Somatic salvation in temporal lobe sclerosis: From resistance to remission with modified electroconvulsive therapy for dual neuropsychiatric symptom relief

INTRODUCTION

Neuropsychiatric disorders often pose diagnostic and therapeutic challenges, particularly when neurological and psychiatric symptoms overlap. Temporal lobe sclerosis, frequently associated with intractable seizures, may also present with mood disturbances.^[1] Standard pharmacological treatments can be limited by drug sensitivity or adverse effects. Modified electroconvulsive therapy (mECT), though underutilized in organic psychiatric conditions, may offer therapeutic benefit.^[2] We present a case of an adolescent with temporal lobe sclerosis secondary to seronegative autoimmune encephalitis (AE), with recurrent mania and treatment-resistant seizures, managed successfully with mECT. Written informed consent was obtained from the patient and his mother for this report.

CASE REPORT

An 18-year-old male (Master A) presented with irritability, grandiosity, pressured speech, aggression, and decreased sleep for 1 week, following a cluster of seizure episodes. He was admitted with a Young Mania Rating Scale (YMRS) score of 44, showing classical manic symptoms. He was already on multiple antiepileptics (brivaracetam, clobazam, and lamotrigine) and was started on risperidone (up to 8 mg/day), oxcarbazepine, and lorazepam. Extrapyramidal symptoms required trihexyphenidyl and an atropine rinse. He also developed hyponatremia due to oxcarbazepine. Despite pharmacological efforts, he had another seizure, and his manic symptoms worsened with minimal improvement. He had no family history of mood disorder or any other psychiatric illness.

His history revealed a diagnosis of seronegative AE with anti-thyroid peroxidase antibodies, hypothyroidism, previously treated with azathioprine, intravenous steroids, and cyclophosphamide. A past manic episode treated with divalproex had led valproate-induced hyperammonemia. Electroencephalogram (EEG) showed rhythmic temporal slowing, and magnetic resonance imaging revealed hippocampal atrophy with parahippocampal hyperintensities and cerebellar atrophy as shown in Figure 1.

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Figure 1:

Magnetic resonance imaging brain findings suggest hippocampal atrophy with prominent temporal horns and T2/fluid-attenuated inversion recovery hyperintensities in parahippocampal areas and diffuse cerebro-cerebellar atrophy (orange arrows).

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Given poor medication response (drop in YMRS score from 44 to 33) and recurrent symptoms, mECT was initiated using the Niviqure SA/VR system after coordination with the multidisciplinary team and shared decision-making with the family. Initial sessions were ineffective due to Anti epileptic drugs (AED) interference. Adjustments included switching anesthesia to etomidate and pre-electroconvulsive therapy (ECT) hyperventilation, leading to effective seizures. After six sessions, the YMRS score reduced from 33 to 15, with no further seizures. He was discharged on risperidone, oxcarbazepine, azathioprine, clobazam, lamotrigine, and brivaracetam. Maintenance mECT was continued bimonthly. Oxcarbazepine (up to 600 mg/day) was continued as a mood stabilizer with Risperidone tapered down to 4 mg/day as no disruptive behavior was seen at home; there were no manic symptoms except fluctuating overtalkativeness and overfamiliarity, which was not distressing and not disabling in terms of the patient’s functioning. Over the next 2 years, the patient remained in remission from mania and seizures, with functional recovery.

DISCUSSION

Organic mania with seizure disorder is often characterized by atypical, pleomorphic, and intermittent symptoms, often complicated by the clustering of seizure episodes.^[1] Intractable seizure disorder has various causes, including structural abnormalities, perinatal injuries, neurometabolic disorders, etc. Such cases can have diagnostic and management difficulties and require a multidisciplinary approach.^[2] Individuals with such neuropsychiatric disorders are susceptible to adverse drug reaction (ADR), as reported in our case.^[3] With the chronic course of intractable seizures, there is a high risk of the development of neuropsychiatric manifestations of epilepsy, such as personality changes, that can further complicate the clinical management. In our case, intractable seizures, poor control of manic symptoms, intolerable ADR, multiple hospitalizations, and frequent visits to the emergency department due to the disruptive behavior were challenging for the treating team as well as burdening the family’s resources, creating therapeutic burnout initially.

Inducing a seizure with mECT is a relatively safer and controlled alternative to modulate gamma-aminobutyric acid levels in the brain, which further inhibits ongoing seizures by raising the seizure threshold, prolonging the refractory period, altering the neural metabolism, and activating inhibitory interneurons.^[4] Evidence suggests using ECT in seronegative AE, but in catatonic presentations, unlike in our case.^[5,6]

Seizure frequency and manic symptoms improved after mECT; hence, to ensure a better, speedier, and more effective control of symptoms, mECT could be recommended as a safer option in acute as well as maintenance therapy to provide a lower risk of clustering of seizures and the associated manic symptoms.

Although ECT is well used in refractory status epilepticus, we also find its utility in reducing seizure frequency in intractable epilepsy. There is no evidence toward the ECT protocol for intractable epilepsy in AE, which could guide the clinician on the type of polarity of ECT, charge delivered, frequency administered, total number of sessions, and effective duration of treatment. In our case, a total of 14 mECT sessions spread over 20-week duration (six sessions over the 4 weeks of inpatient stay and eight outpatient sessions - bimonthly over the next 16 weeks) reduced the frequency and clustering of seizures from 3 times a week to 1–2 times a month (that too brief, partial seizures lasting for a few seconds). Therefore, based on our case, we substantiate the nine biweekly sessions of ECT in the acute phase, followed by five bimonthly sessions with a charge of 360 mC, in young individuals with such presentation. The area remains open for further exploration as one of the modalities of treatment in intractable epilepsy with intolerable ADR to AED. Despite the stigma on the use of ECT in psychiatric disorders, several earlier reports endorse the use of ECT in epilepsy, which can improve the quality of life and give time to clinicians for further treatment optimization and diagnostic workup.

CONCLUSION

Recurrent mania in the case of seronegative AE with intractable epilepsy is a rare presentation and poses challenges for clinical management. ECT is a safer alternative and can be considered earlier in the course of illness, making it a success in such cases. Future research should focus on developing guidelines for ECT in neuropsychiatric illnesses to be adapted by a consultation liaison psychiatrist.