Identification and management of "delirious mania:" A rare clinical entity.

Little is known about the entity of delirious mania, apart from a few reports. Delirious mania, though a potentially lethal condition, has not found a place in the current classification system. We discuss the approach to the identification and management of delirious mania in a female patient presenting with catatonic and delirious features, with a past history of recurrent mood episodes. However, it must be emphasized that it is likely to be a rare clinical presentation of bipolar disorder and should not be the first diagnostic consideration. As a rule, any patient presenting with delirium or clouded consciousness first warrants a thorough medical workup and other specialty referrals.

INTRODUCTION

Little is known about the entity of delirious mania, apart from a few reports. Delirious mania, though a potentially lethal condition, has still not found a place in the current classification system of Diagnostic and Statistical Manual/International Classification of Diseases either as a form of catatonia or a subtype of bipolar disorder (or perhaps even as a sole exceptional, albeit extremely rare, psychiatric underlying cause of delirium).[12] The reason for presenting our case is to bring to notice how the picture of delirious mania can vary from a very severe form of mania as described by Bell in 1849 (“disease resembling some advanced stages of mania and fever”) to relatively atypical clinical picture with delirious features occurring in the context of few affective symptoms, which can be easily missed.[3] It closely resembles catatonia or may often be misdiagnosed as psychosis.[4]

CASE REPORT

A 50-year-old female with 17 years of psychiatric illness, episodic in nature, presented with the current episode for the past 8 months, with acute onset and no apparent stressor. On inquiry, her family reported that the illness began with increased talkativeness in the initial 10–14 days, when she would do her household works with more vigor (including getting up in the middle of the night to wash utensils and preparing food) accompanied by a decreased need for sleep. She started going to market without informing the family members and buying a lot of unwanted grocery. She had anger outbursts and abusive behavior when confronted. After around 2 weeks of onset, she was brought to the outpatient department (OPD) and started on tablet lithium (600 mg/day) and tablet quetiapine (up to 200 mg/day) along with lorazepam 2 mg/day. The family members noted over the next few days that the patient's speech output was markedly reduced. She would keep staring in one direction for long and not participate in any household chores. Her medications were continued for 1 month but she deteriorated. She would occasionally say she is missing some distant dead relatives or talk about her parents who expired around 10 years ago. Subsequently, her appetite and self-care deteriorated as well. She would frequently void urine on bed and had to be assisted by her daughter for changing clothes. Dose of lithium and quetiapine had been increased to 900 mg/day and 350 mg/day, respectively. She was thereafter admitted in August 2014 to the psychiatry ward in view of difficulty in management and a thorough reassessment.

In the past, she had the first episode of postpartum manic episode in the year 1996–1997 at the age of 33 followed by two more episodes (mania and depression) in the years 2002 and 2005-06 respectively. She apparently maintained well for subsequent 5 years till when she had three episodes in quick succession (possible mania followed by 2 weeks' depression, followed by treatment-emergent mania) in the year 2011–2012. The compliance to medications and follow-ups had been highly irregular throughout. Her next episode in 2013 was characterized by running away, disorganized behavior, mutism, and crying spells, after which the treating psychiatrist revised to nonaffective psychosis and started on tablet haloperidol. She again dropped out of treatment after improvement, till she presented in the year 2014 with an episode of persecutory ideas, suspiciousness, anger outbursts, and irritable behavior, which was soon managed again with haloperidol. Thereafter, she came back after an interval of 1 year in the year 2015 with the current episode, for which she was admitted.

Her physical examination was unremarkable except mild pallor and low body mass index (BMI) (19 kg/m2), and low hemoglobin, with normal thyroid function tests, kidney function tests, liver function tests, folic acid, and Vitamin B12 levels.

After admission, the patient was observed to have mutism, reduced activity, occasional crying lasting 30–40 min few times in a day, and would stay awake during the night. With no response to quetiapine in the current episode over the past few months, it was cross tapered with olanzapine, with gradual optimization of dose. Lorazepam was increased to 6 mg/day in divided dosages for catatonic features. The patient showed some improvement over a period of the next 3–4 weeks in terms of her interaction and biological functions, but progress soon plateaued. As the patient started interacting, it was found that she would often call her daughter as her younger sister and misidentify other family members and her doctor. She was unable to identify the place she was in, in spite of repeatedly reorienting her. She would go to the toilet and, at times, was found to be drinking water from the toilet pot. She would not give any explanation for these behaviors. Her sleep–wake cycle was reversed. On three occasions in the ward, she took off her clothes with no regard for surroundings (later on careful questioning, the husband reported that she had expressed to have intercourse with her husband in the ward that time). She would greet the members of the ward, sometimes clinging on to them, but could not be engaged in meaningful conversation. Serial mental status examinations showed disorientation to time/person/place, restricted affect, and no thought or perceptual abnormality. This continued persistently for the next 7–10 days. Medicine, gynecology, and neurology consultations were sought and investigations were ordered to look for the cause of disorientation. Serum electrolytes, creatine phosphokinase levels, magnetic resonance imaging brain, and electroencephalogram came out to be within normal limits. Ultrasound sonography abdomen and pelvis showed incidental finding of a benign ovarian mass (tubo-ovarian abscess). Repeated attempts to perform Mini–Mental status examination on a daily basis yielded a score of 9–11 consistently. There was minimal improvement in her symptoms despite continuing medicines and regular re-orientation cues by family members and ward team.

A re-workup was done after which her diagnosis was revised to bipolar disorder not otherwise specified (mania) with delirium (“delirious mania”) in the 6th week of admission. Lithium was restarted in the 6th week itself after discussion with the family members (serum lithium levels: 0.85 mEq/L). The patient improved within the next 2 weeks. She would now recognize her daughter and her doctor, was able to identify the place she was in, oral intake improved, she gained weight (BMI: 21 kg/m2), and self-care improved markedly. She was discharged in November 2014 on lithium 900 mg, olanzapine 20 mg, and lorazepam 6 mg/day. She had resumed with her household activities, with euthymic mood, normal biological functions, and no residual symptoms. She was seen on regular follow-ups in the OPD till the next 8 months till July 2015.

DISCUSSION

Based on the relative paucity of manic and psychotic features in the patient's presentation, we invite the readers to question whether the classical description of delirious mania is a valid clinical concept.

A literature review based on PubMed using relevant search terms revealed a total of only 14 case reports over the past decade. As it is a clinically rare condition, there are no studies, apart from case descriptions. The above patient met some features of delirious mania as described by other authors. However, the predominant picture was the absence of frank manic or psychotic symptoms. Typical manic features, such as grandiosity, expansive mood, and increased appetite, were not observed. The patient would be awake the whole night, while sleeping during the daytime. We found this to be consistent with the irregular sleep patterns of delirium rather than a biological symptom of mania. These few affective symptoms along with exhaustion of other differential diagnoses during her ward stay helped us consider delirious mania as the underlying pathology. Hallucinations and delusions have also been described as the clinical features of delirious mania in the published literature, but it remains unclear if Mrs. S was experiencing them.[5]

We found this case presentation of particular interest and a diagnostic challenge for several reasons. First, there was a particular concern about missing any medical causes of delirium, especially because she was diagnosed with an ovarian mass as well recently, for which a thorough workup was done. Second, during close observations in the ward stay, she did not demonstrate the characteristic manic symptoms or classical catatonic features. In addition, she responded minimally to lorazepam. She, however, had an excellent response to lithium within a span of a week, which is regarded as a treatment for delirious mania.

Patients with medical illness are at a risk of developing delirium, symptoms of which can mimic neuropsychiatric type of syndrome.[2] Teasing out manic-type symptoms as distinct from the delirious presentation may prove a clinical challenge. Nonetheless, in such cases, delirious mania, being a rare entity, should not be considered as the first possibility.

To summarize the current understanding on the management of delirious mania, electroconvulsive therapy/lorazepam was found to be effective for the treatment of delirious mania in most of the available reports, with few reports providing evidence for mood stabilizers as well.[126789] Nonmalignant delirious mania responds to atypical antipsychotics and mood stabilizers.[1011] Delirium usually resolves faster than mania.[4] Fink and Taylor stated in their review that the commonality in response (benzodiazepines and barbiturates) indicates that “delirious mania, catatonia, malignant catatonia, neuroleptic malignant syndrome, and toxic serotonin syndrome may be best evaluated as diverse manifestations of one syndrome for clinical and neuroscience research purpose.”[12]

This case raises a question whether delirious mania is a valid concept, particularly in such situations where features of delirium may be present alongside or even more prominent than manic or psychotic features. Finally, it must be emphasized again that it is a rare clinical presentation and as a rule, any patient presenting with delirium or clouded consciousness first warrants a thorough medical workup and other specialty referrals.

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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.

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Conflicts of interest

There are no conflicts of interest.