Electroconvulsive Therapy for Catatonia in an 18-Year-Old Patient Presenting with Mixed Features of Schizophrenia and Obsessive-Compulsive Disorder

To The Editor:

Catatonia was first described in 1874 by Karl Kahlbaum, and in the American Psychiatric Association, Diagnostic and Statistical Manual of Mental Disorders, 5th ed. (DSM-5) is a specifier (not an independent disorder) requiring 3 of 12 possible symptoms, including: Stupor, catalepsy, waxy flexibility, mutism, negativism, posturing, odd mannerisms, stereotypies, agitation, grimacing, echolalia, and echopraxia (American Psychiatric Association 2013). Historically linked with schizophrenia, catatonia can also present in other mental disorders, as a result of other medical issues, and drugs. Notably, in situations where there is another mental disorder, as many as two thirds of patients with catatonia meet criteria for a mood disorder, often bipolar disorder (Weder et al. 2008).

Benzodiazepines are first-line treatments for catatonia, and lorazepam response is often considered diagnostically corroborative. Still, only 70% of cases remit with benzodiazepines (Hawkins et al. 1995), and a variety of treatments are now used as adjuncts, including amantadine (Northoff et al. 1997; Hervey et al. 2012), memantine (Thomas et al. 2005; Obregon et al. 2011), and zolpidem (Thomas et al. 1997). In refractory cases, the use of electroconvulsive therapy (ECT) is supported by a limited case literature, with initial remission rates of 75% and sustained recovery at 6 months in 46% of pediatric catatonia cases (Rey and Walter 1997).

In this report, we discuss a young patient admitted to our care following a 1.5-year-long course, whose catatonia responded well to ECT but who, nevertheless, continued to present diagnostic and management challenges.

Case Report

J., an 18-year-old Caucasian male, was transferred to our facility to initiate ECT for catatonia following two outside hospitalizations during the previous 5 weeks. On admission, J. was bedridden and on tube feeds, with stupor, waxy flexibility, mutism, negativism, and posturing. Prior to this, he had undergone extensive evaluation that included brain MRIs, electroencephalogram (EEG), lumbar puncture, a paraneoplastic antibody panel, transthoracic echocardiogram, and serologies for HIV, Herpes simplex virus (HSV), Toxoplasma gondii, Cryptococcus, and West Nile virus. None of these studies were revealing.

Prior to age 17, J. had had an unremarkable history. He first became symptomatic his junior year in high school, when he began having more difficulty making decisions, and withdrew socially. After treatment-emergent anxiety and suicidality on paroxetine, and with positive depressive scores on two mood scales, bipolar disorder became the leading diagnosis. To target bipolar depression, olanzapine, ziprasidone, venlafaxine, sertraline, and quetiapine were successively trialed in outpatient care, but with prohibitive side effects or nonresponse.

J.'s difficulties continued with a romantic breakup and plummeting grades. He mentioned, “My body doesn't feel right.” By spring of his senior year, J. exhibited compulsions, for example, repeatedly exiting and re-entering cars. Soon his deterioration became so severe that he stopped attending school. At this point, the differential diagnosis for J.'s presentation was expanded to include a primary psychotic disorder, obsessive-compulsive disorder (OCD), and encephalitis.

Hospital Course

Because J.'s mental status precluded his ability to consent, court-ordered ECT was secured with family support. Ongoing lorazepam was tapered to 1 mg orally twice a day to facilitate ECT, while adjunctive memantine (from the last hospital stay) was phased out, given its apparent lack of efficacy. Bitemporal ECT treatments were begun on a schedule of three sessions weekly: After the first session, J. showed dramatic improvement with cessation of waxy flexibility, and in his ability to answer questions and eat without tube-feeds. Although this initial transformation was encouraging, his improvement plateaued quickly. Although not catatonic, he had ongoing psychomotor retardation and pronounced cognitive impairment, with a Montreal Cognitive Assessment (MoCA) score of 20 out of 30 (Nasreddine et al. 2005).

J. continued to experience anxiety and decreased executive functioning, with blunted affect, alogia, and vague reports of hearing God and the Devil “arguing.” When he did converse, his speech was notable for extraneous conditionality (often a soft affirmative followed by a reciprocal soft negative). For example, when asked how he was feeling one day, he might answer, “I wanna say worse…or maybe the same.” He also began to demonstrate prominent compulsions, such as retracing steps because his feet “just didn't feel right.”

In the context of additional ECT sessions, several medications were introduced to augment J.'s recovery. Fluoxetine and olanzapine were initiated (as the combination had not yet been tried), but were discontinued after obsessive-compulsive behaviors increased. Clomipramine was used to target obsessive-compulsive behaviors, but was stopped when catatonic symptoms returned. Aripiprazole was tried next, and although J. briefly became more lucid and volitional, his catatonic symptoms eventually returned, and aripiprazole was discontinued after 6 weeks. Whether his regression was triggered by a given medication trial, or was a product of an ongoing requirement for ECT, was ambiguous.

By this time, our formulation had shifted to a primary diagnosis of schizophrenia. Although obsessive-compulsive traits were present, it was felt that this was more likely part of a unifying psychotic syndrome. Because J. regressed into catatonia when ECT was suspended with and without coadministration of clomipramine, we concluded that clomipramine had not provoked his earlier setback, and we successfully reintroduced and titrated this agent to 175 mg daily to target obsessive-compulsive symptoms (OCS).

Following cessation of aripiprazole, we started clozapine therapy to target J.'s treatment-refractory psychotic symptoms. Over the remainder of his 4 month inpatient stay at our facility, this was ultimately titrated to 450 mg daily, which he tolerated well.

After clozapine's initiation, J. demonstrated slow, fluctuating, but progressive improvement. Although he continued to exhibit psychomotor slowing (possibly a result of his obsessions and compulsions), his more extended episodes of “freezing” stopped, and he no longer demonstrated catatonic symptoms. Furthermore, the magnitude of his compulsions decreased with ongoing clozapine titration, despite earlier concerns raised by case reports (discussed subsequently) that clozapine might worsen them. J's conversation also demonstrated greater fluency and detail (with occasional interruptions for compulsive counting). J.'s parents were particularly happy with his improvement, making statements such as “J.'s back!” in the last few weeks of hospitalization. He did not require any further ECT sessions after his 18th, 2 months prior to his discharge.

Discussion

In this report, we have described the complicated course of an 18-year-old male with severe catatonia and psychotic symptoms. Initially, our differential diagnosis favored bipolar disorder, given the large number of catatonia cases associated with that diagnosis (Weder et al. 2008) and a history suggesting that J. had experienced some depression the year prior. However, the patient's subjective report and clinical examination all argued against an acute mood episode. Also, despite crippling obsessions and compulsions, a primary OCD diagnosis was considered unlikely given the presence of hallucinations and J.'s inability to elaborate on the concerns or motivations behind his compulsions. Ultimately, what seemed most diagnostically salient were J.'s heavy burden of negative symptoms, disorganized behaviors, and the hallucinations.

There is a growing literature on schizophrenia with co-occurring OCS, spawning questions about whether patients with both represent a special clinical subpopulation. Reports from the early twentieth century suggested that only 1–3.5% of patients with schizophrenia manifested OCS, whereas more recent studies have estimated 7.8–52%, depending upon how strictly one defines OCS (Bottas et al. 2005).

The “schizo-obsessive” construct has received attention before, and, in older nosologic systems, defied simple categorization into the now-antiquated dichotomy of neurosis versus psychosis (Insel and Akiskal 1986). With DSM-5, it still seems unsatisfactory to apply two diagnostic labels for what may more likely be a single disease process in this patient subpopulation. One explanation offered for the higher recent estimates of OCS in schizophrenic patients is that it is an iatrogenic consequence of using atypical antipsychotics, especially clozapine and olanzapine (Schirmbeck and Zink 2012). The rationale behind this hypothesis is that atypical antipsychotics antagonize serotonergic signaling, whereas the clinical efficacy of antidepressants that treat OCD is attributed to their ability to enhance serotonergic tone through reuptake inhibition (Zohar and Insel 1987). However, the widely variable serotonergic antagonism of atypical agents (Meltzer 2012) in conjunction with the complicated in vivo effects of serotonin reuptake inhibition (Artigas 2013) obfuscate simple pharmacologic explanations. It is also possible that both psychotic and obsessive-compulsive processes are mediated by abnormal function in similar brain regions, such as cortico-striato-thalamic circuitry. Interestingly, the literature is mixed on whether OCS in schizophrenia contributes to greater impairment and negative symptom burden, or is a mitigating factor (Bottas et al. 2005).

With J., OCS did not manifest until the summer prior to his hospitalization with us, after he had already had several months' treatment with olanzapine. However, the reappearance of these symptoms on our inpatient unit occurred just prior to his retrial with olanzapine, and persisted well after this agent was discontinued, making attribution of this symptom cluster to his medication alone questionable. Given J.'s already significant burden of OCS, the potential for exacerbation by clozapine was taken seriously, and hence this agent was initially avoided.

Although ECT resolved J.'s earlier catatonia, it did not appear to be effective against his remaining symptom clusters (e.g., his OCD, negative symptoms, hallucinations, and suicidal thoughts), which is similar to a separately reported case history (Hermesh et al. 1989). The addition of clozapine appeared to be the most definitive intervention after ECT, reducing J.'s symptom burden sufficiently to facilitate his return home. Questions remain about whether clomipramine contributed to this improvement, although we also cannot rule out that clomipramine may have helped blunt iatrogenic OCS caused by clozapine.

This and similar case reports highlight the need for greater characterization of symptoms and clinical course in patients with “schizo-obsessive” presentations, as well as a more systematic vetting of available interventions to delineate best evidence-based practices.