Hindawi Publishing Corporation Case Reports in Psychiatry Volume 2012, Article ID 720354, 7 pages doi:10.1155/2012/720354 Case Report Delirious Mania: Can We Get Away with This Concept? A Case Report and Review of the Literature Rajshekhar Bipeta1 and Majeed A. Khan1, 2 1 City 2 Deccan Nursing Home, Himayath Nagar, Hyderabad 500029, Andhra Pradesh, India College of Medical Sciences, Hyderabad 500058, Andhra Pradesh, India Correspondence should be addressed to Rajshekhar Bipeta,
[email protected] Received 19 July 2012; Accepted 14 October 2012 Academic Editors: L. Dell’Osso, C. Lanc ¸ on, and F. Oyebode Copyright © 2012 R. Bipeta and M. A. Khan. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. Background. Delirious mania (DM) as a clinical entity is well described, yet is often unrecognized in clinical practice. While most often misdiagnosed as acute psychotic episodes of organic delirium, these patients meet the criteria for mania with attendant delirium and pose therapeutic challenges. In addition to the case presentation, this paper also discusses the available literature on DM. Case Presentation. A 29-year-old man with DM was treated with a combination of electroconvulsive therapy (ECT), divalproex 2000 mg/day, loxapine 100 mg/day, and lorazepam 4 mg/day. He demonstrated clinically significant improvement by day 10, which persisted through the twelve-month follow-up period. Conclusions. DM is a severe psychiatric syndrome which should be accurately diagnosed. Patients with DM should be treated aggressively, especially with ECT. Lack of recognition of DM can lead to serious morbidity or fatal outcomes. Though the concept of DM is well established, recent psychiatric literature does not make a mention of this life threatening yet treatable condition. We propose that there is a dire need to keep this concept alive. 1. Introduction Bipolar disorder (BD) is classically viewed as a condition characterized by periods of euphoric excitement and depressive retardation, which is easy to diagnose and treat, whose treatment is exclusively pharmacological, and whose outcome is generally favourable. However, recent advances have shown that the rubric “BD” actually encompasses a variety of conditions. Also, it may have comorbidity with other psychiatric conditions. The diagnosis of BD becomes difficult when there is a variation of the classical picture. Treatment efficacy depends on the type of bipolar state. Several pharmacological agents have been introduced; the choice of the most appropriate drug in the individual patient has become much more complex. Delirious mania (DM), also known as Bell’s mania [1], is characterized by excitement, grandiosity, emotional lability, psychosis and insomnia characteristic of mania, altered consciousness, and disorientation characteristic of delirium [2–6]. The term DM was coined by Kraeplin, but was initially described by Calmeil [7], and reported to have a high morbidity [8] and mortality [1, 9]. Bell, in 1849, reported 40 patients out of 1700 admissions, who had features suggestive of DM and 75% of these patients subsequently died [1]. DM is associated with BD, and its symptoms encompass mania and acute mental confusion [6, 10, 11]. 5–20% of all patients with acute mania show signs of delirium [2]. Klerman proposed staging of manic spectrum as follows: normal, neurotic, hypomanic, manic, and delirious [12]. DM has marked similarity to stage III mania as described by Carlson and Goodwin [13]. The transition of mania to DM is marked by emergence of confusion, more hallucinations, and a marked intensification of the manic symptoms. A dreamlike clouding of consciousness may occur [14]. According to Dunayevich and Keck [15], symptoms in DM are similar to schizophrenia [13], with presence of severe anxiety, frenzied activity, and incoherence [15]. Almost all patients with DM exhibit signs of catatonia [2, 3, 16]. Taylor and Fink theorised DM as a form of catatonia because of the presence of catatonic features, and good response to electroconvulsive therapy ECT [2, 17]. In the absence of ECT. had forgetfulness for recent events. which enabled treatment of his uncontrolled life-threatening manic state. they advised use of high-dose benzodiazepines. the findings in the physical examination. The symptoms increased in severity within the previous six hours. catatonia. especially if there is a history of bipolar disorder [21]. Sociodemographic and Clinical Details. especially the malignant catatonia [11]. The temperature was 98. sleep disturbances. increased heart rate (pulse 102/minute). 2. hyperthermia. and perceptual disturbances. lithium. and malignant catatonia. 2. Otherwise. 2. and valproate cannot be considered first-line treatments in view of a reasonably delayed onset of action. increased sexual desire. The authors concluded that mania should be a differential diagnosis of elderly patients who present with confusion. hypomania. and in three of them. In 1996.3. 11]. However. unemployed graduate man from middle socioeconomic status and urban Asian background. There are no treatment guidelines. increased respiratory rate (24/minute).4. He engaged in continuous charity. including a detailed neurological assessment. Both patients responded well to divalproex (mood stabilizer). and medical workup failed to uncover an organic cause for either mania or delirium. and decreased need for sleep and food. The published literature mostly consists of case series [19]. Physical Examination. They opined that clozapine. He even assaulted the hospital staff. When normal. Fox and Bostwick reported the case of a man with DM who failed to respond to standard antimanic treatment. this is the first case of DM in published literature. The authors conclude that BD patients are at increased risk of delirium. or even aggravate the catatonia. he was finally sedated with propofol. reviewed all cases with mania. Four (out of 14 patients) had a final diagnosis of DM. He was completely off psychotropic medication for six months before the current episode. Our patient was a 29-year-old. He was exhausted. Carlson and Goodwin did a longitudinal study of 20 patients with mania. as many cases used to go unrecognized. were unremarkable. He was seeing images of snakes on fans and would become extremely fearful. He was sometimes urinating and defecating inside the house. Adolescents and children are particularly prone to the very rapid development of DM [14].2. We did pubmed and google scholar search for the available literature regarding DM. with a tendency for recurrence.6 degrees F. They recommend early recognition and aggressive management. In 2001. including the doctors. and a body weight of 102 kg. Barahona-Corrˆ ea et al.2 Many cases are precipitated by medical or neurological conditions [11] or by psychoactive substances [2. He had a two-year history of episodic illness with interepisodic premorbid level of functioning. He presented with an acute onset. overfamiliarity. suggested that the definitive treatment for DM is ECT. Along with these symptoms. 2. Earlier two episodes of mania of very high severity resolved rapidly. Though the concept of DM is now documented in literature. of ten-day duration. and in his clothes. not much is known about its aetiopathogenesis [18] and core clinical features. Nicolato et al. Various terms such as excited catatonia [18]. characterized by assaultive behaviour. our patient was confused. 14% of their patients had medically unexplained delirium. They also cautioned against the use of typical antipsychotics and anticholinergics [19]. with a tendency for recurrence [22]. for previous ten days. History of Present Illness. coprolalia. as they may cause neuroleptic malignant syndrome (NMS). The authors present five cases of DM who had “concurrent manic and delirious symptoms during hospitalization. or mixed affective state from 2006 to 2007. without any signs of stopping it. in 2012. In view of the deterioration of his condition. there is a risk of missing DM. particularly with ECT [23]. he used to stop medication. We describe a case of DM that warranted aggressive management because of extreme agitation and exhaustion. they too cautioned against the use of antipsychotics including the atypical. his relatives saw no coherence in it. The authors concluded that DM occurs rarely in BD. however. ECT was preferred in such cases. Physical examination showed elevated blood pressure (BP) (170/110 mm Hg). Weintraub and Lippmann reported two cases of elderly patients with mania whose initial presentation was delirium. disorientation. autonomic instability. Rapport could not be established. quetiapine. along with the popular term “DM” exist (2). Six (33%) patients developed disorientation suggesting that DM is a frequent occurrence [13]. and spitting at others. They comment that DM does not find mention in the current nosology as a separate diagnosis.” They caution that in view of high medical comorbidity in BD patients. In 2009. He was extremely agitated. and persistence of delirium for over a week. excessive planning. Karmacharya et al. DM cases had longer inpatient stay. with treatment. Various controversies exist regarding proper nomenclature. has typical clinical features. incessant and boastful talk. unmarried. He was continuously reciting from the holy books. The authors concluded that while propofol would not be a practical firstline treatment for agitated psychiatric patients. To our knowledge. He was turning everything . Case Reports in Psychiatry The most recent work on DM is by Lee et al. acute onset of symptoms. reported successful treatment of DM with a combination of olanzapine and ECT. Case Presentation 2. Compared to non-DM cases. cursing everybody. where standardized instruments were used to track symptoms longitudinally. Mental Status Examination. lethal catatonia. there were no signs of dehydration. third episode. it was totally misplaced. it may be helpful for refractory cases [20]. DM was initially thought to be an uncommon syndrome. DM occurred during manic/mixed affective states.1. and was not able to consistently identify even his close family members. Those with delirious features and a diagnosis of DM were specifically reviewed. sleep was better. including complete blood picture. Divalproex was increased to 2000 mg/day. There was echolalia. About two weeks after-discharge. or substance abuse. He had visual hallucinations and could see images of snakes sent to execute him. and said that he was in a railway station. Treatment Details and Course in the Hospital. His insight was relatively improved. jumping on the bed.6. Follow-Up Period. He was confused and had fluctuating levels of consciousness. Investigations. The patient was maintaining the same improvement at day 7. Divalproex was increased to 1500 mg/day. and lorazepam 4 mg/day. The elevated BP was managed with atenolol 50 mg/day. 2. He was having delusions of grandiosity with mood congruent delusions of persecution and reference. most recent episode manic.” ECT was not started as he had high BP. the aggression was better and clinically he was “much improved. there was “marked improvement with no side effects. By day 5. could not recognize his family members.9. 2. The Clinical Global Impression-Improvement scale (CGI-I) is a 7-point scale that assesses change compared to baseline. the patient had shown “marked improvement” and there was no evidence of any psychopathology. there was no relief. Laboratory studies. On day 2. At day 12. He had to be tied securely to the cot. On day 12 (day of discharge). pushing the hospital furniture. flight of ideas. different instruments were applied to measure the illness severity and treatment response (Table 1). because of extreme aggression. zuclopenthixol (Acuphase) 100 mg i/m was given. 2. and loxapine (an antipsychotic drug) 25 mg/day was added. blood sugars. with a mission to eradicate suffering and could communicate with others by telepathy. His insight into his illness was impaired. the consciousness was clear and he was well oriented. The Clinical Global Impression-Efficacy Index (CGI-E) is a 4 × 4 point scale that assesses the therapeutic effect vis-a-vis emergence of side effects [26].” He was now sleeping from 6 to 7 hours per night. There was no history of medical (including hypertension and other cardiovascular disorders) or neurological disorders. He was making sexual advances towards fellow patients and hospital staff. He would suddenly burst into tears. On admission. and clang associations. loxapine 100 mg/day. and not at baseline. Diagnosis. His self-care improved. He was referred to physician for further management of hypertension. He was diagnosed to have “bipolar disorder I. To get an objective evidence of delirium. instead he was started on pharmacological management (Figure 1).7. 2. Table 1 mentions MMSE scores only at day 12. paranoia was coming down and he started accepting food and water. destroying all that which was in his reach. he did cooperate for the same. He was started on divalproex 1000 mg/day. and atenolol 50 mg/day. He was discharged on divalproex 2000 mg/day. Loxapine was increased to 100 mg/day. Other Details. for the previous nineteen days. There was no history to suggest infection in recent past. we administered MMSE at baseline (day 1) and also at day 12 (discharge). The Clinical Global Impression-Severity scale (CGI-S) is a 7-point scale that requires the clinician to rate the severity relative to experience 3 with patients with similar diagnosis. Mini Mental Status Examination (MMSE) is used to assess the severity of cognitive impairment and to document an individual’s response to treatment [27]. By day 3. some meaningless and mostly incoherent words. At various time points. lorazepam was tapered off. By day 4. He was now amenable to suggestions and started calling others with respect. Our patient was premorbidly well adjusted. He strongly suspected that his neighbours were discussing his greatness. severe with psychotic features” as per Diagnostic and Statistical Manual of Mental Disorders (DSM IV criteria) [24]. dragging the mattresses.5. ranging from extreme dysphoria to infectious jocularity. as the BP was under control (130/90 mm Hg).” He did not remember what had happened till one day back. Assessments. clinician-administered scale to measure the severity of mania. A total of four sessions of ECT were administered. however. and haloperidol was discontinued. He had second person auditory hallucinations and would hear threatening voices of devils. 2. stereotypy.Case Reports in Psychiatry upside-down. As there was no relief even after 8 hours. as he was still very disturbed. He believed that the treating psychiatrists were butchers sent by persecutors to finish him off. Though we could not administer MMSE in detail on admission. disoriented in time. liver and thyroid functions. By day 10. lorazepam 4 mg/day. His elder sister was suffering from bipolar disorder. which were envious of his power and wanted to kill him by sending secret executioners. Higher scores reflect more severe psychopathology [25]. bilateral ECT was started. chest X ray. He believed that he was God’s messenger. loudly. urine analysis. Hence. and he was maintained on divalproex 2000 mg/day and loxapine 100 mg/day.10. He demonstrated extreme fluctuations of mood. However. our patient was “extremely ill. renal functions. and electrocardiogram did not reveal any abnormalities. the patient did not respond to all the items at baseline. haloperidol 20 mg/day. He would quietly lie down. 2. but was even pulling the cot. Loxapine was increased to 50 mg/day. echopraxia. his score on discharge was 28/30 (normal range). but was never treated. A brain computed tomography and serum creatinine phospho kinase (CPK) estimation could not be done as the family members were unwilling because of financial constraints.8. He had associated features suggestive of delirium. . The Young’s Mania Rating Scale (YMRS) is an 11-item. olanzapine 10 mg i/m was administered. These prolonged periods of extreme agitation would be followed by brief periods of sudden calmness and muteness. word salad. He was continuously talking. He was inattentive. He maintained euthymia for the next one year and then was lost to follow up. No side effects — Day 7 19 3. Atenolol 50 mg/day Very much improved∗ II ECT Divalproex 2000 mg/day Loxapine 100 mg/day Lorazepam 4 mg/day. normal 1. moderate therapeutic effect. mildly ill 2 05 — Day 10 10 1. Haloperidol 20 mg/day. Scales/scores YMRS (0–60) CGI-S (1–7) CGI-I (1–7) CGI-E (01–16) MMSE (1–30) Day 1 58 7. . MMSE: Mini Mental Status Examination. Haloperidol 20 mg/day Lorazepam 4 mg/day. blood pressure under control Divalproex 1500 mg/day. CGI: Clinical Global Impression. Loxapine 100 mg/day Lorazepam 4 mg/day. Day 1 No change∗ Day 2 Very disturbed Day 2: 8 AM No change∗ Day 2: 4. Atenolol 50 mg/day. Loxapine 100 mg/day Lorazepam 4 mg/day. moderately ill 2. Loxapine 50 mg/day Lorazepam 4 mg/day. Lorazepam 4 mg/day Much improved∗ Atenolol 50 mg/day Divalproex 1500 mg/day. Atenolol 50 mg/day Much improved∗ I ECT Divalproex 1500 mg/day. IV ECT Day 4 Day 5 Day 7 Day 10 Day 12 ∗ CGI-I = Clinical Global Impression-Improvement. very much improved 01. Loxapine 50 mg/day Lorazepam 4 mg/day. NA: Not Applicable. Atenolol 50 mg/day Olanzapine 10 mg i/m stat Zuclopenthixol (acuphase) 100 mg i/m stat Loxapine 25 mg/day No change∗ but sleep better Day 3 Divalproex 1000 mg/day. extremely ill NA NA — Day 4 33 4.40 PM Divalproex 1000 mg/day. marked therapeutic effect. ECT = Electroconvulsive therapy Figure 1: Flowchart depicting the treatment details in our patient. Haloperidol 20 mg/day Loxapine 50 mg/day. CGI-I: CGI-Improvement scale. Lorazepam 4 mg/day. Atenolol 50 mg/day Divalproex 1000 mg/day. much improved 05. Atenolol 50 mg/day Very much improved∗ III ECT Divalproex 2000 mg/day. No side effects — Day 12 7 1 1 01 28 YMRS: Young’s Mania Rating Scale. CGI-S: CGI-Severity scale. Atenolol 50 mg/day Haloperidol discontinued Much improved∗ .4 Case Reports in Psychiatry Table 1: Assessments at various time points in our patient. He suggests that the term “psychogenic” indicates a “mode of explanation. A case of DM as per Bond’s criteria. However. 3. 10]. After starting loxapine. Delirious mania Present Sudden onset/intense excitement Stereotypy Tachycardia Tachypnoea Hypertension Pressured speech/mutism Present Present Grandiosity Emotional lability Delusions Insomnia Disorientation Altered consciousness Negativism Flight of ideas Absent Hyperthermia Posturing Absent Catalepsy Rigidity Cycle from excited state to stuporous state ∗ 5 of meaning with which both those terms are used. (iv) history of mania. 38]. Though case reports find the lowest position in evidence-based research. 36]. GABA-ergic transmission in the orbitofrontal premotor and motor cortices is implicated in the aetiopathogenesis of catatonia. Discussing the relationship between “psychogenic”/“functional. 44]. and the drugs were given at high doses. and (iii) our patient was off all psychotropic medication since six months before the current episode. In Kraines’ word “one seems justified in utilizing just as much medication as necessary. with no appreciable benefit [23]. A point worth mentioning is that our patient’s sleep was disturbed since ten days before admission. Not treating such patients effectively and incorrect attribution to drugs can be dangerous” [31]. The prognosis. but. Catatonia may also be attributed to glutamate (NMDA receptor) hyperactivity. 10]. 19. some researchers consider these to be detrimental in presence of catatonic features [23. serum CPK level could not be ascertained in our patient. There was no muscular rigidity. (i) acute onset of symptoms. Therefore. 40].. He also met Fink’s criteria for DM and catatonia (Table 2) [3. But. Discussion This could well be our “most severe” recorded case of mania. In our patient. This is in keeping with earlier reports [2. 43]. but exacerbation to this height of mania reached within hours. no matter what the standard dosage is. and hyperthermia (i. and this is in contrast to earlier case reports needing prolonged hospitalization [22]. . however. Catatonia∗ Disorganized thoughts Disorganized speech Refusing food and fluids Requires 2 or more signs for ≥24 hours [3]. and (vi) responsivity to treatment for mania [28]. the sleep started improving day 3 onwards. Several researchers argue that loxapine may behave as an atypical antipsychotic [34]. 41]. A classical case of bipolar-I. cautioned that “the recognition of DM is critical once an “organic aetiology” has been excluded. who responded wonderfully to treatment. hence. . Typical antipsychotics are found to be beneficial in delirium [35]. in order to procure sufficient quiet to prevent cardiac failure. Hart talks about the confusion that has resulted from the diversities 4. Our patient had a very brief inpatient stay. (v) family history of bipolar disorder. 38]. obtundation.” [30]. ECT should be considered in early stages of DM [40. Early recognition of sleep disturbance is crucial for the prevention of relapse or recurrence in BD patients [32]. 23. a brief course of four bilateral ECTs was further able to hasten the recovery. One controversy related to our case may be that we changed treatment drastically. glutamate antagonist therapy with amantadine may help [45]. (ii) presence of mania. 8. (iii) features of delirium. 37. which is considered so ominous. But NMS was ruled out in our patient as (i) the neurological examination was within normal limits. some authors caution against their usage in DM [19.e.. NMS was the differential diagnosis considered. altered sensorium. mutism. we should remember that DM is a life-threatening condition and should be treated aggressively..” and “organic” in delirium. in case of nonresponse with lorazepam or ECT. Associated features include akinesia. but with an element of delirium. Swartz et al. The classic features of NMS include muscular rigidity. these continue to guide researchers in planning more rigorous methodologically sound studies.Case Reports in Psychiatry Table 2: Catatonia subgroups: symptoms of delirious mania and catatonia [3. temperature >100. GABA-A potentiators like high-dose lorazepam (3-4 mg/day) may benefit some patients [19. Conclusions Our case illustrates the real world challenges which clinicians face in their day-to-day practice.. might be changed under such treatment” [9]. The serum CPK level is increased in nearly all cases [29]. . Atypical antipsychotics are proposed to be useful in DM [19. 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