Tics as a Presenting Symptom in Catatonia

Catatonia is a complex psychomotor syndrome, including both hypokinetic (eg, catalepsy, mutism) and hyperkinetic features (eg, agitation, verbigeration, stereotypies, echophenomena). Catatonia is often observed in psychiatric conditions (eg, schizophrenia), neurological disorders (eg, N-methyl-d-aspartate receptor encephalitis), and neurodevelopmental conditions (eg, autism spectrum disorder [ASD]).1 Tics are defined as repetitive, stereotyped, nonrhythmic movements or vocalizations. Although some studies have suggested a potential relationship between tics and catatonic symptoms,2-4 tics are not generally recognized as a manifestation of catatonia, nor are they included in catatonia rating scales or the DSM-5 (Diagnostic and Statistical Manual of Mental Disorders, Fifth Edition) description.5, 6 We present 2 cases of patients with catatonia who initially presented with tics. Case 1: 13-year-old boy with autism and intellectual disability (very basic verbal abilities, unable to express detailed thoughts). Parents stated he never had clear tics in the past, only rather nonspecific “mild movements in keeping with his autism.” At age 12, he developed intermittent head and upper-limb tics. This increased explosively in severity to include repetitive facial scrunching, arm movements, back arching, pelvic thrusting, and chest protrusion. He could not explain why he performed the movements/presence of urge. He developed marked sleep disruptions (4 hours/night) and weight loss, and was unable to attend his special needs classroom. Clonidine resulted in sedation and mild reduction in tics. Subsequently, his tics worsened, and he began to have freezing behaviors (spending >30 minutes moving between rooms due to freezing, with repeated tics for extended periods, Video 1). Catatonia was suspected. The patient scored between 20 and 22 on the Bush–Francis Catatonia Rating Scale (excitement, staring, posturing, grimacing, echolalia, stereotypy, waxy flexibility, withdrawal, automatic obedience, ambitendency, perseveration).5 Clonidine was tapered, and lorazepam 8 mg/day led to improvement in tics and catatonia, without sedation. Due to reemergence of catatonia and tics 1 month later, lorazepam was increased to 18 mg/day, again resulting in improvement, without sedation. One year later, he experienced a reemergence of significant tics, associated with sleep/feeding disruption, but no freezing. Lorazepam was increased with little improvement. Aripirazole 7.5 mg daily resulted in improved tics and appetite. Lorazepam was slowly tapered to 9 mg/day. Case 2: a 55-year-old woman with high premorbid functioning and no prior psychiatric history developed erotomanic and persecutory delusions, along with motor (eg neck jerking) and vocal tics (Video 1), for which she described an uncontrollable urge, but no suppressibility. She was diagnosed with adult-onset tic disorder after an extensive workup (including lumbar puncture). There was no identifiable prior drug use/provoking factors, other than casual alcohol consumption, and cannabis use that started in her early forties. Her symptoms progressed to include verbigeration, grimacing, perseveration, and stereotypies. Family collateral indicated mutism, “walking around like she was in a stupor,” decreased oral intake, and withdrawal. She did not respond to treatment with several medications, including quetiapine and clozapine. There was mild response to lorazepam 6 mg/day, not increased to the point of sedation due to lack of robust response, clinician discomfort, and concern for worsening tics requiring alternate therapy. Consultation with a schizophrenia specialist suggested the presentation was in keeping with a psychomotor phenotype of schizophrenia described as periodic catatonia. Ultimately, she responded to electroconvulsive therapy (ECT), combined with lurasidone 40 mg/day, olanzapine 30 mg/day, and sertraline 200 mg/day. We describe 2 cases of patients with catatonia who initially presented with tics: improvement in tics occurred with treatment directed toward catatonia (lorazepam and ECT), which are not generally effective for typical tics. Catatonia treatment is reviewed in depth in other papers.1 The overlapping phenomenology of tics and stereotypies (including complex motor stereotypies/imagery) with the nonspecific symptoms of catatonia may make it difficult for clinicians to diagnose catatonia. For the same reasons, it is possible that tics are underreported in catatonia. The opposite situation would be to overdiagnose symptoms or conditions based on similarity in behavior phenomenology. For example, 1 study concluded that most patients with Tourette's syndrome exhibit features of catatonia4; however, it would be a mistake to infer diagnostic overlap simply based on behaviors that look similar on the surface. In addition, hypokinetic features seen in catatonia may resemble hypokinetic phenomena in tic disorders and other neuropsychiatric conditions. This includes tic “blocking phenomena,” which features a cessation of motor output.7 Blocking phenomena have been reported to co-occur with very severe tics in patients with Tourette's syndrome,8 and they have also been seen in patients with obsessive-compulsive disorder. Although it can be difficult to distinguish blocking from catatonia, if a patient has insight and the ability to describe it, one may elicit an urge or sensation like that described in tics for blocking tics, as opposed to hypokinetic catatonia that may be accompanied by other features of catatonia. Another hypokinetic presentation, obsessional slowness, characterized by disabling limited and slow movements/vocalizations, has been proposed as a potential forme fruste of catatonia.7 These observations may point to a spectrum of phenomenological overlap among neuropsychiatric conditions: movements appearing like tics can be seen in catatonia, whereas symptoms that look like certain features of catatonia can be seen in neurological and psychiatric presentations. This paper presents a novel perspective, suggesting that tics may be an under-recognized feature of catatonia. We suggest that practitioners in neurology and psychiatry consider the possibility of catatonia in patients with atypical presentations of tics, particularly when these occur in the context of ASD, psychosis, or other neuropsychiatric conditions in which catatonia is known to occur. (1) Research project: A. Conception, B. Organization, C. Execution; (2) Manuscript preparation: A. Writing of the first draft, B. Review and critique. L.M.V.: 1A, 1B, 1C, 2A, 2B N.I.: 1A, 1B, 1C, 2A, 2B A.W.: 1A, 1B, 1C, 2A, 2B E.A.-J.: 1A, 1B, 1C, 2B We thank the patients for their consent and contribution to the report, and Dr. Christos Ganos for his comments on the video for case 1 and for sharing articles about tic phenomenologies. Ethical Compliance Statement: An institutional review board review was not required for this study as we are reporting only 2 cases. Patient consent: verbal consent was expressly obtained in direct conversation, from both patients and their family members (parents: case 1; husband: case 2), with consent specific to how the case and video would be shown: including consent to show full face for case 1 and request for facial blurring in case 2. This was documented in the patient chart. We confirm that we have read the journal's position on issues involved in ethical publication and affirm that this work is consistent with those guidelines. Funding Sources and Conflicts of Interest: No specific funding was received for this work. The authors declare that there are no additional disclosures to report. Financial Disclosures for the Previous 12 Months: The authors declare that there are no additional disclosures to report. The data that support the findings of this study are available on request from the corresponding author. The data are not publicly available due to privacy or ethical restrictions.

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