|Year : 2015 | Volume
| Issue : 4 | Page : 159-162
Unfolding the mystery: Rare presentation of Japanese encephalitis as catatonia
Nimisha Doval1, Sujita Kumar Kar1, Hardeep Singh Malhotra2
1 Departments of Psychiatry, King George's Medical University, Lucknow, Uttar Pradesh, India
2 Department of Neurology, King George's Medical University, Lucknow, Uttar Pradesh, India
|Date of Web Publication||19-Oct-2015|
Sujita Kumar Kar
Department of Psychiatry, King George's Medical University, Lucknow - 226 003, Uttar Pradesh
Source of Support: None, Conflict of Interest: None
| Abstract|| |
Catatonia is a syndrome of specific motor abnormalities, closely associated with disorders of mood, thought, and cognition. The principal symptoms of catatonia are mutism, immobility, negativism, posturing, stereotypy, and echo phenomena. Catatonia occurs in various psychiatric illnesses as well as medical disorders like infections of the central nervous system such as encephalitis, autoimmune disorders, cerebrovascular events, systemic metabolic disturbances, and toxic drug states. Catatonia may often mislead the clinician and the patient may be misdiagnosed as primarily suffering from a psychiatric disorder and be treated accordingly. Encephalitis may present with various psychiatric symptoms including catatonic features as its sequel that may sometimes be misunderstood as a primary psychiatric disorder. We highlight a case of a young female in her postpartum period, who presented with initial aggressive behavior and sleep deprivation followed by features of catatonia accompanied with fever. She initially responded well to benzodiazepines; however, her subsequent response was poor. When she was subjected to neuroimaging, it revealed signal intensity alteration in the bilateral basal ganglia and temporal regions suggestive of encephalitis; cerebrospinal fluid (CSF) viral markers examination revealed immunoglobulin M (IgM) positivity for Japanese encephalitis (JE). This case report highlights the diagnostic dilemma and management issues of a case of JE with catatonic symptoms.
Keywords: Benzodiazepines, catatonia, Japanese encephalitis
|How to cite this article:|
Doval N, Kar SK, Malhotra HS. Unfolding the mystery: Rare presentation of Japanese encephalitis as catatonia. Int J Nutr Pharmacol Neurol Dis 2015;5:159-62
|How to cite this URL:|
Doval N, Kar SK, Malhotra HS. Unfolding the mystery: Rare presentation of Japanese encephalitis as catatonia. Int J Nutr Pharmacol Neurol Dis [serial online] 2015 [cited 2021 Sep 17];5:159-62. Available from: https://www.ijnpnd.com/text.asp?2015/5/4/159/167499
| Introduction|| |
Encephalitis is a state of inflammation of the brain parenchyma, commonly resulting from either an infective cause or when the body's own immune system mistakenly attacks the brain tissue., The clinical presentations of acute encephalitis can be in the form of fever, altered sensorium, headache, disturbance of speech, seizure, behavioral abnormalities, and executive dysfunction, ranging from focal to diffuse neurological deficits.
Psychiatric symptoms, in conjunction with neurological features, are commonly seen in the acute phase as well as following the recovery of viral encephalitis. At times, the neuropsychiatric symptoms may be the only presenting feature in these patients., A review reported that psychosis was present in 35% cases and catatonic symptoms were present in 33% cases, leading to misdiagnosis and improper treatment with added risk of side effects of neuroleptics.Herpes simplex virus is the most commonly identified cause of viral encephalitis, simulating a primary psychiatric disorder. One of the main vaccine preventable diseases in Asia and the Western Pacific is Japanese encephalitis (JE). The risk of JE varies among the immigrants in Asia, depending on the destination, duration of travel, season, and activities. Most human infections are asymptomatic or result in only mild symptoms. However, a small percentage of infected persons develop inflammation of the brain (encephalitis), with symptoms such as sudden onset of headache, high fever, disorientation, seizure, tremors, dystonia, Parkinsonian features, and coma. Approximately 25% of the JE cases are fatal. Neuroimaging analysis of patients with JE shows classical changes in the thalamus, basal ganglia, temporal lobes, and midbrain. The presence of dystonia in patients with JE may be suggestive of thalamic involvement, while basal ganglia involvement results in development of Parkinsonian features. Thalamofrontal disconnection or frontal dysfunction presents with impairment of executive functions and perseveration. Patients with JE can present with neuropsychiatric symptoms like cognitive impairment, affective instability, behavioral problems, obsessive compulsive features, and personality changes. Antipsychotics, mood stabilizers, and electroconvulsive therapy may improve the psychiatric symptoms.
| Case Report|| |
A 32-year-old lady presented with the chief complaints of mutism, holding of food in the mouth, prolonged staring looks, psychomotor slowing, and incontinence for 15 days prior to psychiatric consultation at our hospital. Her history was suggestive of childbirth 2½ months back, which had been preceded by fever for 2 days. Fever continued for 5-7 days following childbirth. After childbirth, the patient developed altered sensorium and had multiple episodes of seizures. She was also diagnosed with meningoencephalitis and treated with injectable medications. The patient's condition improved but her altered behavior persisted in the form of muttering to self, aggressive outbursts, lack of concern for her baby, and inappropriate smiling. Over a period of 1 month, the symptoms got aggravated and the patient developed marked aggressive-disruptive behavior along with increased physical activity. She was becoming unmanageable at home. On initial consultation, she was started on oral antipsychotics but upon no improvement within 10 days, she was subsequently given parenteral haloperidol (10 mg) and promethazine (50 mg) twice a day for 5 consecutive days. With these medications, her aggression was controlled; however, in the next 2 weeks, her oral intake and verbal output reduced. She had started holding food and water in her mouth and at times, actively resisted efforts to chew food. With the aforesaid complaints, the patient was referred to our center for further management; the lady was hospitalized. On examination, she had rigidity in all four limbs. Her clinical evaluation revealed mutism and negativism. A provisional diagnosis of organic brain syndrome (catatonia as a postencephalitic sequel) was kept with differential diagnoses of catatonia as a part of postpartum psychosis, and catatonia as a sequel of cerebral vein thrombosis of the deep venous system. The patient was initiated on intravenous lorazepam (4 mg) to which she responded well, and she started speaking and accepting oral feeding. In view of catatonia as postencephalitic sequel, in the initial 2 days, lorazepam (6 mg) in three divided doses was given. Initial response to benzodiazepine was good but subsequently the patient ceased to respond. She continued to hold food in the mouth, along with mutism and negativism. Occasional episodes of crying and anger outbursts were also noted.
Magnetic resonance imaging (MRI) of the brain was done [Figure 1] and [Figure 2], which revealed signal intensity alteration in the bilateral basal ganglia, thalamus, and temporal regions suggestive of encephalitis. Differential leukocyte count revealed 80% lymphocytes, 10% neutrophils, and 10% monocytes; however, the total leukocyte count was in the normal range. Cerebrospinal fluid (CSF) viral markers examination (routine biochemical, microscopic, and viral markers) was done. CSF viral markers examination revealed immunoglobulin M (IgM) positivity for JE. The patient was referred to the Department of Neurology and was hospitalized there, where she was started on oral amantadine (200 mg/day) in divided doses and lorazepam was gradually tapered off. Subsequently, escitalopram 10 mg/day was added. There was an improvement in the patient's oral intake, verbal output, rigidity, and emotional dysregulation over the next 2 weeks. After 3 months, the patient was showing progressive improvement in symptoms and was independent in her activities of daily living.
|Figure 1: MRI of the brain showing signal intensity alteration in the bilateral basal ganglia, thalamus, and temporal regions suggestive of encephalitis|
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|Figure 2: MRI of Brain (Coronal Section) showing signal alteration in bilateral thalamus|
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| Discussion|| |
This case presents a diagnostic dilemma. The patient presented with symptoms of catatonia following encephalitis that was considered as an encephalitic sequel. The patient showed good response to intravenous benzodiazepine (lorazepam); however, she ceased to respond subsequently and so benzodiazepine was withdrawn. The MRI of the brain was suggestive of encephalitis and IgM antibody assessment in CSF was found to be positive for JE, considered to be diagnostic of JE. Autoimmune encephalitis is another common form of encephalitis, characterized by neurobehavioral as well as emotional disturbances and motor disturbances. Autoimmune encephalitis can be an important differential diagnosis; however its possibility was unlikely in presence of positive CSF viral marker for JE, as seen in our case.
Disruption of several frontal subcortical circuits have been found to be associated with behavioral and personality changes. It has been seen that persons with impaired motivation, akinetic mutism, and apathy have involvement of the anterior cingulate circuit. Our patient had symptoms suggestive of dysexecutive syndrome characterized by akinesia, mutism, rigidity, and incapability to perform daily chores, and altered signal intensity was apparent in the basal ganglia, thalamus, and temporal lobe. It is probable that in our case, there was disruption of the anterior cingulate circuit that led to impaired motivation, akinetic mutism, and apathy.
"Akinetic mutism" is a medical term describing patients tending to neither move (akinesia) nor speak (mutism). It most often appears in two different forms: Frontal and mesencephalic. Akinetic mutism can occur in the frontal region of the brain and occurs because of bilateral frontal lobe damage. This state of akinetic mutism varies in intensity but it is distinguished by drowsiness, lack of motivation, hypersomnolence, and reduction in spontaneous verbal and motor actions. The symptoms progress over time and are characterized by lack of motor function, lack of speech, apathy, slowness, and disinhibition. Our rationale behind giving amantadine was its neuroprotective effects, ability to correct dopamine dysregulation at the subcortical level and improve motor slowing, and its function as an antioxidant. Our rationale of giving escitalopram was to improve the serotonin imbalance at prefrontal cortex, thus reducing apathy and increasing volition.
Akinetic mutism can be misdiagnosed as catatonia. A careful evaluation of the evolution of symptoms over time is required to arrive at the correct diagnosis. Catatonia, akinetic mutism, and many more entities lie in the interface of neurology and psychiatry; hence, a collaborative approach is highly essential to make a diagnosis and provide an appropriate treatment.
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[Figure 1], [Figure 2]