(022) Catatonia: Where to Look When ECT Fails

Background: It is well accepted that catatonia has a high prevalence in autoimmune encephalitis and there is support in the literature for incorporating ECT as standard treatment for these patients when immunomodulation therapies are insufficient (Tanguturi, 2019). We present the case of a patient with a history of bipolar I disorder who presented with catatonia refractory to ECT and aggressive medical management. Due to her prior psychiatric history, an autoimmune etiology was not seriously considered at the OSH prior to embarking on ECT. This case report highlights the importance of considering autoimmune catatonia regardless of psychiatric history; and proposes further research into a potential link between anti-basal ganglia antibodies and autoimmune catatonia.

Case: Ms. H is a 65-year-old Caucasian female with psychiatric history of bipolar disorder who presented to an OSH with severe catatonia that was refractory to medical management. The cause of her catatonia had been attributed to her bipolar disorder after LP and MRI were noted to be unremarkable. She was admitted to our facility for ECT and on admission her BFCRS score was 37. Her catatonia remained refractory, despite a course of 15 ECT treatments with concurrent high dose lorazepam augmented with trials of memantine, amantadine and carbidopa-levodopa. On the end of her ECT course, her BCFRS score was 33. At this point we reconsidered her diagnosis of catatonia secondary to a primary psychiatric process and began working through a broader differential including autoimmune entities and disorders of the basal ganglia. Notable results included CSF pleocytosis, borderline GAD 65 antibody on Mayo ENC2 which was not felt to be causative. We also obtained serum analysis of anti-basal ganglia antibodies using the Cunningham panel. Ms. H had elevated titers of anti-D1 receptor antibodies and anti-tubulin antibodies. It was decided to initiate treatment with IVIG at 0.4 gram/kg/day for 5 days followed by 5 sessions of plasmapheresis. Ms. H improved rapidly and her BFCRS was 3 on discharge.

Discussion: Our case confirms that successful treatment of catatonia must take into account the underlying etiology of the catatonia. For Ms. H, standard treatment of catatonia was ineffective until the autoimmune process was appropriately addressed. Interestingly, she never had a positive antibody on the Mayo ENC 2 panel identified, yet her catatonia responded robustly to immunomodulation. Diagnostically, probable seronegative autoimmune encephalitis would be the most unifying diagnosis. In the literature, cortico-striato-thalamo circuits are implicated in the pathophysiology underlying catatonia (Northoff, 2002), which is why we opted to look for presence of anti-basal ganglia antibodies. The Cunningham panel is well known in the PANDAS literature but further research is needed to explore its utility in patients with catatonia caused by seronegative autoimmune encephalitis.
Northoff G. (2002) What catatonia can tell us about "top-down modulation": a neuropsychiatric hypothesis. Behav Brain Sci. 25(5):555-77; discussion 578-604.
Tanguturi, Y., Cundiff, A., & Fuchs, C. (2019) Anti-N-Methyl d-Aspartate Receptor Encephalitis and Electroconvulsive Therapy: Literature Review and Future Directions, Child and Adolescent Psychiatric Clinics of North America, 28(1), 79-89