, Kalayanarooj S, et al. Evaluation of a new commercially available immunoglobulin M capture enzyme-linked immunosorbent assay for diagnosis of Japanese encephalitis infections. J Clin Microbiol 1999;37:3738-41. |
[Figure 1], [Figure 2], [Figure 3]
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