This article was downloaded by: [Simon Fraser University] On: 20 November 2014, At: 05:38 Publisher: Routledge Informa Ltd Registered in England and Wales Registered Number: 1072954 Registered office: Mortimer House, 37-41 Mortimer Street, London W1T 3JH, UK Neurocase: The Neural Basis of Cognition Publication details, including instructions for authors and subscription information: http://www.tandfonline.com/loi/nncs20 Pathological gambling, delusional parasitosis and adipsia as a post-haemorrhagic syndrome: A case report a a a a Gianluca Floris , Antonino Cannas , Monica Melis , Paolo Solla & Maria Giovanna Marrosu a a Dipartimento di Scienze Cardiovascolari e Neurologiche, Sezione Neurologia, Ospedale San Giovanni di Dio, ASL 8 , Università di Cagliari , Cagliari, Italy Published online: 31 Oct 2008. To cite this article: Gianluca Floris , Antonino Cannas , Monica Melis , Paolo Solla & Maria Giovanna Marrosu (2008) Pathological gambling, delusional parasitosis and adipsia as a post-haemorrhagic syndrome: A case report, Neurocase: The Neural Basis of Cognition, 14:5, 385-389, DOI: 10.1080/13554790802385418 To link to this article: http://dx.doi.org/10.1080/13554790802385418 PLEASE SCROLL DOWN FOR ARTICLE Taylor & Francis makes every effort to ensure the accuracy of all the information (the “Content”) contained in the publications on our platform. However, Taylor & Francis, our agents, and our licensors make no representations or warranties whatsoever as to the accuracy, completeness, or suitability for any purpose of the Content. Any opinions and views expressed in this publication are the opinions and views of the authors, and are not the views of or endorsed by Taylor & Francis. 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Terms & Conditions of access and use can be found at http://www.tandfonline.com/page/terms-and-conditions NEUROCASE 2008, 14 (5), 385–389 NNCS Pathological gambling, delusional parasitosis and adipsia as a post-haemorrhagic syndrome: A case report Gianluca Floris, Antonino Cannas, Monica Melis, Paolo Solla, and Maria Giovanna Marrosu Downloaded by [Simon Fraser University] at 05:38 20 November 2014 PATHOLOGICAL GAMBLING POST-HAEMORRHAGIC SYNDROME Dipartimento di Scienze Cardiovascolari e Neurologiche, Sezione Neurologia, Ospedale San Giovanni di Dio, ASL 8, Università di Cagliari, Cagliari, Italy The authors describe the case of a 70-year-old male who developed a peculiar syndrome characterized by pathological gambling (PG), delusional parasitosis and adipsia with mild frontal symptoms. Onset of the syndrome followed a cerebral haematoma involving hypothalamic and bilateral ventromedial prefrontal areas. The potential manifestation of PG following a lesion in the above areas may contribute towards furthering the understanding of pathological conditions underlying this disorder. Keywords: Pathological gambling; Delusional parasitosis; Adipsia; Cerebral haematoma; Orbitofrontal ventromedial areas. INTRODUCTION PATIENT Dysfunction of the orbitofrontal ventromedial cortex is mainly associated with complex behavioural and cognitive deficits, which have not been clarified (Nachev, 2006). Abnormalities in decisionmaking, emotion, impulse control and social functioning have been described in patients with damaged ventromedial prefrontal cortex (VMPC) (Bechara & Van Der Linden, 2005). Indeed, several authors have hypothesized a role of VMPC dysfunction in addictive and compulsive behaviour (Schoenbaum, Roesch, & Stalnaker, 2006). The physiopathology of pathological gambling (PG) is currently the subject of intense debate in the literature. Post-lesional cases of PG have not generally been reported. The authors here describe a peculiar syndrome in which PG, delusional parasitosis and adipsia were manifested following cerebral haematoma. Our patient was a 70-year-old, right-handed man with a high school education. His past medical history revealed no evidence of cranial trauma, substance or alcohol abuse, or neuropsychiatric disorders. The patient was affected by chronic ischaemic cardiomyopathy and arterial hypertension. Treatment with L-thyroxine had been prescribed 16 years previously following a thyroidectomy. Following a headache at the age of 68 years, cranial CT was performed leading to the incidental finding of a left anterior cerebral artery aneurysm at the base of the anterior communicating artery. Two months later the patient underwent surgery for exclusion of the aneurysm. Post-surgery, following a hypertensive crisis, the patient developed a small haematoma in hypothalamic and bilateral orbitofrontal ventromedial areas, predominant on Address correspondence to Gianluca Floris, MD, Clinica Neurologica, Ospedale S. Giovanni di Dio, Via Ospedale 46, 09100 Cagliari, Italy. (E-mail: lgr.floris@tiscali.it). © 2008 Psychology Press, an imprint of the Taylor & Francis Group, an Informa business http://www.psypress.com/neurocase DOI: 10.1080/13554790802385418 386 FLORIS ET AL. Downloaded by [Simon Fraser University] at 05:38 20 November 2014 Figure 1. CT scans show a small haematoma involving suprasellar and bilateral orbitofrontal ventromedial areas predominant on the left side. the left side (Figure 1). He rapidly developed a marked frontally predominant cognitive impairment characterized by an attention deficit, a memory deficit with florid confabulations, insight and reasoning disturbances, logorrhoea, impersistence, impulsivity, dysphoria, irritability, verbal disinhibition and at times confusional episodes. The neuropsychological profile was consistent with a bilateral dysfunction. Moreover, a persistent and marked adipsic hypernatremia (sodium up to 170 mEq/l) was detected. One month later the patient once again underwent neurosurgical intervention for a left subdural fronto-parietal haematoma associated with mild confusional state and right hemiparesis. Neuroradiological check-up demonstrated complete evacuation of the haematoma, with the patient returning to the same clinical conditions observed following initial surgery. After a further 2 months, a potential dysfunction of ADH, hypophysis, thyroid and adrenal glands was ruled out, and diagnosis of hypodipsic hypernatremia was made. In view of a total lack of thirst displayed by the patient, a regular water intake (2 l/die) with sodium correction was prescribed. Normalization of sodium levels led to an improvement of attention and disappearance of confusional episodes. Over the same period the patient developed a delusional personal and environmental parasitosis (pediculosis) accompanied by visual illusions and hallucinations. This delusional parasitosis was characterized by the patient believing his person and the surrounding environment to be infested with lice, frequently observed in his home, his clothes and his food. No criticism of the latter hallucinations and illusions was made, leading to onset of a state of agitation in the patient who subsequently displayed behaviours of avoidance and disinfestation of ‘parasites’. No other forms of delusional ideas or misperceptions were detected. The patient was treated with low doses of olanzapine (5 mg), leading to clinical improvement. Over the next few months, a progressive cognitive improvement was observed, although mild irritability, reduced insight and criticism of his disorders, mild deficit in executive functions and a mild psychosis persisted. Attention, verbal memory and reasoning returned to normal (Table 1) with a good recovery of BADL (Mahoney & Barthel, 1965) and IADL (Lawton & Brody, 1969). Five months after the onset of cognitive disturbances, the patient once again ventured outside the house alone to do his shopping. Subsequently, his relatives noticed how money went missing at times. Indeed, it was later revealed that the patient had rapidly squandered the sums taken (a total of approximately 10,000 Euros with a weekly gambling expenditure of 500–1500 Euros) in lotteries. It was soon established that during his outings the patient spent the majority of his time in betting activities. It should, however, be emphasized that the patient was retired and money spent on gambling was disproportionately higher than his income, leading to his bank account being grossly overextended. In the past, the patient had bet small weekly sums (40 Euros) on the lotteries with no signs of abuse. A follow-up cranial CT and brain MRI revealed the absence of new cerebral lesions. DISCUSSION The patient developed an interesting syndrome characterized by PG, adipsia, psychosis of personal and environmental infestation associated with mild PATHOLOGICAL GAMBLING POST-HAEMORRHAGIC SYNDROME 387 TABLE 1 Neuropsychological findings of the subject in two evaluation after 2 months (first examination) and after 15 months (second examination) from the onset of the cognitive impairment First examination Second examination Cut-off Orientation place/time −/− +/+ +/+ Verbal Memory (15 Rey’s word) Immediate recall Delay recall Recognition 20.1 0 − 29.1 5.2 + >28.52 >4.68 + Logical memory (Short story recall) Immediate recall Delay recall 0 0 3.2 3.2 >3.1 >2.39 Downloaded by [Simon Fraser University] at 05:38 20 November 2014 Test Visuo-spatial memory (Rey–Osterrieth figure) Immediate recall * Delay recall * Digit span forward 4.75 Digit span backward 3 Constructional praxis 6.9 Phonological verbal fluency 11 Semantic verbal fluency 6 Naming + Raven test (coloured matrices) 16.1 Attentional matrices 31.75 Modified Card Sorting Test (MCST) NC * * 4.75 3 8.9 30 14 + 20.1 45.75 + >18.96 >30 + Trail making test A B 92 299 >94 283 130 NC >3.75 3 >7.3 >16 >96 +, normal; −, impaired; NC, not completed; *, performed with limited collaboration. frontal symptoms. An initially marked and bilateral frontally predominant cognitive impairment improved considerably, clearly emphasizing the selective psychotic disorder and the isolated loss of impulse control. The latter was observed as soon as the patient ventured outside his house alone. The patient met DSM-IV clinical criteria for PG (American Psychiatric Association, 1994). He felt compelled to gamble and perpetually engaged in procuring money for gambling, he displayed a marked irritability associated with attempts to cease or reduce gambling. In spite of counselling and regardless of the detrimental personal and economic consequences, the man did not change his gambling habits but merely attempted to conceal the extent of his gambling by lying to his family and therapist, and turning to friends to obtain funds for his gambling or to recoup gambling losses. Furthermore, the behaviour observed was at no time associated with a manic episode. Although the syndrome reported developed progressively, it may reasonably be argued that it originated following cerebral injury caused by parenchymal haematoma. In particular, PG in our patient may be linked to injury to the VMPC. This hypothesis is supported by previous studies demonstrating involvement of the ventromedial frontal cortex in decision-making, reporting how patients with bilateral lesions of VMPC develop cognitive impulsiveness and are insensitive to future consequences of their own decisions, being primarily guided by their immediate prospects (Bechara, Tranel, & Damasio, 2000). The orbitofrontal cortex is critically involved in representing the current relative value of stimuli, playing a crucial role in assessing perceived or expected outcomes to guide decisions. A singular association with structures including the amygdala, nucleus accumbens, hypothalamus and other prefrontal cortex areas is fundamental to this regard. Damage to the VMPC may affect value-based learning and value-based decision-making (Fellows, 2007). Cases of patients with ventromedial frontal lesions associated to impairment of decision-making and abnormal risk-taking behaviour have been widely reported in the literature. Focal frontal lobe Downloaded by [Simon Fraser University] at 05:38 20 November 2014 388 FLORIS ET AL. lesions (ventrolateral and orbital) may be related to the reward-based aspects of decision-making and risky behaviour (Floden, Alexander, Kubu, Katz, & Stuss, 2008). The orbitofrontal cortex is involved in the processing of rewards or punishments (Kringelbach and Rolls, 2004). Patients with orbital frontal damage have difficulty in selecting responses with flexibility based on changes in reward contingencies and on anticipation of outcomes. Moreover, a study of functional MRI in patients with PG showed a dysfunction of ventral striatal and ventromedial prefrontal areas (Reuter et al., 2005). Several studies have demonstrated a higher prevalence of PG in parkinsonian patients, implicating the action of dopaminergic medication on the mesocorticolimbic reward system (Avanzi, 2006; Voon et al., 2006). Brain dopamine plays a central role in the latter system, linked to the ventromedial frontal cortex. PG, together with other impulsive, addictive and compulsive behaviours may be related to dysfunction of the above circuits resulting in a reward deficiency syndrome (Blum et al., 2000). Thus, on the basis of knowledge available to date, it can be assumed that dysfunction of the reward system may be due to genetic and environmental influences (Blum et al., 2000; Bowirrat & Oscar-Berman, 2005) and related to pharmacological, neurodegenerative or, perhaps, lesional causes. Other studies support a role of working memory deficit in decision making in individuals with substance addictions and in perseverative chasing in PG (Bechara & Martin, 2004; Leiserson & Pihl, 2007). However, cases of PG manifested subsequent to cerebral damage have rarely been reported to date (Nakaki et al., 2007), thus rendering the present case of particular interest. The patient investigated was likely affected by a reward-related decision-making dysfunction leading to an inability to control a previously controlled passion for gambling. Delusional parasitosis, frequently reported as an organic syndrome, is not acknowledged as being produced by a single brain lesion. Accordingly, onset of such a manifestation subsequent to the lesion described is likewise of interest. Adipsia with hypernatremia is a rare occurrence following hypothalamic lesions, of particular importance due to the potential severity of outcome. To this regard, the global cognitive improvement observed in our patient following correction of sodium levels should be emphasized. The present report describes a rare post-lesional syndrome that may provide numerous points for discussion. 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