Diagnostic and therapeutic hardships with mixed affective state presenting as catatonia in a patient with intellectual disability

Karthick Subramanian; Siddharth Sarkar; Ravi Philip Rajkumar

doi:10.4103/jmhhb.jmhhb_22_16

CASE REPORT

Year : 2017 | Volume : 22 | Issue : 1 | Page : 69-71

Diagnostic and therapeutic hardships with mixed affective state presenting as catatonia in a patient with intellectual disability

Karthick Subramanian¹, Siddharth Sarkar², Ravi Philip Rajkumar¹
¹ Department of Psychiatry, Jawaharlal Institute of Postgraduate Medical Education and Research, Puducherry, India
² Department of Psychiatry, All India Institute of Medical Sciences, New Delhi, India

Date of Web Publication

14-Jul-2017

Correspondence Address:
Karthick Subramanian
Department of Psychiatry, Jawaharlal Institute of Post Graduate Medical Education and Research, Dhanvantri Nagar, Puducherry - 605 006
India

Source of Support: None, Conflict of Interest: None

Check

DOI: 10.4103/jmhhb.jmhhb_22_16

Abstract

Mixed affective episodes can be misdiagnosed, especially in patients with intellectual disability (ID). We describe the case of an 18-year-old girl with mild ID, who presented with features of catatonia during the first mixed episode. These symptoms responded well to electroconvulsive therapy, following which clear affective symptoms emerged. Her affective episode did not respond adequately to olanzapine but improved significantly after the addition of sodium valproate. The difficulties of diagnosing affective episodes in persons with intellectual disabilities are discussed. This case suggests that mixed affective episodes should be considered in the differential diagnosis when poorly elaborated affective and psychotic symptoms are present in a patient with ID.

Keywords: Catatonia, electroconvulsive therapy, intellectual disability, mixed affective episode

How to cite this article:
Subramanian K, Sarkar S, Rajkumar RP. Diagnostic and therapeutic hardships with mixed affective state presenting as catatonia in a patient with intellectual disability. J Mental Health Hum Behav 2017;22:69-71

How to cite this URL:
Subramanian K, Sarkar S, Rajkumar RP. Diagnostic and therapeutic hardships with mixed affective state presenting as catatonia in a patient with intellectual disability. J Mental Health Hum Behav [serial online] 2017 [cited 2023 Mar 31];22:69-71. Available from: https://www.jmhhb.org/text.asp?2017/22/1/69/210702

Introduction

Mixed affective states are often misdiagnosed,^[1],[2] and less frequently described in individuals with intellectual disability (ID). Only one study discusses diagnosing mixed affective episodes in adolescents with ID.^[3] Clinicians may need to rely on the overt behavioral characteristics than the subjective reporting of cognitive features while diagnosing mood disorders in individuals with ID.^[4] A sizeable proportion of patients with mixed affective episodes can present with catatonic symptoms.^[1],[5]

We present the rare instance of an adolescent with ID in whom catatonia heralded a mixed episode which eventually responded to electroconvulsive therapy (ECT). We also present the diagnostic and therapeutic dilemmas encountered during the patient management.

Case Report

An 18-year-old unmarried female from a low socioeconomic background presented to our emergency services with an acute onset of behavioral oddities that lasted for a week. Irritability, weeping spells, perplexity, motoric restlessness, verbosity, and self-muttering behaviors were present. Her sleep and appetite were compromised for a couple of days. An intact sensorium ruled out delirium. Her premorbid history was marred with delayed milestones, poor academic performance, calculation difficulties, deficits in adaptive functioning such as performing household chores and attending self-care needs. Her mother had suffered from an undiagnosed mental illness. When 3-year-old, she was abandoned by her parents. She was adopted and raised affectionately by her aunt. There was no history of behavioral disturbances in the patient. A provisional diagnosis of acute and transient psychotic disorder (ATPD) and mild ID was considered.

Injectable haloperidol along with promethazine was given to reduce her agitation, and she was concurrently started on risperidone (2 mg/day). She developed sudden onset of muscular rigidity in her extremities, resting tremors, bradykinesia, and dysphagia. The severe extrapyramidal symptoms (EPS) responded to promethazine (50 mg i.m.). After 2 days, she returned to the clinic with signs of mutism, negativism, staring, posturing, echopraxia, and poor oral intake. Her ID limited further elicitation of catatonic signs since the patient could not follow complex instructions. Catatonia was diagnosed based on the symptom profile (Baseline Bush-Francis Catatonia Rating Scale = 18).^[6] Normal creatine phosphokinase (CPK) levels ruled out neuroleptic malignant syndrome (CPK-total = 126 IU/ml). A computerized tomography scan of the brain was normal. The absence of a Kayser–Fleischer ring and normal serum ceruloplasmin levels ruled out Wilson's disease.

The patient was started on lorazepam (up to 16 mg/day). ECT was started due to poor response in catatonic symptoms. Over the next 2 weeks, the improvement was sequentially noticed in food refusal, negativism, posturing, and mutism, and hence, ECT was discontinued after seven sessions. There was no recurrence of catatonia following the termination of ECT. Subsequently, weeping spells were noted, and in interviews, the patient reported sadness and expressed death wishes along with poorly elaborated auditory hallucinations. Olanzapine (7.5 mg/day) was prescribed for the control of auditory hallucinations, and she was sent home for 2 days at her caregivers' request. At home, she was noticed to be more active than usual. She would start a task but would soon shift to another without finishing the previous one. She was noted to be gleeful with her friends and would barge into their houses and demanding them to play with her, to the extent that they found her annoying. She would dance in the streets to her neighbors' amusement. She also spent half the time at home crying loudly reporting a vague distress. The mood fluctuations were unpredictable, and they lasted several hours within a day. The diagnosis was revised to “mixed affective state” (ICD-10 F38.00)^[7] from initial provisional diagnosis of ATPD, as she had alternating depressive and manic symptoms in the course of a day. The Hamilton Depression Rating Scale (HDRS)^[8] score was 15 and Young Mania Rating Scale (YMRS)^[9] score was 15 at the time of diagnostic revision. She was given up to 20 mg olanzapine for 3 weeks. Due to an inadequate response, valproate was given up to 25 mg/kg/day. Her mood symptoms were improved from the 5^th day of inducting valproate therapy. By 2 months, HDRS and YMRS dropped to 2 and 8, respectively. The clinical diagnosis of mild ID was endorsed by an equivalent social quotient of 50 in the Vineland Social Maturity Scale.^[10] The patient was subsequently discharged with no residual catatonic or mood symptoms. The patient was followed up for 3 months without further relapses. The informed consent was obtained from the patient's caregivers for treatment and write-up of this report.

Discussion

The case illustrates the bemusing clinical picture in patients with ID and a comorbid mixed affective episode presenting with catatonia. Eliciting catatonic signs, performing standard measurements of intelligence and elucidating psychopathology proved to be difficult due to ID. In our patient, distinct behavioral changes with affective and psychotic symptoms were noted. The diagnosis of a mixed episode was solely based on such behavioral observations when mood-congruent cognitive symptoms such as grandiosity or hopelessness could not be assessed. The diagnosis was considered tenable as affective episodes in patients with ID can be inferred from their demeanor.^[11],[12] Mood lability alone could not account for the symptomatology as there were concurrent psychomotor changes during the “ups and downs.” Although we experienced some difficulties, we ruled out ultradian rapid cycling since there was a considerable lack of self-report of mood and cognitive symptoms with a cyclical course within a day. Furthermore, at most instances, an admixture of manic and depressive symptoms was present throughout the course of illness. To summarize, the presence of a young age of onset of illness, childhood trauma, fluctuating mood symptoms, extreme degrees of psychomotor agitation or retardation and sensitivity to developing EPS should prompt the clinician to contemplate a diagnosis of bipolar disorder in individuals with ID. Previous studies suggest that ECT can be efficacious in patients who present with a mixed affective episode, similar to our patient.^[1] A single case report has described the successful use of ECT in a female patient with ID and catatonia secondary to depression.^[11] To the best of our knowledge, our patient is the first reported case to describe the efficacy of ECT in a patient with ID presenting in catatonia during a mixed affective state.

Conclusion

Atypical presentations of mixed affective states should be considered in the differential diagnosis of poorly elaborated affective and psychotic symptoms in patients with intellectual impairment, and such patients may require intensive pharmacotherapy.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References

1.	Bräunig P, Krüger S, Shugar G. Prevalence and clinical significance of catatonic symptoms in mania. Compr Psychiatry 1998;39:35-46.
2.	Dilsaver SC, Akiskal HS. High rate of unrecognized bipolar mixed states among destitute Hispanic adolescents referred for “major depressive disorder”. J Affect Disord 2005;84:179-86.
3.	Brunelle J, Consoli A, Tanguy ML, Huynh C, Perisse D, Deniau E, et al. Phenomenology, socio-demographic factors and outcome upon discharge of manic and mixed episodes in hospitalized adolescents: A chart review. Eur Child Adolesc Psychiatry 2009;18:185-93.
4.	Ross E, Oliver C. The assessment of mood in adults who have severe or profound mental retardation. Clin Psychol Rev 2003;23:225-45.
5.	Krüger S, Cooke RG, Spegg CC, Bräunig P. Relevance of the catatonic syndrome to the mixed manic episode. J Affect Disord 2003;74:279-85.
6.	Bush G, Fink M, Petrides G, Dowling F, Francis A. Catatonia. I. Rating scale and standardized examination. Acta Psychiatr Scand 1996;93:129-36.
7.	World Health Organization. The International Classification of Mental and Behavioral Disorders: Clinical Description and Diagnostic Guidelines. 10^th ed. Geneva: WHO; 1992.
8.	Hamilton M. A rating scale for depression. J Neurol Neurosurg Psychiatry 1960;23:56-62.
9.	Young RC, Biggs JT, Ziegler VE, Meyer DA. A rating scale for mania: Reliability, validity and sensitivity. Br J Psychiatry 1978;133:429-35.
10.	Sparrow SS, Balla DA, Cicchetti DV. Vineland Adaptive Behavior Scales, 2^nd ed. Survey Forms Manual. AGS Publishers; 2005.
11.	Aziz M, Maixner DF, DeQuardo J, Aldridge A, Tandon R. ECT and mental retardation: A review and case reports. J ECT 2001;17:149-52.
12.	Esbensen AJ, Rojahn J, Aman MG, Ruedrich S. Reliability and validity of an assessment instrument for anxiety, depression, and mood among individuals with mental retardation. J Autism Dev Disord 2003;33:617-29.