Unexpected Emergence of Catatonia Following Electroconvulsive Therapy in Early Onset Bipolar Disorder

INTRODUCTION Catatonia is a psychomotor syndrome. The condition may be caused by dysfunction of neurotransmitters such as Gamma-aminobutyric acid-A (GABA-A), glutamate, and dopamine.[1] It is well known that benzodiazepines are the first line of treatment for catatonia. In patients who do not respond to benzodiazepines, electroconvulsive therapy (ECT) is the treatment of choice and is also recommended by many treatment guidelines.[2] However, here, we report catatonia signs emerging following sessions of ECT. There are only four case reports and a case series (four cases) reporting this unexpected and rare phenomenon which is contrary to the current literary evidence on ECT and catatonia. CASE REPORT After obtaining informed consent, we report a case of a 20-year-old high school dropout with bipolar affective disorder and lithium induced hypothyroidism, who has been experiencing multiple episodes of depression and mania since the age of 13 years. Despite treatment, she has not responded to multiple antipsychotics (up to 8 mg of risperidone, 20 mg of olanzapine, 1200 mg of amisulpride, and 800 mg of quetiapine) and mood stabilizers, indicating treatment resistance. ECT and medication optimization on amisulpride and quetiapine have helped her remain stable for nearly past 2 years. However, she developed breast tenderness and galactorrhea due to drug-induced hyperprolactinemia and severe constipation. During an attempt to taper the medications to minimize the above-mentioned side effects, she presented in a manic episode with irritability, excessive talking, abuse, dis-inhibition, decreased need for sleep, increased appetite, and poor self-care. Modified ECT was planned due to the patient’s response to previous episodes. However, challenges in securing an IV line for antipsychotics and anesthesia due to her obesity and irritable mood made it difficult. We stabilized her using ketamine and dexmedetomidine infusion and was sedated for 4 h before the next session to prevent pulling off the cannula. Thiopentone sodium, succinylcholine, and atropine were administered as anesthetic premedication before each session. After fourth ECT, she remained unresponsive for 5–6 h. Due to prolonged unresponsiveness, ketamine and dexmetadometine infusions were not administered. Following fifth ECT, she experienced unresponsiveness for 5 days with staring look, minimal verbal output, low-grade fever, tremulousness, and 2 min crying episodes. After ruling out differentials as per Table 1, a trial of lorazepam 6 mg was given and showed improvement within 5 min, confirming the diagnosis of catatonia. We discontinued ECT and lorazepam was tapered over a week until optimal improvement was noted.Table 1: Investigations to rule out other possible conditionsThe patient’s symptoms persisted and she was unmanageable, leading to the decision to start her on clozapine, which showed a significant improvement but also caused hypersalivation and constipation. DISCUSSION Here, we discuss about how catatonia might paradoxically appear while receiving ECT. Pandey and Sharma refuted the anesthesia premedication hypothesis in 1988 by demonstrating catatonia development after discontinuing premedication and utilizing direct ECT. Our case showed no epileptiform changes on electroencephalography, thereby also eliminating the possibility of temporal lobe epilepsy.[3] A 2001 case series suggested recent benzodiazepine cessation may increase the risk of catatonia during ECT. They reported a patient with a history of hypothyroidism and past history of catatonia. Our patient did have hypothyroidism but, was not on benzodiazepine and has no previous or family history of catatonia.[4] According to Shams et al., increased blood–brain barrier permeability following ECT may have resulted in elevated central nervous system (CNS) haloperidol levels, which were proposed as antipsychotic-induced catatonia.[5] This paradox of catatonia following ECT was also discussed in a paranoid schizophrenic patient suggesting this as a variant of drug induced catatonia and attributed it to brain perfusion changes after ECT.[6] A case report from Iran in 2018, showed catatonia after ECT in a case of bipolar disorder. The probable etiology they suggested was a Vitamin B12 deficiency and elevated concentrations of a long-acting antipsychotic in the CNS. They also discussed the literature on brain perfusion changes after ECT in mood disorders, schizoaffective disorders and catatonia.[7] Our patient received ketamine and dexmedatomedine infusions. Ketamine, though initially widely reported as a drug-inducing catatonia, off late there have been reports of it having been used to treat catatonia. This is in light of new knowledge with regard to the role of glutamate and N-methyl-D-aspartate receptor activity in catatonia.[8] In 2023, Ketigian and Kidambi proposed low dose ketamine as a cause for postoperative catatonia. However, their patient also received opioids, resulting in opioid-associated pinpoint pupils. Thus, suggesting, opioid and ketamine may have a complex mechanism in causing catatonia.[9] Our patient was off infusion after the fourth ECT. However, more noticeable symptoms of catatonia continued to appear. Therefore, ketamine can be an unlikely cause in our case. Recent advances in functional neuroimaging have helped us understand that ECT brings about changes in cerebral blood flow and glucose metabolism. It is also demonstrated that repeated administration of ECT causes transient disruption in the blood-brain barrier due to increased blood pressure during the ictal phase. This facilitates leakage of certain neurochemicals, causating changes in the levels of brain-derived neurotrophic factor, angiogenesis, and neurogenesis.[10] Clinicians should be aware of potential catatonia following ECT, especially in patients on long-standing antipsychotics, and further research is needed to understand this rare condition. 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 Nil. Conflicts of interest There are no conflicts of interest.