|Year : 2020 | Volume
| Issue : 1 | Page : 60-62
Catatonia in a young woman with intellectual disability and vitamin D deficiency managed with electroconvulsive therapy
Aarzoo Suman, Anish Shouan, Sandeep Grover
Department of Psychiatry, Postgraduate Institute of Medical Education and Research, Chandigarh, India
|Date of Submission||26-Mar-2020|
|Date of Decision||24-Apr-2020|
|Date of Acceptance||05-Jul-2020|
|Date of Web Publication||7-Oct-2020|
Department of Psychiatry, Post Graduate Institute of Medical Education and Research, Chandigarh
Source of Support: None, Conflict of Interest: None
Catatonia as a syndrome is known to be associated with multiple psychiatric and medical conditions. Although catatonia has been reported in patients with neurodevelopmental disorders such as autism, there is lack of literature on catatonia in subjects with intellectual disability, especially among the adults. In terms of nutritional deficiencies, catatonia has rarely been reported to be associated with Vitamin D deficiency. In this report, we report a 20-year-old female who presented with catatonia in the absence of evidence of any other psychiatric disorders. Physical examination and investigations did not reveal evidence of any other abnormality, except for Vitamin D deficiency. However, the patient required treatment with electroconvulsive therapy for the management of catatonic symptoms followed by olanzapine up to 7.5 mg for managing psychotic symptoms on follow-up.
Keywords: Catatonia, intellectual disability, Vitamin D deficiency
|How to cite this article:|
Suman A, Shouan A, Grover S. Catatonia in a young woman with intellectual disability and vitamin D deficiency managed with electroconvulsive therapy. J Mental Health Hum Behav 2020;25:60-2
|How to cite this URL:|
Suman A, Shouan A, Grover S. Catatonia in a young woman with intellectual disability and vitamin D deficiency managed with electroconvulsive therapy. J Mental Health Hum Behav [serial online] 2020 [cited 2022 Jan 22];25:60-2. Available from: https://www.jmhhb.org/text.asp?2020/25/1/60/297414
| Introduction|| |
Catatonia can be a clinical manifestation of multiple underlying etiologies, varying from a whole range of organic causes to affective disorders, schizophrenia and other psychotic disorders. In terms of developmental disorders, catatonia is mostly reported in patients with autism.,,, However, there are limited data on the association of catatonia with intellectual disability. The available literature on the association of catatonia with intellectual disability is in the form of case reports, and many of the patients reported have another psychiatric disorder or genetic abnormality as a cause of catatonia. In this report, we aim to describe a young female with intellectual disability who presented with catatonia, for which no other definite cause could be ascertained and her catatonia was successfully managed with electroconvulsive therapy (ECT).
| Case Report|| |
A 20-year-old single female presented to the psychiatry inpatient services with complaints of mutism and refusal to eat and drink. On mental status examination, she was found to have mutism, staring, rigidity, hypoactivity, waxy flexibility, negativism, refusal to eat or drink, and intermittent crying spells. Her Bush-Francis Catatonia Rating scale (BFCRS) score was 19. Detailed history revealed that she was born out of an unwanted pregnancy, and her mother had taken some abortion pills during the first trimester of pregnancy, but the abortion failed and resultantly the pregnancy was continued. During the second trimester, the mother had also developed mild fever, which lasted for about 5 days. She was born by forceps delivery and had delayed cry for about 5 min. Since early childhood, she had global delay in developmental milestones. Neck holding was achieved at 6–7 months, sitting without support at 8–9 months, and walking with a limp by 2 years. She could reach for objects at 6–7 months and turn pages of a book by 2 years. She could say mamma and papa at the age of 3 years. She was sent to school at the age of 5 years. At school, she would not interact with children of her class and was not able to keep pace with the class, and resultantly, she dropped out of school after class ninth, at the age of 14 years. At times, in school, peers would make fun of her. In every class, she was made to pass. However, from 12 to 19 years of age, she was able to take care of herself and was able to do minor household works, under the supervision of the family members.
Two months before presentation, over the period of 1–2 weeks, she developed symptoms in the form of decreased speech output with ultimately developing mutism, poor interaction, and staring. She would mostly remain confined to her bed and would keep on staring in one direction for hours together. She would resist all the efforts to move her. Her appetite and self-care deteriorated. Because of the continued symptoms, she was admitted to a local hospital, diagnosed with pyrexia of unknown origin, periventricular leukomalacia due to hypoxic–ischemic injury at birth, and possibly having acute disseminated encephalomyelitis. She was treated with injection ceftriaxone, dexamethasone (20 mg/day), and levodopa (187.5 mg/day). In the hospital, she had two episodes of generalized tonic–clonic seizures (GTCS) 24 h apart. Her electroencephalogram showed evidence of seizure activity, and magnetic resonance imaging (MRI) of brain showed hyperintensities in the periventricular region, suggestive of hypoxic–ischemic injury, but no space-occupying lesions were seen. She was treated with tablet levetiracetam (3 g/day). With these medications, her fever subsided and there was no recurrence of GTCS. However, her catatonic symptoms persisted as such and she started to remain irritable. Given the continuation of symptoms, she was referred to our center. Initially, she was evaluated by the neurology services and tablet levetiracetam was tapered off in view of persistent irritability, and she was started on tablet valproate (600 mg/day) and was referred to the emergency psychiatry services for the management of behavioral disturbances. At the psychiatry outpatient services, she was diagnosed with catatonia and lorazepam challenge test was done, with which her catatonic symptoms reduced (BFCRS came down from 19 to 8). She was investigated for possible underlying organic causes of catatonia. Investigations in the form of hemogram, liver function test, renal function test, serum electrolytes, thyroid function test, blood culture, urine routine and culture, electrocardiograph, X-ray chest posterior-anterior view, serum calcium, serum prolactin, glycated hemoglobin, serum dehydroepiandrosterone levels, serum testosterone levels, and cerebrospinal fluid analysis also did not reveal any abnormality. Her serum ceruloplasmin levels were also found to be in the normal range, and ophthalmology examination did not reveal Kayser–Fleischer ring. Her panel for inborn error of metabolism and porphyria did not reveal any abnormality. However, her Vitamin D3 (5.56 ng/ml; normal range <30 ng/ml) and folic acid (3.60 ng/ml; normal range 4.6–18.7 ng/ml) levels were found to be low. MRI of the brain revealed hyperintensities in the periventricular regions, suggestive of hypoxic–ischemic injury at birth. Diagnosis of catatonia (organic vs. psychotic illness) was kept. Autoimmune encephalitis panel was negative. In view of nutritional deficiencies, she was given tablet folic acid 5 mg/day and Vitamin D sachet (60,000 IU weekly).
After initial improvement with lorazepam, there was worsening of catatonia, despite giving lorazepam 10 mg/day in divided doses. This led to admission to the psychiatric inpatient unit. Given the persistent catatonic symptoms, the family was offered ECT, for which they agreed. After obtaining written informed consent from her mother, she was started on ECT and she received seven effective modified bitemporal ECTs, with which she showed gradual improvement in her catatonic symptoms, with BFCRS coming down from 19 to 0, at the end of the ECT course, without any complications related to ECT. In addition, she was started on tablet olanzapine 5 mg/day, in view of the possibility of a psychotic illness. While receiving ECT, she started swallowing solid food, drinking water, and walking with minimal assistance. Her symptoms of rigidity, negativism, and waxy flexibility disappeared. According to her family members, her ability to interact had returned to her baseline. For further management, tablet lorazepam (5 mg) and tablet valproate (600 mg/day) were continued. Her social quotient was found to be 52. She was discharged after 3 weeks of the inpatient stay. During the initial follow-up, tablet lorazepam and tablet valproate were tapered off, with no recurrence of seizures and catatonic features. On the third follow-up, there was worsening in the form of decreased speech output and poor interaction. Hence, olanzapine was increased to 7.5 mg/day after which she improved. As per family members, with continuation of olanzapine, her interaction improved, and they considered it to be better than her premorbid self. Olanzapine was continued thereafter. She maintained the improvement in all her symptoms, during the follow-up of the next 1 year. Later, a final diagnosis of catatonia possibly associated with intellectual disability was considered. The patient is being followed up further for monitoring and nonpharmacological treatment.
| Discussion|| |
In the index case, in the background of absence of any other psychiatric symptoms and lack of evidence for organic factors (except for a nonspecific history of fever), catatonia could possibly be attributed to the intellectual disability or nutritional deficiencies found at the time of assessment. Although Vitamin D deficiency has been reported to be associated with catatonia, the literature is very scarce and the type of association is not clear., In view of lack of clear evidence in the literature for association of catatonia and Vitamin D deficiency, and possible occurrence of nutritional deficiencies to be secondary to poor oral intake in the 2 month period before the presentation, and lack of persistent improvement with substitution of Vitamin D, catatonia cannot totally attributed to Vitamin D deficiency in the index case. Accordingly, this report suggests that catatonia may also be associated with intellectual disability alone or a combination of intellectual deficiency and Vitamin D deficiency. There are few reports of catatonia in the background of intellectual disability in children and adolescents.,,,,, However, there is limited information about catatonia in young adults and the elderly with intellectual disability., Available literature suggests that intellectual disability in itself cannot cause/lead to catatonia and it is invariably secondary to affective illness, psychotic illness, or medical condition. A study that focused on catatonia in children and adolescents suggests that many children and adolescents have genetic abnormalities, most commonly single nucleotide polymorphisms. There is limited information on the use of ECT in subjects with intellectual disability for various indications. A review of the literature on use of ECT in subjects with intellectual disability revealed 72 case reports, a single retrospective chart review, and a lack of controlled studies. Of the 72 cases, in only four cases, ECT was administered for catatonia in the background of autism and additional affective symptoms. Index case did not have any depressive features at the time of assessment, nor any history to support the diagnosis of autism. However, the catatonia improved with administration of ECT. Accordingly, the present report adds to the limited literature on the topic and suggests that ECT is safe and effective for managing symptoms of catatonia occurring in the background of intellectual disability.
Declaration of patient consent
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.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
Grover S, Sahoo S, Chakravarty R, Chakrabarti S, Avasthi A. Comparative study of symptom profile of catatonia in patients with psychotic disorders, affective disorders and organic disorders. Asian J Psychiatr. 2019;43:170-6.
Wing L, Shah A. Catatonia in autistic spectrum disorders. Br J Psychiatry 2000;176:357-62.
Wachtel LE, Griffin M, Reti IM. Electroconvulsive therapy in a man with autism experiencing severe depression, catatonia, and self-injury. J ECT 2010;26:70-3.
Wachtel LE, Hermida A, Dhossche DM. Maintenance electroconvulsive therapy in autistic catatonia: A case series review. Prog Neuropsychopharmacol Biol Psychiatry 2010;34:581-7.
Zaw FK, Bates GD, Murali V, Bentham P. Catatonia, autism, and ECT. Dev Med Child Neurol 1999;41:843-5.
Dealberto MJ. Clinical symptoms of psychotic episodes and 25-hydroxy vitamin D serum levels in black first-generation immigrants. Acta Psychiatr Scand 2013;128:475-87.
Jacobs J, Schwartz A, McDougle CJ, Skotko BG. Rapid clinical deterioration in an individual with Down syndrome. Am J Med Genet A 2016;170:1899-902.
Chanpattana W. Maintenance ECT in mentally retarded, treatment-resistant schizophrenic patients. J ECT 1999;15:150-3.
Cutajar P, Wilson D. The use of ECT in intellectual disability. J Intellect Disabil Res 1999;43 (Pt 5):421-7.
Lazarus A, Jaffe RL, Dubin WR. Electroconvulsive therapy and major depression in Down's syndrome. J Clin Psychiatry 1990;51:422-5.
Merrill RD. ECT for a patient with profound mental retardation. Am J Psychiatry 1990;147:256-7.
Guze BH, Weinman B, Diamond RP. Use of ECT to treat bipolar depression in a mental retardate with cerebral palsy. Convuls Ther 1987;3:60-4.
Puri BK, Langa A, Coleman RM, Singh I. The clinical efficacy of maintenance electroconvulsive therapy in a patient with a mild mental handicap. Br J Psychiatry 1992;161:707-9.
Desarkar P, Blumberger D, Daskalakis ZJ. Case report: Successful use of the combination of electroconvulsive therapy and clozapine in treating treatment-resistant schizophrenia and catatonia in an adult with intellectual disability. J Autism Dev Disord 2018;48:3637-40.
White M, Maxwell E, Milteer WE, de Leon J. Catatonia in Older adult individuals with intellectual disabilities. Case Rep Psychiatry 2015;2015:120617.
Benarous X, Raffin M, Ferrafiat V, Consoli A, Cohen D. Catatonia in children and adolescents: New perspectives. Schizophr Res 2018;200:56-67.
Raffin M, Consoli A, Giannitelli M, Philippe A, Keren B, Bodeau N, et al
. Catatonia in children and adolescents: A high rate of genetic conditions. J Am Acad Child Adolesc Psychiatry 2018;57:518-250.
Collins J, Halder N, Chaudhry N. Use of ECT in patients with an intellectual disability: Review. Psychiatrist 2012;36:55-60.