Poststroke emotionalism with dacrystic (Crying) episodes - making a case for risperidone.

Emotionalism is the abnormal expression of emotions like crying and laughing and could follow stroke, traumatic brain injury, multiple sclerosis and amyotrophic lateral sclerosis. Emotionalism has been known to respond therapeutically to different classes of drugs including tricyclic antidepressants like imipramine, Selective Serotonin Reuptake Inhibitors (SSRI) like sertraline and citalopram, anticonvulsants like lamotrigine, dopamine precursors like levodopa and NMDA receptor antagonists like dextromethorphan. Classical antipsychotics are hardly prescribed for emotionalism alone without psychotic features. In this case report, an eighty year old woman with a dominant fronto-temporal infarctive stroke with right faciohemiparesis presented with frequent crying (dacrystic) episodes after a month of onset of stroke and who did not satisfy DSM IV criteria for depression nor had other psychotic features. Serial trial of SSRIs and dextromethorphan/quinidine could not help until risperidone, an antipsychotic was introduced with resolution of crying episodes. The response to risperidone after trial of SSRIs and dextromethorphan/quinidine which are considered the gold standard for post-stroke emotionalism (PSE), could be another therapeutic dimension in the management of emotionalism in general and PSE in particular.

INTRODUCTION

Emotionalism is the incongruous manifestation of emotions usually as gelastic (laughing) or dacrystic (crying) or grimacing episodes or a combination of both.[12] It is also variously referred to as pseudobulbar affect or emotional incontinence or pathologic laughing and crying.[3] It is not as common as depression, with which it may be closely associated and the frequency ranges from 20% to 30% within the 1st year after a stroke.[14] Emotionalism could manifest after stroke, traumatic brain injury, multiple sclerosis, cerebral tumors, and amyotrophic lateral sclerosis; could be distressing and embarrassing to sufferers and their families; and often interferes with rehabilitation.[56]

Poststroke emotionalism (PSE) has been seen in both bilateral and unilateral strokes.[6] However, involvement of the dominant, usually left frontal and temporal lobes through the disruption of the neuronal substrate for emotions, has largely been linked.[17] The amygdala, a limbic brain structure, is central to emotional perception. It is known as the neuronal substrate that gives affective meaning to emotional stimulus, be it external or internal.[8] The amygdala robustly connects with the frontal cortex by the ventroamygdalofugal fibers, and the frontal cortex in turn influences the amygdala by giving a cognitive interpretation to the affective perception of emotions. The net effects of the input of the frontal cortex to the amygdala, particularly the orbitofrontal and premotor areas, are inhibitory in the expression of emotion. The frontal cortex via corticopontine fibers inputs brainstem motor nuclei involved in the expression of emotions such as facial, respiratory, and vocal muscles and glands. Also the temporal cortex, particularly the mesial temporal region together with the insula cortex input the amygdala with a net effect that is believed to be excitatory.[9] The amygdala also has afferent and efferent connections to the hippocampus to which it is apposed anatomically and it also connects directly via the stria terminalis to the hypothalamus. The hypothalamus in turn connects to brainstem structures like the periaqueductal gray which are key to the control of autonomic and neuroendocrine manifestation of emotions. Emotionalism, therefore, can be viewed as a limbic-motor disconnection syndrome, in which the faciovocal motor system is released from forebrain afferents carrying information of emotional content, modulated by the cerebellum.[9]

The neurotransmitters involved in the pathways mentioned above are different and include serotonin and glutamate which may form the basis for the response or otherwise to medications such as serotonin-specific reuptake inhibitors (SSRIs) (serotonin) and dextromethorphan/quinidine (glutamate), and serotonin polymorphism may explain the differences among individual in presentation and drug response.[10]

Here, we present a case of PSE with frequent crying episodes in an elderly woman with a dominant hemispheric stroke involving the frontotemporal cortex that persisted for several months until risperidone was introduced after a serial trial of SSRI and dextromethorphan/quinidine (Nuedexta®), a combination that is known as the gold standard of pharmacologic treatment of emotionalism.

CASE REPORT

An 80-year-old, right-handed woman presented in the neurology outpatient clinic with frequent crying episodes after suffering a left frontotemporal infarctive stroke a month earlier. Crying episodes were provoked anytime she attempted to express herself and were of sudden onset and terminated abruptly. Of note, there were no intervening laughing episodes and she was noticed to make unintelligible or jargon speeches in between her crying episode. She had normal sleep–wake pattern and her appetite was also normal. She had a history of hypertension and no history of diabetes mellitus or psychosis or traumatic brain injury. She was on amlodipine, atenolol, and aspirin.

She was chairbound and conscious with a modified Rankin score of 4, had right facial and hemiparesis of MRC 0 with spasticity of modified Ashworth scale of 3, and grade 3 deep tendon reflexes. There were no other craniopathies, had comprehensive aphasia with jargon speech of normal fluency, but unintelligible. She had frequent spells of crying that began when trying to express herself and terminated abruptly. Remarkably, she was calm and responsive with normal graces and affect soon after the crying episodes which were easily terminated when distracted. A left frontotemporal infarct was seen on cranial computed tomography scan; features of hypertensive heart disease with left ventricular hypertrophy and nonspecific ischemic changes on chest radiograph, echocardiography, and electrocardiography, respectively. She had mildly elevated low-density lipoprotein dyslipidemia, serum electrolytes, creatinine, and urea which were normal and her hemogram was satisfactory with a packed cell volume of 38% and normal white cells and platelets count. She did not satisfy the Diagnostic and Statistical Manual of Mental Disorders, Fourth Edition criteria for minor depression, and an impression of PSE was made. She was treated with sertraline 50 mg and later citalopram 10 mg for several weeks without any appreciable improvement and was later administered with a combination of dextromethorphan and quinidine (20/10 mg) for another 1 month with marginal improvement, but the crying episodes persisted for the most part. She was however noticed to have become calm following a trial of 0.5–1 mg of risperidone without dacrystic episodes.

DISCUSSION

The dacrystic episode started within a month of suffering a stroke in this patient which is in consonance with the natural history of the onset of PSE, and usually it begins within the 1st month to 1 year of the preceding vascular event.[6] She had more than one risk factor for a stroke. Her age and heart condition and dyslipidemia were risk factors for a vascular event. Remarkably, she did not satisfy the criteria for the diagnosis of depression, though it is a differential diagnosis. Her poor response to antidepressants is noteworthy as it suggests the absence of depression. The response to conventional treatment like SSRI such as sertraline was poor and the introduction of the relatively expensive dextromethorphan/quinidine which is presently the gold standard for PSE made no remarkable difference. PSE is known to respond with remission and cessation of crying and/or laughing episodes to SSRIs, tricyclic antidepressants, lamotrigine, and a combination of dextromethorphan/quinidine, but this was not the case in this patient.[10]

The complete cessation of crying episodes with the introduction of risperidone, an atypical antipsychotic medication, is the stand-out feature here and in our opinion may convey a novel message on emotionalism generally and PSE in particular.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.