POST STROKE LAUGHTER A CASE REPORT. Amarpreet Kaur*, Nor Zuraida Z*, Ng CG*, Aida SA*

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1 CASE REPORT POST STROKE LAUGHTER A CASE REPORT Amarpreet Kaur*, Nor Zuraida Z*, Ng CG*, Aida SA* *Department of Psychological Medicine, Faculty of Medicine,.. University of Malaya.. ABSTRACT Pathological laughing or crying (PLC) were recognized after the occurrence of stroke, with a prevalence of 15% to 18%. There is no apparent triggering stimulus, and is often misdiagnosed as a mood disorder as it is a disorder of emotional expression rather than a primary disturbance of feeling. We reported a case of a 32 year old lady, who presented with giddiness and altered consciousness progressing to fever and neck stiffness, who s CT showed a massive left cerebellar infarct. No risk factors were identified. Psychiatrically, she developed sudden crying spells after one month and a diagnosis of Major Depressive Disorder was made with subsequent commencement of anti-depressants. A week later, she developed continuous inappropriate laughter without the feeling of elation, which was beyond her control. There were no symptoms of mania or psychosis. Keyword: stroke, Post stroke depression, post stroke laughter Introduction The post-stroke patient is at significant risk for various psychiatric syndromes. The most commonly reported of these in the literature are Post-stroke Depression (PSD) and Post-stroke Dementia (PSDem) (1). Integrating assessment for psychiatric symptoms into the care of post-stroke patients is especially critical in the first 6 months following a stroke, a period of high risk for psychiatric complications. Psychiatric and substance abuse history, past treatment with psychopharmacologic agents, family psychiatric history, and personal and family history of suicidal behavior are important items to bear in mind. Careful attention to caregivers' and family members' behavioral observations is necessary, especially in patients with cognitive impairment or other neurologic barriers to communication, such as residual aphasia. Other Post-stroke Psychiatric Syndromes less frequently seen include pathologic crying, pathologic laughter, apathy, and isolated fatigue (1). These are coded as mental disorder due to a general medical

2 condition not otherwise specified. Pathologic laughter and crying are sometimes grouped as pathologic emotions (PE) with sudden paroxysms of either laughter or crying, irrespective of the ambient mood state. Post stroke PE is a distressing and socially disabling problem. It affects 16 to 29% of all stroke survivors. (2) PE can be triggered by non-specific stimuli or by a low-threshold emotive stimulus. Curiously, the PE do not themselves induce a mood change other than during the affective display, and they are not under voluntary control. Some literature recommends the use of antidepressants for PE; lithium and anticonvulsants are alternatives. Antidepressants, particularly selective serotonin reuptake inhibitors (SSRls), have been increasingly recognized as the treatment of choice for pathologic crying (PC) (2). However, little is known about etiologies and other treatment options for various clinical manifestations of PLC. This case report illustrates a case of post stroke laughter, highlighting the journey that brought her there. Case This is a case of a 32 year old Indian lady with no past medical history, who presented with a sudden onset of giddiness, followed by altered consciousness for one day. She was brought immediately to Kajang Hospital, where she developed a fever and neck stiffness, conscious level dropping to Glasgow Coma Scale of 7/15. A computed tomography (CT) scan of the brain done to investigate and elicit causal factor of illness, showed a massive left cerebellar infarct. She was treated as having meningitis and given a course of antibiotics. She was intubated for cerebral protection. Cerebral resuscitation was commenced and completed after 48 hours. She was transferred to University Malaya Medical Centre (UMMC) for further management of her condition. In UMMC, physical examination showed evidence of tetraparesis. Supported ventilation continued. Unfortunately she developed sepsis secondary to a nosocomial infection. A CTV brain scan was done and a conclusion of an extensive cerebellar and brainstem infarct with no evidence of venous sinus thrombosis was made. Subsequently, a tracheostomy was done and she was weaned off from supported ventilation the next day. The patient had dysarthria and difficulty communicating with others. While in the ward, screening for possible factors that could have precipitated the infarct, such as connective tissue disease and a thrombophilic screen were done, however results were negative. The patient was reviewed by the psychiatric team a month later, after the primary treating team noted that she had sudden crying spells. Based on the history and presentation gathered, a diagnosis of Major Depressive Disorder was made and she was started on Escitalopram 10mg daily. With subsequent observations, a week later she was then noted to have labile mood, characterized by crying spells as well as bouts of laughter. In the meantime, her communication had improved as she was now able to write on an alphabet board to convey information. She was discharged from ward after having been admitted for two months.

3 Subsequent reviews in the psychiatric clinic showed that her mood had improved. Her antidepressant was stopped. However, her family members noted that the crying spells recurred. Antidepressant medication was commenced again three months later. Upon review from December 2007 onwards, the patient had continuous inappropriate laughter. She was not able to explain why she was laughing. She denied feeling elated but felt that her laughter was beyond her control. There were no other accompanying symptoms to suggest mania or hypomania, nor were there any psychotic symptoms. Discussion Sudden, uncontrollable episodes of emotional display or pathological laughing or crying (PLC) were recognized after the occurrence of a stroke.3,4 The prevalence of this morbid condition has been reported to range from 15% to 18%.5,6,7 The episodes either do not have an apparent motivating stimulus or are triggered by a stimulus that would not have elicited such an emotional response before the onset of their neurological disorder. Various terms have been used to describe this condition. These terms are pseudobulbar affect, pathological laughter and crying, emotional lability, emotionalism, emotional dysregulation, forced crying, involuntary crying, pathological emotionality and emotional incontinence.8 The authors use PLC in this report as we believe is a precise descriptive term for the condition. In clinical setting, PLC is often unrecognized. The condition can be often be misdiagnosed as a mood disorder. Patients with PLC exhibit the emotional display in the absence of a pervasive and sustained depressed or elated mood. PLC occurs only paroxysmally and is uncontrollable and involuntarily. Such episodes may even occur in the absence of any congruent changes in the mood of the patient.11 Three primary features were emphasized by Poeck: 1) sudden loss of voluntary emotional control; 2) occurrence in response to non specific, often inconsequential stimuli; and 3) lack of clear association with prevailing mood state.20 Several scales are available to identify and characterize PLC. One of them is Pathological Laughter and Crying Scale.21 It is commonly used in clinical research. PLC is a disorder of emotional expression rather than a primary disturbance of feeling. The laughter or crying behaviours (e.g. the facial expression, the tears etc) in PLC is identical with regular laughter or crying but no associated feeling of happiness or sadness. In the past, it was proposed that the impaired emotional regulation resulted from disinhibition of a presumed brainstem center for laughing and crying due to lesions of the voluntary motor pathways in the descending corticobulbar pathways.9,11however, this explanation has several limitations like patients do sometimes response contradictory to the emotional valence of the triggering stimulus, lack of typical features of pseudobulbar palsy and no clear evidence of single brainstem center for laughing and crying.11 Based on current studies, other structures like prefrontal cortices9, cerebellum12 and globus pallidus13 are suggested to be associated with PLC. A thorough understanding of the pathophysiology of PLC is needed.

4 Although there is lack of understanding regarding the exact etiologies of PLC, the condition was found improved markedly with the administration of antidepressants14 like sertraline15, fluoxetine16, amitriptyline17, nortriptyline18 and citalopram.19 The possible explanation for the beneficial affect of selective serotonin reuptake inhibitor (SSRI) and other antidepressants would be that by altering the operation of higher order cortical areas involved in cognitive processing, the drugs would alter the cognitive context enough to raise the threshold at which stimuli engage the system in PLC, reducing, in short, emotional lability.11 It is important to note that even though the antidepressant is helpful in treating PLC, does not necessary imply a causal relationship between the agent and the condition. However, understanding how serotonergic substitution can improve emotional experience in patients with mood disorder while also being effective in patients who have pathological regulation of emotional experience is important.8 References 1. Bourgeois JA, Hilty DM, Chang CH (2004) Poststroke neuropsychiatric illness: an integrated approach to diagnosis and management. Curr Treat Options Neurol 6: Derex L, Ostrowsky K, Nighoghossian N, Trouillas P (1997) Severe pathological crying after left anterior choroidal artery infarct. Reversibility with paroxetine treatment. Stroke 28: Ghika-Schmid F, Bogousslavsky J (1997) Affective disorders following stroke. Eur Neurol 38: Dark FL, McGrath JJ, Ron MA (1996): Pathological laughing and crying. Aust NZ J Psychiatry 30: Huse A, Dennis M, Molyneux A, Warlow C, Hawton K (1989) Emotionalism after stroke. Br Med J 298: MacHale SM, O Rourke SJ, Wardl0aw JM, Dennis MS (1998) Depression and its relation to lesion location after stroke. J Neurol Neurosurg Psychiatry 64: Morris PLP, Robinson RG, Raphael B (1993) Emotional lability after stroke. Aust NZ Psychiatry 27: Parvizi J, Arciniegas D. B., Bernardini G. L., Hoffmann M. W., Mohr J. P., Rapport M.J., Schmahmann J. D., Silver J. M.,Tuhrim S. (2006) Mayo Clin Proc 81(11): Oppenheim H, Siemerling E, Mitteilungen uber Pseudobulbarparalyse und acute Bulbarparalyse. Berl Klin Wochenschr. 1886; Wilson SAK (1924) Some problems in neurology, II: pathological laughing and crying. J Neurol Psychopathol. 4: Parvizi J, Anderson SW, Martin CO, Damasio H, Damasio AR. (2001) Pathological laughter and crying: a link to the cerebellum. Brain 124:

5 12. McCullagh S, Moore M, Gawel M, Feinstein A. (1999) Pathological laughing and crying in amyotrophic lateral sclerosis: an association with prefrontal cognitive dysfunction. J Neurol Sci. 169; Jong S. Kim (2002) J Neurol 249: House Ao, Hackett ML, Anderson CS, Horrocks JA. (2004) Pharmaceutical interventions for emotionalism after stroke. Cochrane Database of Systematic Reviews. Issue 2. Art No.:CD DOI: / CD pub Burns A, Russell E, Stratton- Powell H, Tyrell P, O Neill P, Baldwin R. (1999) Sertraline in stroke-associated lability of mood. Int J Geriatric Psych. 14: Brown K.W., Sloan R.L., Pentland B. (2007) Fluoxetine as a treatment for post-stroke emotionalism. Acta Psychiatrica Scandinavica Vol98. 6; Ohkawa S, Mori E, Yamadori A. (1989) Treatment of pathological laughing with amitriptyline. Clin Neurol 29: In Japanese. 18. Parikh Rm, Robinson RG, Lipsey JR, Price TR. (1989) Nortriptyline treatment of poststroke emotional lability a double blind study. Neurology. 5(suppl 1):177. Abstract. 19. Anderaon G, Vestergaard K, Riis JO. (1993) Citalopram fro post-stroke pathological crying. Lancet 342: Poeck K (1969): Pathophysiology of emotional disorders associated with brain damage, in Handbook of Clinical Neurology, vol3, edited by Vinken PJ, Bruyn GW. Amsterdam, North-Holland, pp Robinson G.B., Parikh R. M., Lipset J. R., Starkstein S.E., Price T. R. (1993) Pathological laughing and crying following stroke: Validation of a measurement scale and a double-blind treatment study. Am J Psychiatry 150:2 pp Corresponding address: Dr Amarpreet Kaur, Department of Psychological Medicine Faculty of Medicine, University of Malaya preetsingapore@yahoo.co.uk

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