catatonic schizophrenia case study

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Introduction, case report, considerations.

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The Curious Case of a Catatonic Patient

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John H. Enterman, Dyllis van Dijk, The Curious Case of a Catatonic Patient, Schizophrenia Bulletin , Volume 37, Issue 2, March 2011, Pages 235–237, https://doi.org/10.1093/schbul/sbq110

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Catatonia is a syndrome characterized by the coexistence of psychiatric and motor symptoms. 1 It is associated with a wide range of psychiatric, medical, neurological, and drug-induced disorders. 2 The concept of catatonia was first described by the German psychiatrist Kahlbaum in 1874. 3 It is more frequently found among patients diagnosed with mania, depression, and neurotoxic syndromes than among those with schizophrenia. Yet, it is mainly classified as a form of schizophrenia. 4 The exact cause of catatonia has not been elucidated.

The syndrome of catatonia is defined by the objective presence of motor signs, over 40 of which have been described. These catatonic signs are listed in table 1 . There is no agreed threshold for the number or duration of symptoms that should be present to justify a diagnosis of catatonia. Research has suffered from this, and studies can rarely be compared with confidence. 7

Principal Features of Catatonia 5 , 6

There are consistent clinical reports that benzodiazepines are effective in acute catatonia syndromes, particularly stuporous conditions, but no placebo-controlled randomized studies have been published. 8 , 9 However, benzodiazepines are the drugs of choice for catatonia. 10 In most cases, lorazepam is administered parenterally or orally beginning with 3 mg/d and increasing rapidly to effective resolution. Dosages of 20–30 mg/d are occasionally necessary. 5 Patients who are unresponsive or insufficiently responsive to benzodiazepines need electroconvulsive therapy (ECT). 5 , 10

Patient A is a 28-year-old male of Mediterranean origin diagnosed with paranoid schizophrenia at the age of 23. He was hospitalized several times due to psychotic episodes characterized by religious delusions and auditory and visual hallucinations. He is living in an assisted living facility, where the medication is offered to the residents, but where they have to take it by themselves. He uses cannabis daily and does not use any other substances. Drug history mentions the use of risperidone and flupentixol decanoate, the latter since 2008 up to the present. At the end of 2008, he developed a progressive condition in which he showed less mimicry, staring, negativism, mutism, and immobility. There were no signs of autonomic dysregulation, such as increased body temperature or unstable blood pressure.

Because catatonia was assumed in April 2009, he was orally treated with lorazepam, starting at 2 mg a day. The lorazepam dose was increased based on the clinical state until 40 mg a day without any subjective or objective effects. He was admitted to the psychiatric ward to receive parenterally administered lorazepam up to 60 mg daily. After 2 days, there still was no measurable effect nor was there any effect on his consciousness. We resumed oral treatment with lorazepam 40 mgs daily and patient agreed to undergo ECT. During the lorazepam and ECT treatment, the patient continued to receive 30 mg of flupentixol decanoate every 2 weeks. After 3 ECT sessions (Mecta 5000, bilateral, 1 ms, 40 hz 2 s, 128 mC, 800 mA, [a relatively low, common, dosage]), the catatonic signs receded rapidly and patient refused to take the lorazepam, because “he was cured.” He soon afterward developed an acute catatonic state, in which he was found completely immobile next to his bed. He received lorazepam immediately and ECT the following days. After 2 more ECT sessions, the catatonic signs receded again. During the weeks afterward, patient received 40 mgs lorazepam daily, which was reduced and finally stopped on his demand.

A few months afterward, patient presented to the acute psychiatric service with signs of acute dystonia (cervical dystonia and dysphagia). He was treated with biperiden 2 mg and the dystonia almost immediately disappeared. Flupentixol decanoate dosage was lowered to 20 mgs every 2 weeks. Patient denied the use of any drugs except cannabis and urine examination confirmed this. After this episode, patient experienced several other episodes of dystonia, each time successfully treated with biperiden 2 mgs.

This case has many remarkable features. To begin with, the simple fact of a slowly progressive, during multiple months, catatonic state emerging elicited our curiosity. We could not relate it to a mood disorder nor to excessive cannabis use. Then again, the administration of doses of lorazepam up to 60 mg per day without any effects whatsoever seems remarkable, especially in the case of a young man not habituated to benzodiazepines. Of interest to those practicing ECT is the remarkable fact that the quality of the ECT did not suffer under the administration of high doses of benzodiazepines. We used the dosage titration method to determine the energy level needed for the ECT. We chose to temporarily halt the action of the lorazepam with the administration of 0.5 mg of flumazenil i.v. immediately prior to the ECT and achieved a therapeutically sufficient convulsion at a relatively low energy level. After 2 ECT sessions in this manner, we chose to try a treatment session without the use of flumazenil. This had no influence on the energy necessary for the ECT; on the contrary, we obtained a convulsion of the same length and electroencephalogram waveform as we did using the flumazenil, at precisely the same energy level. After the fifth treatment session, the patient did not return for further treatment sessions, in spite of his incomplete remission and in spite of his having been warned of the possibility of relapse. He was observed to be in worse condition in his home, but he himself seemed to be less distressed by his condition than his caregivers did, in spite of the many observations that catatonia is usually accompanied by anxiety. Because of the outpatient situation, there were limitations according to physical and blood examinations and the medication intake during the (acute) catatonic state of our patient. We have had our doubts of his acceptance of the benzodiazepines, but during his stay on the ward, the administration has been closely supervised by trained psychiatric nursing staff. Unfortunately, we did not determine a plasma level of benzodiazepines. Other laboratory results were unremarkable.

Should we consider other diagnoses than catatonia, and, if so, which? Perhaps a syndrome caused by cannabis consumption?

Does such a diagnosis explain the progression, over months, of the “catatonic-like” state?

How is the absence of an effect on the necessary ECT energy level by benzodiazepines to be explained?

How should we interpret the absence of distress?

Have we used the correct treatments or should we have had other considerations?

Submissions should be sent to the email address as listed in the author information. Any outcome will subsequently be published in this journal.

The Authors have declared that there are no conflicts of interest in relation to the subject of this study.

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Brain Motor Region Diffusion Tensor Imaging in Patients with Catatonic Schizophrenia: A Case-Control Study

Affiliations.

• 1 Geha Mental Health Center, Petah Tikva, Israel.
• 2 Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel.
• 3 Laboratory of Biological Psychiatry, Felsenstein Medical Research Center, Petah Tikva, Israel.
• 4 Department of Psychosis Studies, Institute of Psychiatry, Psychology and Neuroscience, King's College, London, UK.
• 5 Department of Diagnostic Imaging, Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel.
• 6 Advanced Technology Center, Sheba Medical Center, Tel Hashomer, Israel.
• 7 Department of Diagnostic Imaging, Sheba Medical Center, Tel Hashomer, Israel.
• PMID: 34672443

Background: Only a small proportion of schizophrenia patients present with catatonic symptoms. Imaging studies suggest that brain motor circuits are involved in the underlying pathology of catatonia. However, data about diffusivity dysregulation of these circuits in catatonic schizophrenia are scarce.

Objectives: To assess the involvement of brain motor circuits in schizophrenia patients with catatonia.

Methods: Diffusion tensor imaging (DTI) was used to measure white matter signals in selected brain regions linked to motor circuits. Relevant DTI data of seven catatonic schizophrenia patients were compared to those of seven non-catatonic schizophrenia patients, matched for sex, age, and education level.

Results: Significantly elevated fractional anisotropy values were found in the splenium of the corpus callosum, the right peduncle of the cerebellum, and the right internal capsule of the schizophrenia patients with catatonia compared to those without catatonia. This finding showed altered diffusivity in selected motor-related brain areas.

Conclusions: Catatonic schizophrenia is associated with dysregulation of the connectivity in specific motoric brain regions and corresponding circuits. Future DTI studies are needed to address the neural correlates of motor abnormalities in schizophrenia-related catatonia during the acute and remitted state of the illness to identify the specific pathophysiology of this disorder.

• Cerebellum / diagnostic imaging
• Cerebellum / physiopathology
• Connectome / methods
• Corpus Callosum / diagnostic imaging
• Corpus Callosum / physiopathology
• Correlation of Data
• Diagnostic and Statistical Manual of Mental Disorders
• Diffusion Tensor Imaging / methods*
• Internal Capsule / diagnostic imaging
• Internal Capsule / physiopathology
• Motor Cortex* / diagnostic imaging
• Motor Cortex* / physiopathology
• Psychiatric Status Rating Scales
• Schizophrenia, Catatonic* / diagnosis
• Schizophrenia, Catatonic* / physiopathology

• Case report
• Open access
• Published: 06 July 2018

Acute catatonia on medical wards: a case series

• Elisabeth Doran 1 &
• John D. Sheehan 1  

Journal of Medical Case Reports volume  12 , Article number:  206 ( 2018 ) Cite this article

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Catatonia is a behavioral syndrome which presents with an inability to move normally. It is associated with mood disorders and schizophrenia, as well as with medical and neurological conditions. It is an expression of the severity of the underlying condition.

The awareness of catatonia among general medical doctors and even psychiatrists is poor. It is often seen as an historical diagnosis. Because of this, catatonia is often not recognized. If patients in catatonic states are not diagnosed, their condition is likely to progress with a risk of increased morbidity and potentially fatal outcomes.

Case presentation

We present a case series of three acutely unwell, frail, elderly medical patients (a 65-year-old Irish woman, a 75-year-old Irish woman, and a 68-year-old Irish woman) with a background of longstanding well-controlled psychiatric illnesses, who developed acute catatonia while being treated for medical conditions in a general medical in-patient setting.

Conclusions

Catatonia is common in acute medical settings but is underdiagnosed due to the low awareness of the condition among both general medical doctors and psychiatrists. Within a short time period, we diagnosed and successfully treated three acutely unwell patients in acute medical settings. We would like to increase the awareness of catatonia among medical doctors.

Peer Review reports

Catatonia is a behavioral syndrome which presents with an inability to move normally. It is associated with mood disorders and schizophrenia, as well as with medical and neurological conditions. The presence of catatonia denotes the severity of the underlying illness. Acute medical and neurological conditions, as well as drug withdrawal and toxic drug states, can trigger catatonia [ 1 ].

According to Diagnostic and Statistical Manual of Mental Disorders , Fifth Edition (DSM-5), catatonia is associated with a mental disorder and is diagnosed when the clinical picture is dominated by at least three of the following: stupor, catalepsy, waxy flexibility, mutism, negativism, posturing, mannerisms, agitation, stereotypy, grimacing, echolalia, or echopraxia (Table 1 ) [ 2 ].

The awareness of catatonia among general medical doctors and even psychiatrists is relatively poor, and it is often seen as an historical diagnosis [ 3 ]. Due to this low level of awareness catatonia is likely to be underdiagnosed. If patients in catatonic states are not diagnosed their condition is likely to progress with a risk of increased morbidity and a potentially fatal outcome. As catatonia is treatable, timely diagnosis and treatment significantly improves patient outcomes. Patients suffering from catatonia are at high risk of developing medical complications such as dehydration, malnutrition, deep vein thrombosis, pulmonary embolism, pressure ulcers, contractures, constipation, urinary tract infections, and aspiration pneumonia [ 4 ].

Between December 2015 and June 2016 three cases of catatonia were diagnosed and successfully treated in the Mater Misericordiae University Hospital (MMUH), Dublin. We feel that it is not well known among medical doctors, that patients, particularly if they have a history of psychiatric illness, can be at risk of becoming catatonic when acutely unwell.

All patients or next of kin of the patients have provided written informed consent for the publication of their cases.

The first case is of a 65-year-old Irish woman with a background of schizoaffective disorder, which had been stable in recent years, and a medical history of chronic renal failure, type 2 diabetes mellitus, atrial fibrillation, arterial hypertension, previous stroke with a right arm contracture, and aortic stenosis. For her schizoaffective disorder she was on a risperidone depot and escitalopram 20 mg once a day. She was admitted medically in December 2015 to the MMUH with a urinary tract infection, acute renal failure, and deranged international normalized ratio (INR).

The Liaison Psychiatry service was consulted shortly after admission. The family gave a collateral history of low mood in our patient since her brother had become ill 2 months earlier and her dose of antidepressant had been increased a month earlier. On review, she was at her baseline mental state, engaging well in conversation and denying low mood, which was confirmed by the community mental health nurse, to whom the patient was well known. No changes were made to her management.

A week later the neurology service was asked to review the patient due to altered level of consciousness. On examination she presented with waxy flexibility, negativism, new onset increased tone of her left arm, posturing, and catalepsy. Her mobility had deteriorated, with selective speech, mute episodes, and poor oral intake noted by medical staff over the preceding day. The impression was that she was suffering from acute catatonia. An magnet resonance imaging (MRI) of her brain showed no acute changes. Nasogastric (NG) feeding was established to ensure oral intake.

The psychiatry service was again consulted, and acute catatonia was confirmed. She was diagnosed as having schizoaffective disorder with catatonia, as per DSM-5 (Table 2 ). A trial of lorazepam was advised for the treatment of catatonia. The dose was titrated to 3 mg per day. The dose was well tolerated and her mental state improved significantly over the following 2 weeks. She became verbally interactive again and returned to her baseline verbal interaction; her mood was euthymic and tone normalized. She was discharged to her own home at a physical baseline that compared to her pre-admission physical state and had remained so at 6-month follow-up. The likely cause for this episode of catatonia was thought to be her medical deterioration.

The second case is a 75-year-old Irish woman with a psychiatric history of bipolar affective disorder, stable for several years on olanzapine and valproate, enabling her to lead an independent lifestyle. There was no history of cognitive impairment. She suffered from multiple medical conditions including: atrial fibrillation, type 2 diabetes mellitus, obstructive sleep apnea, and a recent mitral valve repair complicated by postoperative delirium.

She was admitted medically to a rural Irish hospital in November 2015 for management of a raised INR. During the admission she developed sudden onset left-sided weakness and altered levels of consciousness, as well as rigidity and one isolated temperature spike. The concern was raised that she may be or might have been suffering from neuroleptic malignant syndrome and her neuroleptics were stopped as a precaution (Table  3 ). She was transferred to the intensive care unit (ICU) in the MMUH in Dublin with a suspicion of neuroleptic malignant syndrome or encephalopathy. Computed tomography (CT) brain imaging was normal at the time. As neuroleptic malignant syndrome was suspected, olanzapine was stopped. However, her creatinine kinase levels were normal as was her body temperature. Hence, neuroleptic malignant syndrome was deemed to be unlikely. An electroencephalogram during admission showed changes suspicious of encephalopathy and MRI imaging showed no acute abnormality. A working diagnosis of metabolic encephalopathy was established but extensive investigations yielded no cause for the encephalopathy.

Due to prolonged altered levels of consciousness and unexplained altered mental state, the Liaison Psychiatry service was consulted in January 2016.

On examination, she responded with a mouthed single word greeting, but made no other attempt at verbal interactions. She inconsistently followed the examiner with her gaze, but stared out of the window for most of the examination. On physical examination she presented with waxy resistance to passive movement and psychomotor retardation. The impression was that these features were most likely related to a catatonic exacerbation of her bipolar affective disorder, in the absence of an organic explanation. She was diagnosed as having bipolar I disorder with catatonia as per DSM-5 (Table 2 ).

Delirium was raised as a differential diagnosis (Table 3 ), but she had been reviewed in September 2015 by the Liaison service, when she was delirious after her valve replacement and her presentation was distinctly different on that occasion.

She was initially treated with intravenously administered lorazepam, but became drowsy, with a significant drop in Glasgow Coma Scale (GCS). As such the treatment was abandoned. Instead, olanzapine was cautiously reintroduced, which led to a significant improvement in her mental state within days. On follow-up review, she was mildly confused but engaged well at interview, and was euthymic with no evidence of thought disorder or movement disturbance. Subsequently she was discharged back to her own home. She was not reviewed at 6-month follow-up as she was living in a rural area and was followed up in her local service.

Of note, in 2017, the same patient was readmitted to the MMUH ICU, from the same peripheral hospital, in a very similar state to the presentation in November 2015. Again her neuroleptics had been stopped when she was acutely unwell and she developed typical traits of acute catatonia. She was trialled on lorazepam, which she did not tolerate and reinstitution of her neuroleptics brought no improvement. The therapy was then escalated to electroconvulsive therapy (ECT), to which she had a dramatic response and significant improvement of her mental state.

The third case is of a 68-year-old Irish woman who presented to the MMUH in April 2016 with acute laryngitis. She had a background of bipolar affective disorder which had been stable for the past 30 years on monotherapy with lithium. There had been a recent history of lithium toxicity secondary to a deterioration of her renal function, which had been managed at her local psychiatric hospital. After the episode, she had been restarted on a low dose of lithium as well as a low dose of valproate.

On presentation to the MMUH she was initially treated jointly by the ear, nose, and throat (ENT) team and medical team and was managed in an ICU environment due to respiratory compromise. She had no oral intake for multiple days. Once stabilized she was transferred to an acute medical ward but an acute onset confusional state with bizarre behavior was noted over a period of 2 days. Due to her psychiatric history the Liaison Psychiatry service was consulted. On review she was severely thought disordered and confused. She was only able to produce a word salad and showed echolalia. She had motor retardation, increased tone, negativism, and posturing on examination. The impression was that she was suffering from acute catatonia. Brain imaging did not reveal acute abnormalities. She was diagnosed as having bipolar I disorder with catatonia as per DSM-5 (Table 2 ).

Advice was given to treat her with paliperidone. Her mental state improved slightly as a result, but she remained severely thought disordered and confused for 2 weeks. Eventually, lithium was cautiously reintroduced under close monitoring of her renal function. The reintroduction of lithium was well tolerated and she improved significantly over a 2-week period. At discharge she was no longer thought disordered, she was well orientated, and back to her fully independent baseline. She continues to live independently to date.

Discussion and conclusions

In all three cases, the catatonia was caused by an acute medical deterioration, and in two of the cases there was also a withdrawal of longstanding psychiatric medication secondary to the acute illness.

The treatment of catatonia recommended by the Maudsley Guidelines [ 5 ] is a challenge with lorazepam. If that is not successful, then ECT may be an alternative option. Use of antipsychotics has been controversial.

Our first patient responded well to treatment with lorazepam. Benzodiazepines are the recognized first-line treatment for catatonia [ 5 , 6 ]. As she was on a depot preparation of her longstanding antipsychotic mediation she did not experience withdrawal of her medication when she became medically unwell and her oral intake diminished, unlike the other two cases.

In the second case, the patient had deteriorated suddenly; due to a suspicion of neuroleptic malignant syndrome, which was subsequently ruled out, her longstanding maintenance medication olanzapine was stopped. She received an extensive medical work-up and catatonia only became a suspicion and was diagnosed after a prolonged intensive care stay of over a month. When she was diagnosed as having catatonia, first-line treatment with lorazepam was trialled, but not tolerated, probably due to her frail physical state. As a second option, and following multidisciplinary discussions with medical and neurological colleagues, we opted for the cautious reintroduction of olanzapine, which had been held since there was a suspicion of neuroleptic malignant syndrome. This management proved to be effective for the patient and she recovered. Interestingly, the same patient re-presented with catatonia 2 years later and did not respond to olanzapine but required ECT.

In the literature there is clear advice against using first-generation antipsychotics in catatonia, as these have been shown to exacerbate the condition [ 7 , 8 ]. The use of second-generation antipsychotics is controversial, but there are a number of case reports showing good outcomes for patients when treated with second-generation antipsychotics. Cassidy et al. [ 9 ] described a patient with bipolar affective disorder who responded selectively to high-dose olanzapine and Babington and Spiegel [ 10 ] reported on a similar case, in which a catatonic patient did not respond to lorazepam, but showed dramatic reduction of catatonic features with a combination of olanzapine and amantadine. Valevski et al. [ 11 ] described two patients who responded well to risperidone. There are multiple positive reports on the treatment of catatonia with clozapine, but due to the slow introduction and close monitoring required with clozapine this may only be an option limited to chronic, severe, and treatment-resistant cases [ 12 , 13 , 14 ].

The third case is slightly more unusual. The patient had been well maintained on lithium monotherapy for many years, but it had been reduced to a sub-therapeutic level due to renal impairment, preceding her medical illness. Due to her inability to swallow she did not receive any of the psychotropic medications she was on at the time of admission. She relapsed with pronounced psychotic features, but also obvious catatonic features. Due to the acute change in her mental state, she was diagnosed very early on in her catatonic state. Consequently, the decision was made to treat the psychotic features with a second-generation antipsychotic that could be safely used in chronic renal impairment. She showed some improvement in her mental state with this regime, but only recovered fully when the decision was made to reintroduce the lithium that had kept her stable for the past 30 years. The reintroduction of lithium was a difficult decision due to her comorbidities and history of lithium toxicity, but there was a consensus among the treating physicians and psychiatrists that in her case the benefits were likely to outweigh the risks of the treatment. This decision was made in agreement with her relatives by explaining that there was some evidence that showed mood stabilizers to be beneficial in the treatment of catatonia [ 15 ]. The decision proved to be very beneficial for the patient and she agreed with the action herself once she became well enough to have insight into her episode of catatonia.

In all three cases catatonia was diagnosed as per DSM-5. In DSM-5, catatonia can either be diagnosed in the context of a general medical disorder or as a specifier for a major psychiatric illness [ 1 ]. All our patients have well-controlled major psychiatric illnesses. Our impression in all cases was that the catatonic episodes were probably triggered by the acute medical illness rather than an exacerbation of the psychiatric illness. This particular circumstance is not reflected in DSM-5. Patients with major psychiatric illnesses are probably more prone to reacting to acute medical illness with catatonic states than patients who do not suffer from psychiatric illnesses, for this reason it is important for medical doctors to be aware of catatonia.

All three patients improved significantly once diagnosed and treated but the treatment varied with each patient. All three improved physically and mentally to such a point that discharge home was made possible. Recognition and prompt diagnosis of catatonia is crucial, since outcomes for treated catatonia are very good, but untreated catatonia may lead to chronic morbidity and can be fatal.

In conclusion catatonia is a severe psychomotor syndrome which has a good prognosis if diagnosed and treated in a timely manner. Acute medical deterioration can trigger catatonia, particularly if patients have a history of mental illness. It is important to be aware of catatonia and have a high index of suspicion when longstanding psychiatric medications have been stopped in the course of the medical management or in patients with reduced oral intake. As these patients are likely to be under the care of a medical team, rather than a psychiatrist, it is important to increase awareness of catatonia among physicians and encourage prompt treatment.

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Both authors were involved in the patients’ care, including diagnosing and managing the patients’ conditions. ED collected the patient information and drafted the article. JS revised the article critically for intellectual content. Both authors approved the final version of the article. ED and JS take public responsibility for appropriate portions of the content and are accountable for all aspects of the work.

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Correspondence to Elisabeth Doran .

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JS has been a Consultant in Liaison Psychiatry for many years and has experience and qualifications in Medicine, Psychiatry, and Psychotherapy.

ED complete 1 year of training in psychiatry and is currently training in neurology, but also has experience in geriatric medicine.

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Doran, E., Sheehan, J.D. Acute catatonia on medical wards: a case series. J Med Case Reports 12 , 206 (2018). https://doi.org/10.1186/s13256-018-1714-z

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Received : 18 January 2018

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DOI : https://doi.org/10.1186/s13256-018-1714-z

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CASE REPORT article

Two sides of the same coin: a case report of first-episode catatonic syndrome in a high-functioning autism patient.

• 1 Department of Psychiatry and Psychotherapy, Central Institute of Mental Health, Medical Faculty Mannheim, University of Heidelberg, Mannheim, Germany
• 2 Institute of Physiology and Pathophysiology, University of Heidelberg, Heidelberg, Germany

Background: Catatonic phenomena such as stupor, mutism, stereotypy, echolalia, echopraxia, affective flattening, psychomotor deficits, and social withdrawal are characteristic symptoms of both schizophrenia and autism spectrum disorders (ASD), suggesting overlapping pathophysiological similarities such as altered glutamatergic and dopaminergic synaptic transmission and common genetic mutations. In daily clinical practice, ASD can be masked by manifest catatonic or psychotic symptoms and represent a diagnostic challenge, especially in patients with unknown or empty medical history. Unclear diagnosis is one of the main factors for delayed treatment. However, we are still missing diagnostic recommendations when dealing with ASD patients suffering from catatonic syndrome.

Case presentation: A 31-year-old male patient without history of psychiatric disease presented with a severe catatonic syndrome and was admitted to our closed psychiatric ward. After the treatment with high-dose lorazepam and intramuscular olanzapine, catatonic symptoms largely remitted, but autistic traits persisted. Following a detailed anamnesis and a thorough neuropsychological testing, we diagnosed the patient with high-functioning autism and catatonic schizophrenia. The patient was discharged in a remitted state with long-acting injectable olanzapine.

Conclusion: This case represents an example of diagnostic and therapeutic challenges of catatonic schizophrenia in high-functioning autism due to clinical and neurobiological overlaps of these conditions. We discuss clinical features together with pathophysiological concepts of both conditions. Furthermore, we tackle social and legal hurdles in Germany that naturally arise in these patients. Finally, we present diagnostic “red flags” that can be used to rationally select and conduct current recommended diagnostic assessments if there is a suspicion of ASD in patients with catatonic syndrome in order to provide them with the most appropriate treatment.

Schizophrenia and autism spectrum disorders (ASD) are common, highly heritable, and severe neurodevelopmental disorders with overlapping genetic, neurobiological, and clinical characteristics. Schizophrenia is a complex brain disorder with an average prevalence of about 0.6% ( 1 ). ASD are classified as pervasive developmental disorders in the Diagnostic and Statistical Manual of Mental Disorders, Fifth Edition (DSM-5) with an estimated prevalence of 0.6–1% in the general population ( 2 ). The core symptoms of ASD are impairments in social interaction, impairments in communication, and restricted as well as repetitive behaviors ( 3 ). ASD are usually diagnosed in childhood or adolescence. If the patient exhibits flamboyant psychotic and catatonic symptoms, clinicians are confronted with considerable difficulties in diagnosing both disorders. Due to potentially life-threatening consequences, fast diagnostic workup is required in order to provide correct treatment.

From a clinical perspective, catatonic phenomena are nosologically unspecific and involve complex affective, motor, and behavioral symptoms ( 4 , 5 ). According to International Classification of Diseases (ICD-10), catatonia can only be diagnosed within the group of schizophrenia spectrum disorders or within the organic catatonic disorder. However, the majority of studies in recent years have made no distinction between the different forms of schizophrenia ( 6 ). The new classification systems DSM-5 and International Classification of Diseases (ICD-11) have also abandoned the traditional differentiation into paranoid, catatonic, hebephrenic, and disorganized subtype. DSM-5 classifies catatonia as a specifier in order to characterize other mental disorders presenting with catatonic features. Catatonia in the ICD-11 (https://icd.who.int/dev11) will be listed as an independent entity belonging to the mental, behavioral, or neurodevelopmental disorders. However, such discrepancies in the definition of catatonic syndrome might confound the diagnosis and delay appropriate treatment of patients presenting with severe catatonia. In this case report, we will discuss current evidence-based practice guidelines for accurate assessment, treatment, and management of catatonic syndrome in adult patients with high-functioning ASD.

Case Presentation and Clinical Assessment

A 31-year-old Caucasian male patient with catatonia was admitted to our closed psychiatric ward. In the emergency contact, he was disoriented as to the situation, time, and place; confused; anxious; and mutistic. Besides incoherently expressed psychotic fears of poisoning and other incoherent phrases, he was not open for exploration. Most information was gathered from his accompanying parents. According to them, he had never moved out but had been living his whole adult life in the basement of their house. Usually socially withdrawn and very calm, his behavior had changed rapidly approximately 5 days prior to admission toward agitation, repetitive movements, verbal and physical aggression, and sexual disinhibition. The patient had no prior personal or family psychiatric history and no history of drug abuse. Besides being underweight (Body Mass Index = 18.4 kg/m 2 ), he was in good physical health and had never taken medication.

On the ward, the patient initially refused water, food, and medication. Remaining in bed in a rigid posture, appearing confused and anxious, and avoiding eye contact and any kind of communication, he exhibited classic psycho-motoric symptoms of catatonia such as stupor, waxy flexibility, and mutism. The Positive and Negative Syndrome Scale (PANSS) ( 7 ) on admission added up to 148 points; the Northoff Catatonia Rating Scale (NCRS) ( 5 ) added up to 30 points in total, indicating severe psychosis and catatonia, respectively (see also Figure 1B ). Due to a sudden state of agitation, the patient had to be temporarily restrained. Initial blood tests, clinical examination, and cranial magnetic resonance tomography ( Figure 1A ) were without significant pathological findings. In particular, no gross abnormalities (e.g., tumor, space-occupying cystic lesion greater than 3 mm, signs of bleeding, contusion, infarction, and major gray or white matter lesions) were found. Due to reduced health conditions of the patient (dehydration, fever, and elevated CreatinKinase) on day 3, treatment with intravenous lorazepam and electrolyte solutions was initiated under physical constraints to prevent malignant catatonia. This led to stabilization of the patient’s state with partial remission of catatonic symptoms. Although some level of communication could be achieved, the patient, however, still refused any kind of treatment and demanded discharge. Therefore, his father was installed as legal guardian; the patient was involuntarily committed to treatment according to German law, and coerced antipsychotic medication with olanzapine and lorazepam was legally approved. Administration of oral olanzapine 20 mg daily over the course of 2 weeks and a subsequent intramuscular injection of 300 mg of its depot formulation resulted in further remission ( Figure 1B ) and allowed for a more in-depth exploration and neuropsychiatric testing.

Figure 1 Clinical time course. (A) Patient’s MRI, sample horizontal planes (3 mm) at the height of ventricles, taken on a 3-Tesla scanner. Upper image: Turbo-Inversion Recovery-Magnitude (TIRM) sequence with fluid suppression; bottom: T2 blade sequence. (B) The time course of NCRS and PANSS scores is depicted on the upper and the lower subplots, respectively. The patient was independently scored at five time points throughout treatment and follow-up (as marked on the upper x axis) by the two first authors of the paper; then, the mean score was calculated. Big cyan-filled circles represent the total scores; small red, blue, and green dots represent the subdomains of the tests. Major therapeutic and diagnostic milestones are marked on the bottom x axis. Treatment 1–4: 1, enforced medication with lorazepam and isotonic infusions; 2, enforced medication with lorazepam and oral olanzapine 10–15 mg daily; 3, enforced medication with intramuscular olanzapine 300 mg biweekly; 4, voluntary medication with intramuscular olanzapine 300 mg biweekly.

Five weeks after admission, the patient began to leave his room for dinner and ward rounds if requested. However, he never addressed someone actively. He was fully oriented and did not show any positive psychotic symptoms. Psychomotorically, he was calm and slow. He barely showed emotions, his affect was flat, and his voice and body language were monotonous. Any kind of communication, however, evoked apparent unease in the patient, accompanied by vegetative arousal signs such as sweating and tachycardia. In conversations, he never held eye contact. He still insisted on discharge, stressing out his will to return to his usual daily activities undisturbed by others. The patient admitted having been in critical conditions on admission, but rejected disease-related medical explanations. He refused voluntary medication, repeating that all it required to maintain his health was good food and his daily routine, which mostly comprised playing computer games, watching television, and cooking for himself. He reported an inverted circadian cycle of nightly waking and sleeping from early in the morning to late in the afternoon, so—despite living under one roof—he would rarely interact with his parents and younger sister. He would not leave the house for weeks except for short walks. He did not work and relied financially on his parents who also did all the shopping and housework. He reported not having had any friends or social contacts outside the family for 3 to 4 years. He never had a sexual relationship, albeit being sexually aroused by women, e.g., on television. Asked about his future plans, the patient seemed to be fully convinced that his current living situation would continue unchanged for an indefinite amount of time and considered this positive. When aging and potential mortality of his parents were addressed, he seemed not to be able to conceptualize this possibility. When asked about his parents’ and sister’s ages, he missed it by more than a decade (estimating them younger), albeit being fully oriented and exactly knowing his own age and date of birth.

The patient was born in Germany in a middle-class family. According to his parents, he was a healthy, normal-weight infant without any complications during pregnancy or birth ( via naturalis ). No infections of mother or child were memorable. His motor development was retarded; reportedly, he started to walk at the age of 2. Later, he would have coordinative difficulties to prepare bread or to walk downstairs. The speech development was reportedly unaltered. At the age of 3, he attended kindergarten. After the birth of his younger sister (by 5 years), he would have reacted “properly” and care for her. At the age of 7, he was sent to school. His teacher in the first grade advised the parents to put him in a special school due to his “odd” behavior, but they refused. During kindergarten and school, he was reported to have a hard time finding friends. The patient passed middle school with mediocre grades. According to himself, he did not like a special subject, but was bad at math and reading. He acquired a driver’s license at 18. After school, he finished a full training for a computer technician but did not attend the final exams. According to his parents, this was a breaking point in the patient’s life. He withdrew socially and ceased to adapt to social norms. He was dismissed from an internship at a warehouse due to inability to cope with the social structure at work and disrespect toward supervisors. Until clinical presentation, he had been living in the basement without much contact to the outer world. Interestingly, a stress situation could be described, which might have contributed to the acute psychotic state. The family reported that a renovation took place in the patient’s basement. Therefore, he had to move temporarily upstairs, and his daily routine was disrupted. Asked about this situation, he reported that the inability to withdraw and to follow his routines evoked feelings of anger and fear of change.

We performed a series of neuropsychological tests on the patient. In contrast to the clinical impression and his education, he performed poorly on the Intelligence Structure Test (IST-2000R) IQ test ( 8 ), reaching an overall IQ of 74 ( Table 1.1 ). However, the patient worked extremely slowly, over-carefully and anxiously, not proceeding adequately, leaving the majority of tasks uncompleted. He showed a substantial lack of motivation and worked only on tasks corresponding to his interests, leaving everything else out. Those items that he completed were solved correctly. Therefore, the results might underrepresent his intelligence. Furthermore, the Marburger Beurteilungsskala zum Asperger Syndrom (MBAS) ( 9 ), the Structured Clinical Interview for DSM-IV-TR (SCID II) ( 10 ), and a facial emotion recognition test were carried out. In the MBAS, the patient reached the cutoff for high-functioning ASD ( Table 1.2 ). He could identify all emotions except for anger ( Table 1.4 ). The patient underwent Autism Diagnostic Observation Schedule (ADOS-IV) after the partial remission of psychotic symptoms. This module is used with adolescents and adults with fluent speech ( 11 ). In this testing, he fulfilled the criteria for ASD and showed deficits mainly in communication and social interaction. The total score of ADOS-IV was 17 points ( Table 1.5 , A4 = 1; A8 = 2; A9 = 2; A10 = 1; B1 = 2; B2 = 2; B6 = 2; B8 = 2; B9 = 1; B10 = 1; B11 = 1; D1 = 1; D2 = 2; D4 = 1; and D5 = 1) and he reached the cutoff criterion for ASD. Autism Diagnostic Ineterview (ADI-R) was not performed. Still, the patient’s parents were interviewed extensively about his development.

Table 1 Neuropsychological measures.

Continued lack of patient’s therapeutic adherence and legal restrictions resulted in an interruption of therapy with olanzapine depot. Within a few weeks after discontinuation, the patient returned to a mutistic state, mostly refused to communicate, to leave his bed, and to participate in any social activities. He re-expressed high levels of anxiety and psychomotor agitation accompanied by vegetative symptoms. Resumption of forced medication with biweekly intramuscular olanzapine depot led once again to rapid remission. To encourage the patient to engage in social activities, we installed a behavioral plan where participation in ward rounds, ergotherapy, and meals was rewarded by computer time. In cooperation with psychologists and social workers, we strongly involved the parents in the therapeutic process. After multiple extensive psychoeducational sessions, the patient started to accept his biweekly medication. He was discharged in good physical and mental condition after 101 days of treatment (Figure 1B). Currently, he biweekly visits our outpatient office for medication and clinical controls and has been in remission for over a year.

To the best of our knowledge, we present the first case report of an adult male patient with an acute catatonic syndrome on the background of high-functioning ASD. The patient’s clinical presentation and autobiography clearly fulfill the diagnostic criteria of high-functioning ASD. He exhibited profound deficits in socio-emotional behavior and nonverbal communication, had rigid daily routines, and was involved in very few specific social activities. According to his parents, these traits have already been present in early childhood. Despite these psychosocial deficits, he managed to finish middle school, pass the driver’s exam, and receive an Information Technology (IT) education. This is also in line with previous evidence. Although ASD are lifelong neurodevelopmental disorders, the improvement of the autistic core symptoms in the course of the disease is a frequent phenomenon. People with autism and normal intelligence adapt to social and interactional demands as they grow older. This means that the autistic core symptoms might improve across childhood and adolescence ( 6 , 12 ). However, the occurrence of comorbid psychiatric diseases such as depression, anxiety disorders, or psychoses can lead to a further deterioration of the autistic core symptoms because the compensatory mechanisms no longer function. In our case, the current psychotic exacerbation associated with catatonic syndrome appeared to be triggered by disruption of his daily routines and unusual exposure to social stress and stimuli. Typically triggered by environmental stress, psychotic episodes are a known phenomenon in ASD with prevalence between 4% and 20% ( 13 – 15 ). Catatonic syndrome is also described in ASD with a similar prevalence of about 17% ( 16 ). However, we believe that the patient’s biography and clinical course cannot be explained by a single psychotic episode, but rather by the diagnosis of catatonic schizophrenia according to ICD-10 or schizophrenia with catatonia as a specifier according to DSM-5 ( 17 , 18 ). A clear breaking point can be observed in the patient’s biography, marked by a sudden onset of psychotic symptoms including disorganization, negative symptoms, and psychomotor abnormalities, causing a severe drop in the patient’s social functionality far below his previous level. This is a typical manifestation age of schizophrenia spectrum disorders, which can be followed by an extended prodromal phase without apparent positive symptoms ( 19 ). The sudden onset in the young adult patient also makes the diagnosis of schizotypal personality disorder highly unlikely, which usually manifests gradually during adolescence ( 20 ). Furthermore, the exacerbation of psychotic symptoms associated with catatonic syndrome upon disruption of antipsychotic therapy in contrast to stable state under medication hints towards catatonic schizophrenia according to ICD-10. In the last two decades, several studies pointed towards possible co-occurrence of both diseases, especially if ASD had been diagnosed during childhood ( 21 ). Volkmar et al. ( 22 ) found that about 1% of patients with ASD develop schizophrenia spectrum disorders. The prevalence of catatonia in adolescent and young adult ASD patients is described as ∼10% ( 23 ). Remarkably, some authors argue that catatonic symptoms and schizophrenia in children with ASD are different manifestations of a single underlying form of brain pathology—a kind of “iron triangle” of symptomatology—rather than three separate illnesses ( 24 ). In the last two decades, a growing number of studies have reported ASD with comorbid catatonic syndrome ( 23 , 25 – 28 ). However, only two case reports described high-functioning ASD patients exhibiting catatonic syndrome. Ellul and colleagues described a child ( 29 ) and Takaoka et al. reported a woman with high-functioning ASD ( 30 ). However, the overall risk of catatonic syndrome or schizophrenia in ASD is unknown. The currently reported low prevalence may be due to the fact that ASD prior to onset of schizophrenia may be strongly unrecognized ( 31 ). Indeed, positive, negative, and motor symptoms may occur in both conditions and therefore present a diagnostic challenge ( 32 – 34 ). Intriguingly, according to the results of a parental interview, about 50% of patients with schizophrenia spectrum disorders retrospectively fulfilled ASD criteria ( 35 ). This staggeringly high co-occurrence is not surprising if one takes into account the pathophysiological similarities between these conditions. Although there has been an increase in ASD diagnoses in recent years, the increasing prevalence rates are mainly related to children or adolescents with early childhood autism and mental retardation. ASD patients with normal intelligence often become manifest in adulthood by a different psychiatric diagnosis as their life circumstances or needs change. Along with clinical overlaps, both schizophrenia and ASD are considered as neurodevelopmental disorders and manifold overlap in their genetics, pathophysiology, and symptoms to an extent that some authors discuss them as entities on one disease spectrum ( 36 ). Thus, pregnancy-related complications were identified as significant risk factors for both conditions ( 37 ). Common genetic risk factors for both ASD and schizophrenia include genes encoding neurotrophic factors such as neurexin-1 ( 38 ) and synaptic proteins such as PSD-95 ( 39 ) and copy number variations in several genomic regions (e.g., 1q21, 16p11.2, and 22q11) regulating synaptic structure and function ( 40 ). These mutations are also successfully employed to model these diseases in rodents ( 41 , 42 ). Both conditions furthermore share various pathophysiological endophenotypes including altered evoked auditory potentials and sensorimotor gating deficits ( 40 , 43 ), imbalances in the excitatory–inhibitory activity ( 44 ), and overlapping findings in imaging studies such as altered gray matter volumes within the limbic–basal ganglia loop system ( 45 , 46 ). In particular, it has been shown that basal ganglia are important structures in the pathogenesis of ASD, because of their crucial role in motor and cognitive functioning ( 47 ). Thorough examination enables the clinical practitioner to distinguish between these conditions in unclear cases such as the one presented. Neuropsychologically, patients with ASD were shown to be predominantly impaired in executive functioning tasks such as planning/organization and flexibility of thought, whereas patients with schizophrenia had impairments in a wide range of intellectual abilities ( 48 ). Furthermore, patients with schizophrenia showed significantly larger motor coordination deficits (neurological soft signs) as compared to ASD patients ( 46 ). Additionally, the patient’s history, especially careful parental anamnesis, can provide crucial clues to differentiate between ASD and schizophrenia or both even in an adult, unknown patient with no history of disease.

Concluding remarks

In unknown adult patients presenting with catatonic syndrome, ASD need to be considered among potential differential diagnoses. Physicians need to be aware that both conditions may co-occur in one individual. Despite diagnostic and therapeutic challenges, thorough examination and meticulous anamnesis can lead to correct diagnosis and facilitate treatment. Even when typical autistic features are missing due to severe catatonic syndrome, ASD should be considered, even early in the diagnostic process in order to correctly classify the interactional difficulties with such patients. Elicitation of incongruous or unusual psychopathological findings should be followed up by careful psychometric and neuropsychological examination as well as by case history through a third party (e.g., parents or siblings).

Clinical Warning Signs That Enable Earlier Diagnosis of Autism Spectrum Disorders, Therapy, and Better Prognosis

● Behavioral features such as repetitive and/or stereotyped patterns of movement, clumsy gait, poor muscle tone, imbalance, impairment of motor skills, abnormal involuntary movements, monotonous voice and body language, mutism, and negativism

● Sudden onset of catatonic and/or psychotic symptoms following stressful life events or sensory overload (CAVE: hyper- and/or hyposensitivity to external stimuli)

● Difficulties to maintain a conversation with others and orienting to people in a social environment due to impairments in social interaction, language, and related cognitive skills

● Limitations with maintaining turn-taking in interactions with others and difficulties in responding to bids for interaction often leading to vegetative arousal signs such as sweating, tachycardia, and internal unrest

Consent for publication

The father of the patient gave his written consent for information about his son to be published in a scientific journal. He understood that the information will be published without his son’s name attached, but that full anonymity cannot be guaranteed. He understood that the text and MRI images published in the article will be freely available on the Internet and may be seen by the general public. The MRI images and text may also appear on other websites or in print and may be translated into other languages or used for commercial purposes. The father has been offered the opportunity to read the manuscript. A copy of the written consent is available for review by the editor of this journal.

Ethics Statement

Not applicable. Written informed consent was obtained from the patient and his legal guardian for publication of this case report and any accompanying images. A copy of the written consent is available for review by the editor of this journal.

Author Contributions

DHe and CT equally contributed to this work. DHe and CT performed the treatment, neuropsychological tests, took the patient history and third-party anamnesis, and wrote the paper. PG supervised all steps of diagnoses and treatment and wrote the paper. DHi supervised neuropsychological testing and wrote the paper.

This work was supported by the Physician Scientist Program of the Medical Faculty of the Heidelberg University. We acknowledge financial support by Deutsche Forschungsgemeinschaft within the funding program Open Access Publishing, by the Baden-Württemberg Ministry of Science, Research and the Arts and by Ruprecht-Karls-Universität of Heidelberg.

Conflict of Interest Statement

The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.

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Keywords: autism, catatonia, schizophrenia, diagnostic challenge, acute psychiatry, coercive treatment

Citation: Hefter D, Topor CE, Gass P and Hirjak D (2019) Two Sides of the Same Coin: A Case Report of First-Episode Catatonic Syndrome in a High-Functioning Autism Patient. Front. Psychiatry 10:224. doi: 10.3389/fpsyt.2019.00224

Received: 21 October 2018; Accepted: 26 March 2019; Published: 12 April 2019.

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*Correspondence: Peter Gass, [email protected]

† These authors have contributed equally to this work.

Disclaimer: All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article or claim that may be made by its manufacturer is not guaranteed or endorsed by the publisher.

• Case report
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• Published: 13 March 2018

A case report of schizoaffective disorder with ritualistic behaviors and catatonic stupor: successful treatment by risperidone and modified electroconvulsive therapy

• Yuanhan Bai 1 ,
• Xi Yang 1 ,
• Zhiqiang Zeng 1 &
• Haichen Yang 1  

BMC Psychiatry volume  18 , Article number:  67 ( 2018 ) Cite this article

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Ritualistic behaviors are common in obsessive compulsive disorder (OCD), while catatonic stupor occasionally occurs in psychotic or mood disorders. Schizoaffective disorder is a specific mental disorder involving both psychotic and affective symptoms. The syndrome usually represents a specific diagnosis, as in the case of the 10th edition of the International Classification of Diseases (ICD-10) or the 5th edition of the Diagnostic and Statistical Manual of Mental Disorders (DSM-5). However, symptom-based diagnosis can result in misdiagnosis and hinder effective treatment. Few cases of ritualistic behaviors and catatonic stupor associated with schizoaffective disorder have been reported. Risperidone and modified electroconvulsive therapy (MECT) were effective in our case.

Case presentation

A 35-year-old man with schizoaffective disorder-depression was admitted to the hospital because of ritualistic behaviors, depression, and distrust. At the time of admission, prominent ritualistic behaviors and depression misled us to make the diagnosis of OCD. Sertraline add-on treatment exacerbated the psychotic symptoms, such as pressure of thoughts and delusion of control. In the presence of obvious psychotic symptoms and depression, schizoaffective disorder-depression was diagnosed according to ICD-10. Meanwhile, the patient unfortunately developed catatonic stupor and respiratory infection, which was identified by respiratory symptoms, blood tests, and a chest X-ray. To treat psychotic symptoms, catatonic stupor, and respiratory infection, risperidone, MECT, and ceftriaxone were administered. As a result, we successfully cured the patient with the abovementioned treatment strategies. Eventually, the patient was diagnosed with schizoaffective disorder-depression with ritualistic behaviors and catatonia. Risperidone and MECT therapies were dramatically effective.

Making a differential diagnosis of mental disorders is a key step in treating disease. Sertraline was not recommended for treating schizoaffective disorder-depression according to our case because it could exacerbate positive symptoms. Controversy remains about whether antipsychotics should be administered for catatonic stupor. However, more case studies will be needed. Risperidone with MECT was beneficial for the patient in our case.

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Diagnosis is the first step toward correctly curing disease. Unlike internal or surgical diseases, mental disorders are largely symptom-based diagnoses [ 1 , 2 ]. In the process of interviewing, syndromes are always associated with certain diagnoses according to the ICD-10 or DSM-5. Repetitive behaviors or ritualistic behaviors may be linked with OCD [ 3 ]. Immobility, mutism, negativism, and peculiar motor behavior represent catatonic stupor [ 1 , 2 ], which is a psychotic diagnosis because approximately 10–15% patients with catatonic stupor meet the criteria for schizophrenia [ 4 ]. Typical ritualistic behaviors and catatonic stupor may represent OCD and psychotic disorders, respectively. However, these patients are not “textbook” cases, which means that complex and complicated symptoms may lead to misdiagnosis. Schizoaffective disorder is a specific mental disorder involving both psychotic and affective symptoms [ 5 ]. It is classified as “schizophrenia, schizotypal, and delusional disorders” by ICD-10 [ 1 ] and “schizophrenia spectrum and other psychotic disorders” by DSM-5 [ 2 ]. The complex symptomatology of schizoaffective disorder makes it highly likely that patients will be misdiagnosed.

Sertraline is a selective serotonin reuptake inhibitor [ 6 ] that is used to treat depression and OCD [ 7 ]. Previous studies have summarized the effective use of antidepressants in schizoaffective disorder [ 8 ]. However, the risk of exacerbation of positive symptoms of antidepressants should be considered [ 9 , 10 , 11 ]. A review of the treatments for catatonia has shown that MECT is effective, while antipsychotics remain controversial [ 12 ]. On the other hand, Huang et al. published other papers suggesting that the Lorazepam-Diazepam protocol can rapidly and safely relieve catatonia in schizophrenia, mood disorder, and organic lesions [ 13 , 14 , 15 ].

In this paper, we present the case of a patient who was initially suspected of having OCD but who actually suffered from schizoaffective disorder-depression with ritualistic behaviors and catatonic stupor. Sertraline exacerbated the psychotic symptoms. The ritualistic behaviors that were actually secondary to psychotic symptoms may have prevented us from making an accurate diagnosis. Finally, risperidone and MECT were effective strategies for this patient.

The patient was a 35-year-old male. He was apparently normal before the age of 27 years without any medical problems. He was intellectually normal and worked as a security guard. He was never married and had no children, often living with his older sister. He had been smoking for approximately 10 years or more, denying alcohol or other psychoactive substance abuse. He is the third child, with one older brother and one older sister. His father often became drunk and violent, going outside for no reason, and committed suicide many years ago. There were no detailed records for his father because he failed to see a doctor.

In 2009, the patient gradually became depressive; showed diminished pleasure, insomnia, and fatigue; and was unwilling to talk to others. Meanwhile, he developed delusions of persecution and reference, which made him believe that someone had insulted him and planned to kill him without evidence. Later, he came to the outpatient clinic of our hospital and was prescribed paroxetine 20 mg/d and sulpiride 0.2 g/d. He took these drugs irregularly, with minimal improvement in depressive symptoms and delusions. In August 2013, the patient was sent to the hospital for schizoaffective disorder-depression. During the following month, with quetiapine 600 mg/d and lithium carbonate sustained-release tablet 0.6 g/d, his depressive and positive symptoms improved. Taking these drugs, he almost enjoyed normal life and work. Unfortunately, he discontinued these medications in May 2017. Again, the patient gradually developed fear of the sound of water, a lack of any pleasure and negative ideas, and claimed that he could not trust anyone. Furthermore, the patient performed ritualistic behaviors, such as walking with a specific order. Once again, he was sent to our hospital by his older sister. At the time of admission, the patient presented depressive symptoms as well as ritualistic behaviors and distrust.

Upon admission, liver and kidney function, routine blood test, computed tomography (CT) of the head, and electrocardiograph (ECG) were normal. Depressive symptoms, delusions, and ritualistic behaviors were found upon psychiatric interview. By day 9 in the hospital, we followed outpatient therapeutic strategies with quetiapine 100 mg/d and lithium carbonate sustained-release tablet 0.6 g/d, observing that the depressive symptoms and distrust had moderately improved. However, the ritualistic behaviors gradually worsened. Before waking up, he would swing his arms up and down four times, did sit-ups four or five times, and sat at the edge of the bed for a few minutes, all of which took him approximately 8 min. These disturbed or interrupted behaviors made the patient anxious. Because of the predominant ritualistic behaviors, OCD was suspected first. Here, we wanted to reduce these behaviors by adding sertraline at a dose of 50 mg/d and titrating it to 100 mg/d.

Then, the symptoms further worsened, and the patient developed agitation, pressure of thoughts, and delusion of control. He felt that his ritualistic behaviors gradually became out of control, realizing that “unknown thoughts” and “a black shadow” affected his mind. Meanwhile, he felt sad, and there was nothing that brought him pleasure. Considering the clinical picture and depressive and psychotic symptoms with equal importance, the diagnosis of schizoaffective disorder-depression was eventually made according to the ICD-10. Sertraline 100 mg/d was immediately ceased. We planned to change the ineffective quetiapine to risperidone, which was more effective on positive symptoms according to our own clinical experiences when the patient developed catatonia. Mutism, posturing, nonverbal communication, hyper-myotonia of the limbs, and saliva collected in the mouth were observed. Redness and swelling of the pharynx and hyperthermia (38 °C) were present. Routine blood tests showed that the white blood cell (WBC) count was 12.85 × 10 9 /L (normal range 3.5–9.5 × 10 9 /L), and the neutrophil granulocyte (NEUT) count was 10.6 × 10 9 /L (normal range 1.8–6.3 × 10 9 /L). Chest computed tomography indicated a high-density streak like a shadow in the lower lobe of the left lung, which was clear at admission. After the case discussion, we decided to initiate risperidone at a dose of 1 mg/d and gradually titrate it to 4 mg/d to control the positive symptoms. A twice-daily intravenous injection of ceftriaxone 1 g in 250 mL 0.9% physiological saline was administered to treat the respiratory infection. Meanwhile, MECT was added three times a week to ameliorate the catatonia. MECT was administered with the SPECTRUM-5000Q device used in the bilateral mode. The treatment parameters included frequency (30 Hz), stimulus duration (2.5 s), electric charge (120 Mc), energy (21.1 J), and constant current (800 mA). Vital signs were stable; specifically, body temperature was not over 37.2 °C before starting MECT in this patient. Before and during the MECT, anesthesia was induced with etomidate 10 mg and muscle relaxation with succinylcholine 50 mg, while arterial oxygen saturation, heart rate, and electrocardiogram were continuously monitored. Each time, the patient experienced adequate generalized seizures measured with an electroencephalogram. The patient was ventilated with 100% oxygen until the resumption of spontaneous respiration.

On hospital day 23, inflammation of the pharynx disappeared, and normal WBC and NEUT counts suggested that the respiratory infection had been clinically cured. Ceftriaxone was ceased when we found negative blood bacterial culture results. We continued risperidone and MECT (a total of nine times) treatments. The patient gradually began to talk with doctors and other patients, joining some activities in the ward. The pressure of thoughts and delusion of control almost disappeared. Furthermore, it took him less and less time to perform the ritualistic behaviors. On hospital day 31, we stopped MECT and added lithium carbonate sustained-release tablets 0.6 g/d. Oral risperidone 4 mg/d was introduced, in which the blood concentration was 8.7 μg/L (normal range 2–60 μg/L). No obvious side effects were observed. Finally, we titrated the lithium carbonate sustained-release tablets to 0.9 g/d and maintained risperidone 4 mg/d. The patient remained normopyretic, and his psychotic and depressive symptoms were stable. We told the patient to review and check the blood lithium carbonate concentration 1 week later in the outpatient setting.

For the publication of this case report, written informed consent was obtained from the patient and his older sister.

Here, we describe a case of schizoaffective disorder-depression with ritualistic behaviors and catatonia. After admission, we suspected a diagnosis of OCD because of the dominance of ritualistic behaviors and depression, as the patient reported that the ritualistic behaviors must be performed or else he would feel sad. The ritualistic behaviors proved to be secondary to delusion of control. The patient’s insomnia, fatigue, and unwillingness to talk to others were not explained by the ritualistic behaviors. Ultimately, we made a diagnosis of schizoaffective disorder-depression. The symptom-based diagnostic criteria [ 1 , 2 ] as well as the multivariate symptoms made diagnosis and treatment difficult. Our case suggests that time, patience, and detailed observation are essential factors for making clinical decisions.

Selective Serotonin Reuptake Inhibitors (SSRIs) are beneficial for depression and OCD [ 6 ], among which sertraline is an effective therapeutic strategy [ 7 ]. In early studies and guidelines, psychiatrists were concerned about the risk of exacerbating psychosis when prescribing antidepressants to schizophrenic patients [ 9 , 10 , 11 ]. Rebecca Schennach et al. identified the exacerbation of positive symptoms in patients with antidepressant augmentation compared with patients without any antidepressants during the course of the study, and patients with antidepressant add-on treatment suffered from more severe psychopathological symptoms and greater psychosocial impairments at discharge [ 16 ]. However, a recent review does not support these points, as no studies found that add-on antidepressants worsened positive symptoms [ 17 ]. Although many studies have described the use of antidepressants in schizophrenia with depression, controversies remain about whether to administer antidepressants for schizophrenia spectrum disorders. We suggest that therapeutic strategies for schizoaffective disorder-depression might not include additional antidepressants, for sertraline may have exacerbated positive symptoms in our case.

Catatonia is a neuropsychiatric syndrome with psychomotor inhibition that occurs in approximately 8% of patients admitted for mental disorders, such as schizophrenia or mood disorders [ 18 ]. Schizophrenia and other psychotic spectrum disorders are more commonly presented as catatonia than mood disorders [ 19 ]. Catatonic stupor is a psychiatric emergency due to a broad range of complications [ 20 , 21 ]. Neuroleptic malignant syndrome (NMS) typically presents with fever, muscle rigidity, and altered mental status [ 22 ] and should be differentiated from catatonic stupor complicated by respiratory infection. In this case, the clear consciousness of the patient with psychomotor inhibition, redness and swelling of the pharynx, fever without muscle rigidity, increased WBC and NEUT counts, and a high-density streak of shadowing in the lower lobe of left lung in the chest X-ray at admission suggested a status of catatonic stupor complicated by respiratory infection. Therefore, our case describes schizoaffective disorder-depression with catatonic stupor complicated by respiratory infection. Controlling the infection and improving the catatonic stupor were important in treating this patient. Three treatment strategies were employed in this case. Supportive measures included high-level nursing care, intravenous fluids, and gastrointestinal support to reduce the risk of bedsores and deep vein thrombosis caused by immobility and to improve poor nutrition and dehydration. Antibiotic treatment was considered due to the redness and swelling of the pharynx, hyperthermia, increased WBC and NEUT counts, and abnormal chest X-ray. These symptoms successfully responded to the administration of 7 days of ceftriaxone. MECT was an effective strategy for improving catatonic stupor [ 23 , 24 ]. Although benzodiazepines proved to rapidly and effectively relieve catatonia, we did not prescribe these kinds of medications for the patient considering his poor nutrition, respiratory infection, and risk of respiratory inhibition. However, the role of antipsychotics in the treatment of catatonia is controversial. Several authors have suggested that antipsychotics may exacerbate the catatonic state and increase the risk of NMS [ 25 , 26 ]. Studies have found that second-generation antipsychotics (SGAs) have weak γ-aminobutyric acid (GABA)-agonist activity and 5-hydroxytryptamine 2 (5-HT 2 )-antagonism that could stimulate dopamine release in the prefrontal cortex and thus alleviate catatonic symptoms [ 20 ]. Several articles have suggested a beneficial effect of risperidone [ 27 , 28 ]. A case report identified MECT together with olanzapine, which resulted in improvement of catatonic stupor [ 29 ]. Given the results of the abovementioned studies, risperidone was cautiously administered at a low dose (2 mg/d). Once catatonic stupor improved and MECT came to a stop, risperidone would be titrated from 2 to 4 mg/d to target residual psychotic symptoms, such as the pressure of thoughts and delusion of control. Furthermore, lithium carbonate is also an effective strategy for patients with schizoaffective disorder, as it can reduce the rate of re-hospitalization [ 30 ].

Here, we describe a case of schizoaffective disorder-depression with secondary ritualistic behaviors complicated by catatonic stupor, which induces respiratory infection. Sertraline might not be recommended in patients with schizoaffective disorder to improve depression, which could have exacerbated the positive symptoms in our case. Supportive measures have an important role in the treatment of catatonic stupor. Once again, MECT has been shown to improve catatonic stupor. Although there is controversy over whether antipsychotics should be administered in the catatonic status, we considered risperidone to be beneficial in our case. Further studies should focus on the effectiveness and safety of antipsychotics associated with MECT in patients with catatonia.

Abbreviations

5-hydroxytryptamine 2

Computed tomography

5th edition of Diagnostic and Statistical Manual of Mental Disorders

Electrocardiograph

γ-aminobutyric acid

10th edition of International Classification of Diseases

• Modified electroconvulsive therapy

Neutrophil granulocyte

Neuroleptic malignant syndrome

Obsessive compulsive disorder

Second-generation antipsychotics

Selective Serotonin Reuptake Inhibitors

White blood cell

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Acknowledgements

We would like to thank the patient and his older sister for their collaboration.

This case report was partially supported by the Sanming Project of Medicine in Shenzhen (Grand No. SZSM201612006). The funding agency had no role in this case report; analysis or interpretation of data; or the preparation, review, or approval of the manuscript. We received no support from any pharmaceutical company or other industry.

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YHB and XY were involved in the management of the patient. ZQZ and HCY were the primary clinicians involved in the assessment, management, and follow-up of the patient. The article was written by YHB. YHB, XY, ZQZ, and HCY provided final approval of the version to be published and agreed to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. All authors read and approved the final manuscript.

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Bai, Y., Yang, X., Zeng, Z. et al. A case report of schizoaffective disorder with ritualistic behaviors and catatonic stupor: successful treatment by risperidone and modified electroconvulsive therapy. BMC Psychiatry 18 , 67 (2018). https://doi.org/10.1186/s12888-018-1655-5

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Module 11: Schizophrenia Spectrum and Other Psychotic Disorders

Case studies: schizophrenia spectrum disorders, learning objectives.

• Identify schizophrenia and psychotic disorders in case studies

Case Study: Bryant

Thirty-five-year-old Bryant was admitted to the hospital because of ritualistic behaviors, depression, and distrust. At the time of admission, prominent ritualistic behaviors and depression misled clinicians to diagnose Bryant with obsessive-compulsive disorder (OCD). Shortly after, psychotic symptoms such as disorganized thoughts and delusion of control were noticeable. He told the doctors he has not been receiving any treatment, was not on any substance or medication, and has been experiencing these symptoms for about two weeks. Throughout the course of his treatment, the doctors noticed that he developed a catatonic stupor and a respiratory infection, which was identified by respiratory symptoms, blood tests, and a chest X-ray. To treat the psychotic symptoms, catatonic stupor, and respiratory infection, risperidone, MECT, and ceftriaxone (antibiotic) were administered, and these therapies proved to be dramatically effective. [1]

Case Study: Shanta

Shanta, a 28-year-old female with no prior psychiatric hospitalizations, was sent to the local emergency room after her parents called 911; they were concerned that their daughter had become uncharacteristically irritable and paranoid. The family observed that she had stopped interacting with them and had been spending long periods of time alone in her bedroom. For over a month, she had not attended school at the local community college. Her parents finally made the decision to call the police when she started to threaten them with a knife, and the police took her to the local emergency room for a crisis evaluation.

Following the administration of the medication, she tried to escape from the emergency room, contending that the hospital staff was planning to kill her. She eventually slept and when she awoke, she told the crisis worker that she had been diagnosed with attention-deficit/hyperactive disorder (ADHD) a month ago. At the time of this ADHD diagnosis, she was started on 30 mg of a stimulant to be taken every morning in order to help her focus and become less stressed over the possibility of poor school performance.

After two weeks, the provider increased her dosage to 60 mg every morning and also started her on dextroamphetamine sulfate tablets (10 mg) that she took daily in the afternoon in order to improve her concentration and ability to study. Shanta claimed that she might have taken up to three dextroamphetamine sulfate tablets over the past three days because she was worried about falling asleep and being unable to adequately prepare for an examination.

Prior to the ADHD diagnosis, the patient had no known psychiatric or substance abuse history. The urine toxicology screen taken upon admission to the emergency department was positive only for amphetamines. There was no family history of psychotic or mood disorders, and she didn’t exhibit any depressive, manic, or hypomanic symptoms.

The stimulant medications were discontinued by the hospital upon admission to the emergency department and the patient was treated with an atypical antipsychotic. She tolerated the medications well, started psychotherapy sessions, and was released five days later. On the day of discharge, there were no delusions or hallucinations reported. She was referred to the local mental health center for aftercare follow-up with a psychiatrist. [2]

Another powerful case study example is that of Elyn R. Saks, the associate dean and Orrin B. Evans professor of law, psychology, and psychiatry and the behavioral sciences at the University of Southern California Gould Law School.

Saks began experiencing symptoms of mental illness at eight years old, but she had her first full-blown episode when studying as a Marshall scholar at Oxford University. Another breakdown happened while Saks was a student at Yale Law School, after which she “ended up forcibly restrained and forced to take anti-psychotic medication.” Her scholarly efforts thus include taking a careful look at the destructive impact force and coercion can have on the lives of people with psychiatric illnesses, whether during treatment or perhaps in interactions with police; the Saks Institute, for example, co-hosted a conference examining the urgent problem of how to address excessive use of force in encounters between law enforcement and individuals with mental health challenges.

Saks lives with schizophrenia and has written and spoken about her experiences. She says, “There’s a tremendous need to implode the myths of mental illness, to put a face on it, to show people that a diagnosis does not have to lead to a painful and oblique life.”

In recent years, researchers have begun talking about mental health care in the same way addiction specialists speak of recovery—the lifelong journey of self-treatment and discipline that guides substance abuse programs. The idea remains controversial: managing a severe mental illness is more complicated than simply avoiding certain behaviors. Approaches include “medication (usually), therapy (often), a measure of good luck (always)—and, most of all, the inner strength to manage one’s demons, if not banish them. That strength can come from any number of places…love, forgiveness, faith in God, a lifelong friendship.” Saks says, “We who struggle with these disorders can lead full, happy, productive lives, if we have the right resources.”

You can view the transcript for “A tale of mental illness | Elyn Saks” here (opens in new window) .

• Bai, Y., Yang, X., Zeng, Z., & Yang, H. (2018). A case report of schizoaffective disorder with ritualistic behaviors and catatonic stupor: successful treatment by risperidone and modified electroconvulsive therapy. BMC psychiatry , 18(1), 67. https://doi.org/10.1186/s12888-018-1655-5 ↵
• Henning A, Kurtom M, Espiridion E D (February 23, 2019) A Case Study of Acute Stimulant-induced Psychosis. Cureus 11(2): e4126. doi:10.7759/cureus.4126 ↵
• Modification, adaptation, and original content. Authored by : Wallis Back for Lumen Learning. Provided by : Lumen Learning. License : CC BY: Attribution
• A tale of mental illness . Authored by : Elyn Saks. Provided by : TED. Located at : https://www.youtube.com/watch?v=f6CILJA110Y . License : Other . License Terms : Standard YouTube License
• A Case Study of Acute Stimulant-induced Psychosis. Authored by : Ashley Henning, Muhannad Kurtom, Eduardo D. Espiridion. Provided by : Cureus. Located at : https://www.cureus.com/articles/17024-a-case-study-of-acute-stimulant-induced-psychosis#article-disclosures-acknowledgements . License : CC BY: Attribution
• Elyn Saks. Provided by : Wikipedia. Located at : https://en.wikipedia.org/wiki/Elyn_Saks . License : CC BY-SA: Attribution-ShareAlike
• A case report of schizoaffective disorder with ritualistic behaviors and catatonic stupor: successful treatment by risperidone and modified electroconvulsive therapy. Authored by : Yuanhan Bai, Xi Yang, Zhiqiang Zeng, and Haichen Yangcorresponding. Located at : https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5851085/ . License : CC BY: Attribution

Catatonia associated with pediatric postoperative cerebellar mutism syndrome

• Published: 17 April 2024

Cite this article

• Walter Jaimes-Albornoz   ORCID: orcid.org/0000-0003-2292-9614 1 ,
• Peter Wu   ORCID: orcid.org/0009-0005-9003-7783 2 ,
• Lydia García de Mendaza-Martínez de Icaya 1 ,
• Farah Rozali 3 ,
• María Martínez-Querol 1 ,
• Rowena Smith 2 ,
• Marco Isetta   ORCID: orcid.org/0000-0002-3878-7553 4 ,
• Ángel Ruiz de Pellón Santamaria 1 &
• Jordi Serra-Mestres   ORCID: orcid.org/0000-0003-1935-7109 5  

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To ascertain the presence of catatonia in cases of pediatric postoperative cerebellar mutism syndrome (PPCMS).

A systematic review of PPCMS case reports of patients aged 0–17 years with sufficient clinical information to extract catatonic phenomena was undertaken following PRISMA guidelines. Standardized catatonia rating scales were applied to selected cases retrospectively to ascertain whether diagnostic criteria for catatonia were met. A case known to the authors is also presented.

Two hundred twenty-one suitable full-text articles were identified. Following screening and application of inclusion criteria, 51 articles were selected plus seven more from their references, reporting on 119 subjects. All cases met Bush and Francis (BF) diagnostic criteria for catatonia, 92.5% Pediatric Catatonia Rating Scale (PCRS), 52.9% ICD-11, and 44.5% DSM-5. All patients presented with mutism. The next most frequent signs were immobility/stupor (77.3%), withdrawal (35.3%), mannerisms (23.5%), and excitement/agitation (18.5%). Most cases presented with stuporous catatonia (75.6%). Catatonia most frequently occurred following resection of medulloblastoma (64.7%). Preoperative hydrocephalus occurred in 89 patients (74.8%).

Catatonia was frequent in this PPCMS sample, with a predominant stuporous variant; it should be considered in patients with PPCMS and assessed with reliable and validated instruments for prompt diagnosis and management.

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Conceptualization: W.J.-A. and J.S.-M. Methodology: W.J.-A., J.S.-M., and M.I. Validation: W.J.-A., J.S.-M., P.W., L.G.M.-M.I., F.R., M.M.-Q., R.S., M.I., and A.R.P.S. Search strategy and database search: M.I. Formal analysis: W.J.-A., J.S.-M., P.W., L.G.M.-M.I., F.R., M.M.-Q., R.S., M.I., and A.R.P.S. Data curation: W.J.-A., J.S.-M., P.W., L.G.M.-M.I., F.R., M.M.-Q., R.S., M.I., and A.R.P.S. Writing–original draft preparation: W.J.-A., J.S.-M., P.W., L.G.M.-M.I., F.R., M.M.-Q., R.S., M.I., and A.R.P.S. Writing, review, and editing: W.J.-A., J.S.-M., P.W., L.G.M.-M.I., F.R., M.M.-Q., R.S., M.I., and A.R.P.S. All authors have read and agreed to the published version of the manuscript.

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Jaimes-Albornoz, W., Wu, P., de Mendaza-Martínez de Icaya, L.G. et al. Catatonia associated with pediatric postoperative cerebellar mutism syndrome. Childs Nerv Syst (2024). https://doi.org/10.1007/s00381-024-06392-x

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• Schizophr Bull
• v.38(2); 2012 Mar

Catatonic Schizophrenia: A Cohort Prospective Study

Karine kleinhaus.

1 Department of Psychiatry, New York University School of Medicine, 550 First Avenue, New York, NY 10016

2 Department of Environmental medicine, New York University School of Medicine, NY

Susan Harlap

Mary c. perrin.

3 Hebrew University-Hadassah Braun School of Public Health, Jerusalem, Israel

Mark Weiser

4 Department of Psychiatry, Sheba Medical Center, Tel Hashomer, Israel

5 Sackler School of Medicine, Tel Aviv University, Ramat Aviv, Israel

Jill M. Harkavy-Friedman

6 New York State Psychiatric Institute, NY

7 Department of Psychiatry, Columbia University College of Physicians and Surgeons, NY

Pesach Lichtenberg

8 Department of Psychiatry, Herzog Hospital, Jerusalem, Israel

9 Hadassah Medical School of the Hebrew University of Jerusalem, Jerusalem, Israel

Dolores Malaspina

Background:.

In the 20th century, catatonia was usually deemed a subtype of schizophrenia. Recently, the nature and classification of catatonia are being reconsidered. This study is the first to describe catatonia using prospectively collected data and to examine how catatonic schizophrenia differs from, or resembles, other types of schizophrenia.

Data were analyzed in a cohort of 90 079 offspring followed from birth till ages 29–41 years. Proportional hazards models were used, calculating time to first psychiatric hospital admission, to compare risk factors for catatonic schizophrenia vs “other schizophrenia.”

Of 568 cases of schizophrenia, 43 (7.6%) had catatonic schizophrenia. The sexes were equally at risk for catatonic schizophrenia in contrast to other schizophrenia, for which the incidence was higher in males (1.70, 1.42–2.03, P < .0001). Advancing paternal age had no influence on the risk of catatonic schizophrenia in contrast to other schizophrenia, in which the risk to offspring of fathers age 35+ was 1.27 (1.03–1.57, P = .03) compared with those of younger fathers. Those with catatonic schizophrenia were somewhat more likely to have older mothers (aged 35+) (relative risk = 2.14, 0.85–5.54) while maternal age was not related to other schizophrenia. Both were equally affected by parental history of schizophrenia. Patients with catatonia were significantly more likely to attempt suicide ( P = .006).

Conclusion:

Patients with catatonic schizophrenia show a somewhat different profile of risk factors from those with other types of schizophrenia in this cohort and are more likely to attempt suicide. This lends some support to the hypothesis that catatonic schizophrenia may have a distinct etiology.

Introduction

Since the early 20th century, catatonia has been regarded primarily as a subtype of schizophrenia. This view was first put forth by Kraeplin and Bleuler 1 and is currently upheld in both the Diagnostic and Statistical Manual of Mental Disorders, Fourth Edition (DSM-IV) and the International Statistical Classification of Diseases and Related Health Problems 10th Revision (ICD-10) . 2 , 3 In the past several decades, however, the many descriptions of catatonia as a syndrome in patients with mood disorders, medical disorders, and toxicities have put the syndrome in a new light. Some authors argue that catatonia should be considered as a separate syndrome, 1 , 4 – 6 or not exclusively as a subtype of chronic schizophrenia, 4 pointing out that catatonia presents more frequently in psychiatric inpatients diagnosed with mood disorders than in those with schizophrenia. 2 Catatonic symptoms also co-occur with other psychiatric diseases and with general medical conditions. 2 , 3

Although the nature, proper diagnostic classification, and treatment of catatonic schizophrenia are under debate, there are no population-based studies of this condition, in which the data were collected prospectively. To meet this need, we analyzed information from a large birth cohort for the purpose of quantifying demographic and clinical features of patients with catatonic schizophrenia within a population and assessing whether established risk factors for schizophrenia (sex, parental age, and family history of psychiatric disease) 7 – 11 might affect catatonic schizophrenia in the same way as other subtypes.

We used data from the Jerusalem Perinatal Study, a population-based cohort derived from all 92 408 births in 1964–1976 to mothers resident in West Jerusalem. The cohort includes linkages within nuclear families and a 29- to 41-year follow-up. Core information from the notification of birth was supplemented with other data from multiple sources. The methods and the characteristics of the population were described in detail by Harlap et al. in 2007. 12 The cohort has been linked with Israel's population registry to trace and verify identity (ID) numbers, dates of birth, and basic demographic characteristics and to ascertain vital status and dates of death.

Information on psychiatric morbidity was drawn from Israel's national Psychiatric Registry, to which the cohort has been linked. Run by the Ministry of Health since 1950, 13 the Registry contains a record of all admissions to psychiatric wards and day facilities and includes dates of admission and discharge and a single discharge diagnosis for each episode, assigned by a board-certified psychiatrist. These diagnoses are coded with the ICD; codes from earlier years have been updated to 10th Revision (ICD-10), and those for psychotic disorders have been validated recently. 14 This validation, however, must be considered in light of the fact that the diagnoses in the whole registry are compared with diagnoses in subjects selected for research. Israel's Psychiatric Registry includes a separate rubric for whether a suicide attempt was made in the 60 days prior to admission. The research was approved by institutional review boards at Hebrew University, Jerusalem, and at New York University Medical Center and exempted from the requirement for written informed consent.

Operational Definitions

Catatonic schizophrenia was defined as a record of at least 1 hospital admission prior to January 1, 2005 with a discharge diagnosis coded in ICD-10 as F20.2 (“catatonic schizophrenia”). “Other schizophrenia” was defined by at least 1 record of admission for F20 but no admissions for F20.2. For the purposes of this study, the diagnoses did not have to be at first admission or consistent over all admissions. Note that schizophrenia spectrum disorders (coded F21–29) were not included in this definition of other schizophrenia. Mood disorders were defined as at least 1 discharge diagnosis coded in ICD-10 as F30–39 (ICD-10 category of “Mood [affective] disorders”) with no admissions for any type of schizophrenia. The date of diagnosis was considered to be the date of first admission to a psychiatric facility, regardless of the diagnosis at that time. Diagnoses were coded as present or absent. Diagnoses in the parents were also taken from the psychiatric case registry and handled in the same way as in offspring.

SAS 9.1 (SAS Institute Inc, Cary, North Carolina) was used to analyze the data. After tabulating characteristics of the 2 groups of cases, proportional hazards models were constructed to analyze and compare the effects of risk factors on their incidence. Time to event was handled as completed years since birth, ie, age, till the first hospital admission or death. Survivors were censored on December 31, 2004. Ties were handled by Efron's method. A robust sandwich estimate of the covariance matrix was used to take into account correlation between siblings. 15

Potential risk factors were modeled as dichotomies or sets of mutually exclusive categories, coded 1 (if present) or 0 (absent). Included were male sex; maternal and paternal years of age (35+ vs <35); and parental history of schizophrenia. Other variables tested, but not included in the final models, were ethnic origin, socioeconomic status, and seasonal effects. Relative risks (RRs) and 95% CIs were estimated, both in univariate and multivariate models. To assess whether, in the adjusted models, differences in RRs for the 2 types of schizophrenia were statistically significant, we first considered whether their CIs were overlapping or not. We also constructed proportional hazards models within the 586 cases of schizophrenia; in these analyses, the outcome was catatonic schizophrenia vs other schizophrenia.

Of the original cohort of 92 408, there were 949 (1.0%) stillbirths and 1380 (1.5%) whose ID numbers could not be traced in the Population Registry, leaving 90 079 offspring available for this study. There were 568 offspring diagnosed with schizophrenia (ICD-10 = F20), and of these, 43 had catatonia (F20.2). The additional 292 with schizophrenia spectrum disorders (ICD-10 = F21–F29 without F20) are not considered further in this article.

Table 1 shows characteristics of the 2 groups of schizophrenia patients. Those with catatonia were evenly distributed between males and females, in contrast to those with other schizophrenia who showed a preponderance of males. Those with catatonic schizophrenia were somewhat more likely to have older mothers and to be in the lowest socioeconomic class, and more had a parent with schizophrenia. Age at first admission was similar for the 2 groups.

Characteristics of Patients with Catatonic Schizophrenia and Other Schizophrenia

Suicide attempts were recorded in 44% of the patients with catatonic schizophrenia compared with 25% of those with other schizophrenia ( P = .006). We assessed the association of suicide attempts with different diagnoses, including mood disorders, among those with catatonic or other schizophrenia. Schizophrenia patients with catatonia showed less evidence of mood disorders than those with other types of schizophrenia. Of the 43 cases with catatonia, only 3 (7%) had any admission episodes coded as a mood disorder. These 3 patients had 7 recorded suicide attempts, but only one of these occurred prior to an admission for a mood disorder (“recurrent depressive disorder, current episode severe with psychotic symptoms” [ICD-10 code F33.3]). The other attempts were in conjunction with catatonic schizophrenia or other schizophrenia, except for 1 admission for an organic disorder. The 40 patients with catatonic schizophrenia but no history of admission for a mood disorder had 48 attempted suicides recorded. The suicide attempts in these 40 were in conjunction with catatonic schizophrenia or other schizophrenia, except for 1 case whose attempt was in conjunction with “Paranoid personality disorder” (F60) and another offspring who had 1 attempt with catatonic schizophrenia but the other 2 together with “Mental and behavioral disorders due to multiple drug use and use of other psychoactive substances” (F19). For the attempts recorded together with discharge for a type of schizophrenia, 11 suicide attempts coincided with admissions for catatonic schizophrenia and 34 with other types of schizophrenia.

For the 43 cases with catatonic schizophrenia, only 25 of their 386 admissions were for diagnoses other than a type of schizophrenia. The admissions were for personality disorders, for unspecified dementia, for organic mental disorders, psychoactive substance use, mood disorder, neurotic or somatoform disorders, or mental retardation. Eleven of the 43 cases had catatonic schizophrenia diagnosed in less than 20% of their total number of admissions; for all of them, as well as the others diagnosed with catatonic schizophrenia, most admissions were for some type of schizophrenia. Analyses were rerun with these 11 reclassified as having a history of schizophrenia but not the catatonic subtype. The effects of male sex, parental history of schizophrenia, and parental age were in the same directions. Males were still less likely to have catatonic schizophrenia than other schizophrenia, but the effect was no longer statistically significant, possibly due to the smaller number of cases (data not shown). Those with catatonic schizophrenia still had a much higher percentage of suicide attempts; of those with the more conservative definition of catatonic schizophrenia, 44% had a suicide attempt, while there were 25% among those with other schizophrenia. This was a significant difference ( P = .02).

In those with catatonic schizophrenia, the proportions with 1, 2, 3, 4, and 5+ admissions to a psychiatric hospital were 5%, 14%, 9%, 2%, and 70% while of those with other subtypes the corresponding proportions were 18%, 18%, 12%, 13%, and 39%. There were 29 cases of catatonic schizophrenia (67%) who had their first admissions for catatonic schizophrenia within their first 5 admissions to hospital; however, multiple admissions for the diagnosis were rarely consecutive. Given the limited stability of subtype diagnoses, however, those with more admissions have a greater likelihood of receiving a diagnosis of catatonic schizophrenia during any one of those admissions. Nine percent of those with catatonic schizophrenia and 6% of those with other types had court-ordered admissions, while 2% vs 3%, respectively, had at least 1 admission for an organic disorder coded “Organic, including symptomatic, mental disorders” (F00–F09) in the ICD-10. These included unspecified dementia (F03), “Other mental disorders due to brain damage and dysfunction and to physical disease” (F06), including “Organic delusional (schizophrenia-like) disorder” (F06.2).

Turning to the univariate proportional hazards models (data not shown in a table), we found that sex and mother's and father's ages were each associated with a difference in risk for the 2 types of schizophrenia, as was parental history of schizophrenia. The multivariate model is shown in table 2 . While sex was not a risk factor for catatonic schizophrenia, it did markedly affect other schizophrenia, the risk of other schizophrenia being higher in males. The CIs for these RRs showed virtually no overlap; furthermore, a test of the difference, based on the model restricted to cases alone, showed them to differ significantly ( P = .03), as reported in the right hand column of table 2 .

Numbers of Cases With and Without Schizophrenia ( N ), Relative Risk (RR), and 95% CI by Type of Schizophrenia and Risk Factors for Schizophrenia

Catatonic vs other schizophrenia did not differ significantly in relation to older age of parents in the multivariate model; however, they did show opposing trends ( table 2 ). While older paternal age seemed unrelated to the risk of catatonic schizophrenia, it was a significant risk factor for other types of schizophrenia. In contrast, older maternal age tended to be associated with catatonic schizophrenia while it was unrelated to other types.

The psychiatric diagnoses of the parents, like those of the offspring, were determined from the national psychiatric case registry. Overall, there were 8 cases with catatonic schizophrenia and 66 with other schizophrenia who had any parental history for admission for schizophrenia or mood disorder. There were 3 catatonic patients with a mother with schizophrenia and 4 with a father, for a total of 9 parents diagnosed with the disorder. Those with other schizophrenia had 32 mothers and 19 fathers diagnosed with the disorder, making 51 overall. Parental history of admission for schizophrenia, as expected, was a strong and highly significant predictor of schizophrenia in the cohort. This relationship was somewhat stronger for catatonic schizophrenia, but the effects of parental history did not differ significantly between the 2 types of cases, as shown by their overlapping confidence limits and by the P value seen in the right hand column of table 2 . One of the patients with catatonic schizophrenia had a mother with an admission for affective disorder, and another had a father with it. Of those with other schizophrenia, 19 had a mother and 10 had a father with affective disorder. Numbers were too small to allow for analysis of the effects of a parent with mood disorder or for a separate model of paternal or maternal history of schizophrenia or of mood disorder.

The salient findings from this study are that catatonic schizophrenia shows no difference between the sexes and no relation to paternal age, unlike other schizophrenia, for which these are strong and robust risk factors. Another finding is that those hospitalized for catatonia have been more likely to attempt suicide than those with other schizophrenia. Our results lend support to the hypothesis that catatonic schizophrenia might be biologically distinct from other types of schizophrenia, as previously suggested. 1 , 4 – 6 More tenuous is the finding that catatonic schizophrenia may have a stronger relationship to maternal ageing and/or to mental illness in the father compared with other schizophrenia.

Prior reports on catatonic schizophrenia have been limited to cross sectional and case control studies, and case series. They have assessed the patients' characteristics and have examined prognosis but could not measure effects of risk factors. For example, 1 study, in contrast to our findings, reported that for the catatonic subtype, men outnumbered women by a ratio of 6:1 ( P = .04) 16 but could not measure the risk conferred by male or female sex.

Regarding the prevalence of catatonic schizophrenia, Rosebush and Mazurek found that in a group of patients with catatonia, 20% had a concurrent diagnosis of schizophrenia, 17 while another study reported that among a group of inpatients, 15% had catatonia and of those 54% had schizophrenia. 18 Rosebush and Mazurek also reviewed other research of the prevalence of catatonia overall in psychiatric inpatients and reported that an estimated 9%–15% of patients admitted to a typical acute care psychiatric service meet diagnostic criteria for catatonia. 17 Other studies not included in the review, which also used standardized rating scales to report on the prevalence of catatonia in psychiatric hospitals, found prevalences within this same range. 19

Our study found that people with catatonic schizophrenia attempted suicide more frequently than those with other types of schizophrenia, and the difference was statistically significant. This is notable because those with schizophrenia are already considered at high risk for suicide. Studies have shown that depressive disorders are important risk factors for suicide attempts and suicide deaths and may be stronger contributors than schizophrenia. 20 , 21 It could be considered that because catatonic symptoms appear more frequently in the context of mood disorders than psychotic ones, 2 those with catatonic schizophrenia were more likely than those with other schizophrenia to have admissions for mood disorders and that this could explain the higher rate of suicide in patients with catatonic schizophrenia in our cohort. We did not find evidence in support of this, however; although many patients with catatonic schizophrenia also had admissions for other diagnoses, only 3 had a history of mood disorder. The misdiagnosis, at times, of those with mood disorder as patients with catatonic schizophrenia would have been likely to materialize as more history of admissions for mood disorders. Thus, the link between catatonic schizophrenia and suicide in this cohort is not easily explained by the presence of mood disorder.

Our finding of an increase in suicide attempts with catatonic schizophrenia is in agreement with a cross sectional study of 31 patients hospitalized for catatonic syndrome as adolescents. The standardized mortality ratio (SMR) for suicide in these adolescents with catatonia (SMR = 54 945, 95% CI = 5189–202 066) corresponds to a 500-fold increased risk of suicide when compared with the general population of same sex and age and is clearly higher than the 9-fold increase measured in schizophrenia (SMR = 900, 95% CI = 842–962). 22 Although this suggests that adolescents diagnosed with catatonia were more likely to die from suicide than those with schizophrenia taken as a whole, a comparison of suicide in catatonic schizophrenia could not be made due to lack of reported SMR for subtypes of schizophrenia in the literature. 22 In a study of patients previously hospitalized for schizophrenia, 29% of those who eventually committed suicide had psychomotor agitation during their hospital stay compared with less than half (13%) of those who did not. 24 Psychomotor agitation is characteristic of catatonia and catatonic schizophrenia, 2 , 3 so that perhaps catatonia was also associated with an increased suicide risk in that study. Our finding adds to this literature, suggesting that catatonic schizophrenia confers a higher risk.

The lower proportion of mood disorders in those with catatonic schizophrenia compared with others with schizophrenia must also be considered in relation to the absence of sex differences we found. If mood disorders were diagnosed more frequently in those with catatonic schizophrenia, that could be thought to explain the finding of an equal rate in men and women as compared with higher risk in males for other schizophrenia. Because those with catatonia are less likely to have admissions for mood disorders, the findings on sex cannot be explained in that way.

This study faced some limitations. First, data are for hospitalizations for treatment; we could not ascertain the criteria for diagnosis but only that the final discharge diagnosis was made by a board-certified psychiatrist using ICD coding. These data must be interpreted with caution because notwithstanding the course of illness or any change of diagnosis during each hospitalization, the registry records only a single discharge diagnosis for each hospital episode. In addition, the reliability of the diagnosis of schizophrenia subtypes is generally considered low, and this could not be rectified in population-based data where the diagnoses were discharge diagnoses from a database. Another limitation is the low stability of subtype diagnoses in schizophrenia. In order to try to address this, we reclassified as other schizophrenia the offspring who were diagnosed with catatonic schizophrenia at less than 20% of their total number of admissions. The predictors seemed to affect the risk for catatonic schizophrenia in a similar fashion, implying that a misclassification of these cases due to low stability of subtype diagnosis does not necessarily explain our findings in this cohort.

An additional limitation is that the Jerusalem cohort's data on offspring are truncated in the late 1930s, so that differential lifetime incidence cannot be evaluated. Our study includes too few cases to speak to catatonic schizophrenia with certainty beyond ages 35+; the cohort will have to age before this question can be addressed.

There is no consensus in the literature on whether patients with catatonia in the context of schizophrenia constitute a distinct subtype of patients with schizophrenia, whether some patients with schizophrenia are more likely to develop catatonia, or whether catatonic symptoms can appear in patients with schizophrenia but do not delineate a distinct subgroup of patients; rather, catatonia can simply co-occur with several psychiatric as well as medical diagnoses. 1 , 5 , 6 , 23 Both the ICD-10 and the DSM-IV list catatonic schizophrenia as a subtype of schizophrenia; DSM-IV, however, also includes catatonic symptoms in other categories and states that catatonic symptoms occur more frequently in the context of mood disorders than schizophrenia. The ICD-10 contains a category for organic catatonic disorders and also includes “stupor” as a feature of several other categories; stupor in the ICD-10 has many features in common with psychomotor disturbances seen in catatonia. Some suggest that the current classification of traditional subtypes in schizophrenia should be discarded because different subtypes can present over the course of illness with schizophrenia and because the traditional classifications do not help with prediction of treatment response or prognosis. 24 These researchers propose that schizophrenia should be described with symptom dimensions, analogous to subtypes, including a dimension for the presence of motor symptoms. A description of the severity and variation in these dimensions in a patient over time would be more accurately reflecting distinct etiopathologies and genetic effects. 24 In our data, although 67% of cases had their first admissions for catatonic schizophrenia within their first 5 admissions to hospital, multiple admissions for the diagnosis were not commonly consecutive in each patient, illustrating, as noted above, that different subtypes can present over the course of illness with schizophrenia. Perhaps, at present, the best course may be to describe phenotypes until more information emerges about the correct classification and true biological underpinnings of catatonic schizophrenia or other subtypes.

In addition to the debate about the nature of catatonic schizophrenia, there are questions about its proper treatment, and more research may be needed to determine optimal treatment protocols for catatonia in the context of chronic schizophrenia. 17 , 25 – 28 Whether it is a subtype of schizophrenia, a co-occurrence of catatonia and schizophrenia, or a symptom dimension, a better understanding of the features and etiopathology of catatonic schizophrenia would facilitate further research into the best treatments.

Certain findings in basic science experiments hint that perhaps, in some cases, catatonic schizophrenia may have a distinct underlying molecular phenotype than other types of schizophrenia. Patients with catatonic schizophrenia have been reported to have lower serum brain-derived neurotrophic factor protein levels than patients with paranoid schizophrenia and residual schizophrenia ( F = 3.27, df = 2,12, P = .04). 29 In addition, levels of C-reactive protein in serum were reported to be increased in patients with schizophrenia and were even higher in those patients with schizophrenia and prominent catatonic features ( P = .04), in comparison to those with schizophrenia but without prominent catatonic symptoms. 30

Patients with catatonic schizophrenia show a different profile of risk factors and outcomes than those with other types of schizophrenia in this cohort study. This lends support to the hypothesis that their etiology differs from other cases and suggests that in future genetic and clinical studies, there may be some benefit from considering this phenotype separately. A better understanding of catatonic schizophrenia may also shed light on the etiology of schizophrenia and of catatonia presenting in other clinical contexts.

National Institute Mental Health ( K08 MH085807 to K.K., 2 RO1 MH59114 to D.M., 2-K24 MH01699 to D.M.); National Cancer Institute ( 5K07-131094-2 to M.C.P., CA080197 to S.H.); National Alliance for Research on Schizophrenia and Depression (M.C.P., S.H.)

Acknowledgments

The Authors have declared that there are no conflicts of interest in relation to the subject of this study.