Delirious mania with catatonia succesfully treated with electroconvulsive therapy: a case report

Delirious mania is a severe neuropsychiatric syndrome characterized by the acute onset of mania, delirium, and psychosis, often accompanied by catatonic features and requiring urgent multidisciplinary evaluation [1][2].This case aims to emphasize the importance of a comprehensive differential diagnostic approach to exclude neurological, infectious, autoimmune, and metabolic disorders in a patient presenting with catatonia within the spectrum of delirious mania. Additionally, the case underscores the therapeutic role of electroconvulsive therapy (ECT) and psychopharmacological interventions, as well as the potential confounding effects of treatment on biochemical markers.

A 22-year-old male patient presented to the emergency department with a one-week history of insomnia, irritability, and aggression, followed by three days of refusal of oral intake, incoherent speech, and repetitive behaviors. He was admitted to the psychiatric ward.

On examination, he was uncooperative, had no eye contact, and failed to provide meaningful responses. Negativism, perseverative speech, and severe psychomotor agitation were observed. Oral intake was significantly reduced, with selective acceptance of liquids. He had not defecated for one week and demonstrated urinary retention. Physical examination revealed a distended bladder, rigidity, and diaphoresis.

His psychiatric history revealed first-onset psychotic symptoms at age 17, including insomnia, hallucinations, disorganized behavior, and grandiose/mystical delusions; hospitalization twice to date, treatment with clozapine and remission.

The patient was initially considered to be in a catatonic state, and treatment with benzodiazepines with clozapine was initiated. Supportive care (hydration and nutrition) was provided, and urinary catheterization was required.

ECT was started quickly, clozapine was titrated with administration of ECT on alternate days. After the third session, oral intake improved significantly. Following the fourth session, meaningful communication and appropriate responses were observed, although clinical status remained fluctuating. A differential diagnostic workup was performed. Meningitis panel oligoclonal bands, and autoimmune encephalitis panels in both serum and Cerebrospinal Fluid were negative. Serologic testing for Human Immunodefficiency Virus(HIV), Hepatitis B virus, Hepatitis C virus, syphilis, and brucellosis was negative. Brain Magnetic Resonance Imaging(MRI) with contrast, susceptibility-weighted imaging (SWI), and Electroencephalography(EEG) revealed no abnormalities.

Given early-onset, atypical, and treatment-resistant course, metabolic evaluation was conducted. Lysosomal, peroxisomal, organic acid, and fatty acid oxidation disorders were largely excluded. Although elevated urinary porphyrins were detected, the absence of porphobilinogen and lack of clinical correlation did not support a diagnosis of porphyria[3][4]. Elevated 7-dehydrocholesterol levels raised suspicion for Smith-Lemli-Opitz syndrome; however, absence of developmental delay and dysmorphic features, along with overall biochemical profile, made the diagnosis unlikely[5]. These abnormalities were considered secondary to metabolic stress and pharmacological effects.

By the seventh ECT session, spontaneous urination resumed, and progressive improvement in social interaction, and oral intake was observed. Subsequently, a manic switch was noted, characterized by decreased need for sleep, increased energy, elevated mood, hyperactivity, grandiose and mystical delusions. Valproate was initiated. The patient was discharged on clozapine 350 mg/day and valproate 1250 mg/day in a clinically stable condition, with resolution of catatonic symptoms and improvement in affective instability.

This case emphasizes that delirious mania and catatonia require a careful multidisciplinary approach to rule out organic causes. ECT proved to be an effective treatment in this complex presentation.

References

1. JACOBOWSKI, NATALIE L. MD; HECKERS, STEPHAN MD; BOBO, WILLIAM V. MD, MPH. Delirious Mania: Detection, Diagnosis, and Clinical Management in the Acute Setting. Journal of Psychiatric Practice 19(1):p 15-28, January 2013. | DOI: 10.1097/01.pra.0000426324.67322.06 2)Tyler Wheeler, Sarayu Vasan, Treatment of delirious mania with catatonia in a community teaching hospital: A case series, Psychiatry Research Case Reports, Volume 2, Issue 2,2023,100190,ISSN 2773-0212, https://doi.org/10.1016/j.psycr.2023.100190. 3) Eliane Sardh, Pauline Harper, Dan E.H. Andersson, Ylva Floderus, Plasma porphobilinogen as a sensitive biomarker to monitor the clinical and therapeutic course of acute intermittent porphyria attacks, European Journal of Internal Medicine, Volume 20, Issue 2,2009,Pages 201-207,ISSN 0953-6205, https://doi.org/10.1016/j.ejim.2008.06.012. 4)Duque-Serrano L, Patarroyo-Rodriguez L, Gotlib D, Molano-Eslava JC. Psychiatric Aspects of Acute Porphyria: a Comprehensive Review. Curr Psychiatry Rep. 2018 Feb 2;20(1):5. doi: 10.1007/s11920-018-0867-1. PMID: 29392446. 5) Diaz-Stransky A, Tierney E. Cognitive and behavioral aspects of Smith-Lemli-Opitz syndrome. Am J Med Genet C Semin Med Genet. 2012 Nov 15;160C(4):295-300. doi: 10.1002/ajmg.c.31342. Epub 2012 Oct 5. PMID: 23042585.