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Reevaluating the Place of Cycloid Psychoses: Case Study Reavaliando o Lugar das Psicoses Ciclóides: A Propósito de um Caso Clínico Bruno Gonçalves Teixeira*, João Ferreira Perestrelo*, Ângela Venâncio*

RESUMO explorada, mais investigação será necessária Introdução: O conceito de psicose ciclóide foi dado o seu prognóstico favorável e a sua pa- descrito pela primeira vez por Karl Kleist. Mais tofisiologia e tratamento potencialmente dis- tarde, Leonhard propôs a corrente conceptua- tintos. lização descrevendo três subtipos da doença Palavras-Chave: Psicose Ciclóide; Psicose e Perris desenvolveu os primeiros critérios Aguda; Reação Psicótica Breve; Perturbação diagnósticos operacionais. O diagnóstico de Psicótica Transitória; Leonhard; Perris. psicose ciclóide possui uma longa tradição na psiquiatria europeia, mas o conceito ciclóide ABSTRACT não está explicitamente patente nos esquemas Introduction: Karl Kleist primarily de- internacionais de diagnóstico (DSM 5 e ICD- scribed the concept of cycloid psychosis. Le- 10) suscitando um debate controverso quanto onhard set the basis for the current concep- à sua utilidade e validade. tualization of the disorder describing three Objetivos: O presente artigo pretende, a partir overlapping subtypes and Perris developed de um caso clínico, abordar o conceito de psi- the first operational diagnostic criteria. The cose ciclóide enfatizando a sua importância à diagnosis of cycloid psychosis has a long luz da psiquiatria atual, discutindo o uso do tradition in European psychiatry, but the conceito e a sua validade clínica e preditiva. cycloid concept is not explicitly included in Métodos: Os autores apresentam um caso clí- standard international diagnostic schemes nico de psicose recorrente com total remissão (DSM 5 and ICD-10) leading to a controver- interepisódica e afetação funcional mínima. sial debate about its utility and validity. Resultados e Discussão: O artigo ilustra a Aims: This article intends to evaluate, from importância de estarmos atentos ao diagnósti- a case study, the cycloid psychosis concept co de psicose ciclóide dado o seu prognóstico e highlighting its importance and its clinical tratamento distinto das restantes psicoses. and predictive validity. Conclusão: Enquanto esta perturbação, de Methods: The authors present a case study of incidência desconhecida, não for devidamente recurrent psychosis with total inter-episode

* Serviço de Psiquiatria e Saúde Mental do Centro Hospitalar de Vila Nova de Gaia/Espinho [email protected]. Recebido / Received: 03.12.2014 - Aceite / Accepted: 30.10.2015.

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remission and minimal functional impair- -depressive and schizophrenia-like features3. ment. The concept of “cycloid marginal psychoses” Results and Discussion: The article il- was firstly presented characterizing atypi- lustrates the importance of the diagnosis of cal, acute, or recurrent psychoses. Then, two cycloid psychosis given its distinct prognosis syndromes related to cycloid psychosis were and treatment response compared with oth- introduced: motility and confusional psy- er psychoses. choses. Conclusion: While this disorder, of un- Kleist considered that patients with cycloid psy- known incidence, is not well reported, it is choses had a predisposition to phasic illnesses worthy of further investigation and clinical with spontaneous remission that contrast with attention given its generally favorable prog- phases of confused excitement alternating nosis and potentially distinct pathophysiolo- with stupor, or hyperactivity alternating with gy and treatment. inactivity. These illnesses, like manic-depressi- ve insanity, presented with full recovery and no Key-Words: Cycloid Psychosis; Acute Psy- apparent deficits2. chosis; Brief Psychotic Disorders; Transient Cycloid psychoses had unique characteristics Psychotic Disorders; Kleist; Leonhard; Perris. involving spells of excitement and inhibition, classically lasting two to four weeks contras- INTRODUCTION ting with the extended phases of mania and “For me this is not a matter of some special melancholia in manic-depressive insanity3. diagnostic label, since it does not matter According to Kleist, there were temperamental what names are used for these psychoses. predispositions underlying cycloid psychoses However, as I understand it, the diagnosis of mechanisms. He stated that both the cycloid a mental illness includes a prognosis. When and manic-depressive disorders seamed to rise the diagnosis of cycloid psychosis is made, from constitutional liability or dysregulation then it necessarily follows that a complete of affective, psychomotor, and cognitive func- remission will occur and even if the illness tions, with comparable phasic courses and recurs no defect will be left behind. This is, fluctuations between extremes of excitement no doubt, very important.” Karl Leonhard and inhibition, with labile, polymorphous, (1961)1. and rapidly changing manifestations, typically In the beginning of the twentieth century, without deterioration3. when attentions were focused in the distinc- Karl Leonhard, Kleist’s colleague, later intro- tion of the idiopathic psychoses proposed by duced the concept of “endogenous atypical Emil Kraepelin, Karl Kleist proposed the con- psychoses”, as illnesses characterized by psy- cept of cycloid psychosis2. chotic symptoms with an episodic pattern and Kleist considered that some cases of atypical without deterioration of function. He also in- psychoses should be classified as independent vestigated the “anxiety psychoses” described nosological entities presenting with manic- by Wernicke, specifically those presenting with

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symptoms of perplexity, paranoid ideation, ments. The increase or decrease of motor acti- ideas of reference, and perceptual disturban- vity is related with the reactive and expressive ces, with abrupt shifts in mood, going from movements that are based in psychomotor ac- anxiety to elation. He called these cycloid psy- tivity itself and do not need conscience consi- choses and proposed three major types: anxie- deration or intention. Both types of movement ty-elation, confusional excited-inhibited, and cease in akinesia. In complete akinesia the hyperkinetic-akinetic motility disorders2. patient is almost motionless1. All forms included interchange between a res- By the mid-twentieth century, Leonhard consi- tricted-inhibited phase and an expansive-exci- dered cycloid psychoses as distinct and as lying ted phase, involving a phasic and cyclic course conceptually between episodic mood disorders and full inter-episode recovery2. and chronic schizophrenia2. In anxiety-elation psychosis only the affecti- Operational diagnostic criteria for cycloid psy- vity is involved. The anxiety pole is associated choses were later developed by Carlo Perris and with ideas of reference. The elation psychosis his colleagues4. The key features stated by Per- courses not only with expansive ideas, but, ris included confusion or distressed perplexity, frequently, these patients want to make other and shifting, polymorphous symptoms. These people happy. They regularly believe that they investigators did not emphasize subtypes, as are able to bring salvation to mankind by re- Leonhard, instead they followed a “symptom ligious or political means. Anxiety psychosis is collection” approach, which is also found in more common than elation psychosis1. ICD-9 and -10 and DSM-IV and DSM 52. The The thinking process is affected in confusion diagnostic criteria for cycloid psychosis, accor- psychosis. In the excited pole of the illness it ding to Perris, can be found in Table I. shows as incoherence but very different from Several investigators following Leonhard and the incoherence of confused mania. Mutism is Perris have supported the nosological validity the distinguishing clinical picture in inhibited of cycloid psychosis. Others have considered it confusion psychosis. As these patients with as a variant of major affective disorder closely inhibition of thought are not capable to un- related to bipolar disorder, as an atypical form derstand the surrounding environment they of schizophrenia, or as excessively heteroge- become perplexed, so that “perplexed stupor” neous for reliable use. This lack of consensus (Kleist) represents the typical syndrome of has been associated with a decline in interest inhibited confusion psychosis. The inhibition in cycloid psychosis since the 1980s, although of thought also leads to false interpretation1. the continuing lack of a satisfactory system The motility psychosis has two poles (hyperki- for categorizing acute, remitting psychotic netic and akinetic) named according to the di- illnesses encourages renewed interest in the rection of the change in psychomotor activity. concept2. In both cases, the change implies an excess or The international classification systems divide deficiency of activity. There are no qualitative both clinicians and researchers. To increase changes concerning the execution of move- the diagnostic validity and reliability patients

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presenting with cycloid psychoses are categori- In this article, we report a case of cycloid psy- zed as acute polymorphic psychotic disorders, chosis and review the concept, nosological with or without symptoms of schizophrenia status, diagnostic features, associated clinical (F23 in ICD-105) and brief psychotic disorder characteristics and the etiopathological varia- (298.8 in DSM 56,7,8). bles involved in the condition.

Table I. Diagnostic criteria for cycloid psychosis by Carlo Perris2,4,9.

1. An acute psychotic condition, not related to the administration or the abuse of any drug or to brain injury, occurring for the first time in patients between 15 and 50 years old; 2. The condition has a sudden onset with a rapid change from a state of health to a full- -blown psychotic condition within a few hours or at most a few days; 3. At least four of the following: a. Confusion of some degree, mostly expressed as perplexity or puzzlement; b. Mood incongruent delusions of any kind, most often with a persecutory content; c. Hallucinatory experiences of any kind, often related to themes of death; d. An overwhelming, frightening experience of anxiety, not bound to particular situations or circumstances (pananxiety); e. Deep feelings of happiness or ecstasy, most often with a religious coloring; f. Motility disturbances of an akinetic or hyperkinetic type that are mostly expressional; g. A particular concern with death; h. Mood swings in the background that are not severe enough to justify a diagnosis of affective disorder; 4. No fixed symptomatological combination; on the contrary, the symptomatology may change frequently during the episode and have a bipolar characteristic.

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CASE REPORT On transfer to our unit he presented himself The case we report is about a 45-year-old man sleepy, dysarthric and mildly ataxic. that will be addressed as Mr. J. He was medicated with risperidone 3 mg (once Mr. J. was a construction worker who studied a day) and observed by a neurologist in order for only four years. He lived with his wife and to exclude any organic disease. Mr. J. was also teenage daughter. His family described him submitted to a detailed investigation, inclu- as a truly polite, respectful and hard-working ding computerized tomography (CT) scan, he- man, though very shy, having some difficulties matologic and biochemical routine tests and a with interpersonal relationships. neuropsychological evaluation. Nothing was Mr. J. had no psychiatric background up un- detected in CT and the analytic study showed til March 2005, when he was admitted to our no important changes. No substance misuse inward with a clinical picture of behavioural was detected. The MMSE (Mini Mental State disturbance with sexual disinhibition and Examination) and the clock drawing test di- persecutory delusions. There was no history dn’t reveal any deficit (he punctuated 25/30* of substance misuse and he wasn’t on medica- in the MMSE). The psychological evaluation tion when admitted to our department. diagnosed a mild intellectual disability. In March 2005, considering his financial pro- Two days after his hospitalization, he showed blems, he went to work in The Netherlands. an appropriate behavior, revealing no delusio- It was the first time he was living in ano- nal activity. So he was discharged to outpatient ther country and he had never travelled to follow-up being medicated with risperidone 3 anywhere so far away from home. After only mg (once a day). He abandoned this therapeu- one week abroad, he developed a clinical pic- tic regimen one week after he was discharged. ture of behavioral disturbance with aggres- He also abandoned the outpatient follow-up siveness, sexual disinhibition, coprolalia and later. total insomnia. He was sent back to Portugal In 2007, due to another financial crisis, Mr. J. a day after the beginning of the symptoms decided to emigrate again, but this time went and while waiting at the airport he started to work in Spain. A mere three days after he had looking at the men around him with distrust arrived at the country, he developed a clinical stating they were trying to seduce him and picture of psychomotor restlessness, walking intended to have sexual relationships with in the streets adrift stating he was following a him. He also believed someone had mixed light that was leading him somewhere. He ex- drugs in his cigarettes because they had a pressed himself very anxious with the fact that bad taste. he didn’t understand the Spanish language. Already in Portugal, he was evaluated in the As a result, he was sent back to Portugal soon. psychiatry emergency room and hospitalized Already at home, he maintained the behavior in our inward. disturbance. He woke up his wife in the middle

* The MMSE scale adapted for the Portuguese population considers the existence of cognitive impairment when the patient punctuates 15 or less and is illiterate, 22 or less and has between 1 and 11 years of study, 27 or less if he had studied more than 11 years.

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of the night stating he was extremely rich and inhibited confusion subtype. In both episodes that she should have sex with him immedia- (2005 and 2007), Mr. J. clearly presented with tely, if not, he would do it with their teenage two distinct clinical phases. Initially, he was daughter or even with their neighbour. excited, sexually disinhibited and talkative, His wife accompanied him to the psychia- with accelerated thought and incoherent spee- tric emergency room and while waiting to be ch, predominantly confused and hyperactive evaluated he presented himself restless and with fleeting ideas of reference. Then he fell very disinhibited (he started masturbating in into a perplexed, sleepy and dysarthric phase, front of other men and spitting over everyone being in mutism in the second episode. around him). He also assumed someone had The acute onset, the switching of polarity with mixed drugs in his food and that everyone in polymorphous clinical symptoms, the short Spain was gossiping about him. duration of the episodes – phasic course – He was hospitalized for the second time and on with complete remission and no defect left transfer to our department he was perplexed, behind support the diagnosis for this case11,12. almost in mutism. This case also meets Perris’s criteria for cy- Again, he was submitted to a detailed inves- cloid psychoses. Mr. J., a 45-year-old man, de- tigation including CT scan, hematologic and veloped an acute psychosis of unknown cause biochemical routine tests and also electroen- with sudden change from health to psychosis cephalography (EEG). The clinical investiga- within hours to a few days. He presented shif- tion didn’t identify any relevant changes and ting polymorphic symptoms, with opposed po- there was no substance misuse detected. larities within a same episode, showing mood He was medicated with risperidone 2 mg (once swings not sufficient to support a diagnosis of a day) with quick remission of the symptoms. primary major affective disorder, deep feeling On the 5th day of hospitalization, Mr. J. was dis- of happiness or ecstasy, confusion and para- charged to our outpatient unit and medicated noid features. with risperidone 2 mg (once a day). The case is difficult to categorize within the DSM5. Bipolar disorder with psychotic fea- DISCUSSION tures would be improbable to present with The diagnostic stability shown by cycloid psy- rapidly fluctuating symptoms in multiple do- choses seem to be reason enough to consider it mains and multiple modes of perceptual dis- as an independent nosological entity9,10. turbances without a mood disturbance prece- Leonhard’s classification can help identify a ding the episode. Schizoaffective disorder or subset of acute and transient psychotic disor- schizophrenia would be rejected considering ders that have a distinctly favorable progno- the complete remission of symptoms between sis8. episodes with no remaining defect. Other re- We believe this case is closest to the descrip- levant categories of non-affective psychotic tions of cycloid psychosis, with symptoms episodes in the DSM are brief psychotic di- predominantly from the Leonhard’s excited– sorder and schizophreniform disorder which

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are based on time criteria and do not provide They can be associated with severe akinetic any psychopathologic distinctions on how to complications, which are often mistaken for predict course. The ICD-10 category of “acute a malignant neuroleptic syndrome, thus the and transient psychotic disorders” specifies management of akinetic psychosis predomi- more detailed descriptive criteria although nantly demands observation and the use of this can be criticized for its heterogeneity13. anxiolytic substances16. However, it should A cohort study over three years found a high always be kept in mind that these phases also level of diagnostic instability for this cate- remit spontaneously. Electroconvulsive thera- gory14. The identification of cycloid psychoses py (ECT) may be the first choice treatment if as a subset of this category may be worthy dangerous akinetic complications are presen- of further study, with the hope of better cha- ted. Neuroleptic long-term medication should racterizing this group and thereby improving not be prescribed to these patients although prognostic validity8. it frequently is, unfortunately, due to “schi- Cycloid psychoses are only partially taken into zophreniform” symptoms. Clinical experience consideration in the formal diagnostic systems indicates that for the phasic repetitions pro- and several studies that examined its nosologi- phylactic treatment with lithium or anticon- cal status have shown they do not correspond vulsive agents may be superior to neuroleptic to any category included in modern classifi- maintenance therapy, although more studies cations. Most cases of cycloid psychosis are should be carried out to support their benefits. diagnosed by these systems as brief psychotic In his initial studies, Perris found neuroleptics disorder, schizophreniform disorder, schizoaf- far less effective than lithium for prophylactic fective disorder, mood disorder with psychotic treatment of cycloid psychoses17. On the other features, or psychotic disorder not otherwise hand there is evidence that after a sudden sus- specified15. pension of long-term neuroleptic treatment of The diagnosis of a cycloid psychosis may be cycloid psychoses the rate of relapse may in- truly important essentially with respect to the- crease7,12. rapeutical consequences. Clinical experience Treatment of cycloid psychosis with antipsy- has proved that the specific subform may play chotics, mood stabilizers, benzodiazepines, a considerable role in the pharmacothera- or electroconvulsive therapy has mainly an peutic approach. The acute therapy of anxiety empirical basis. Antipsychotic drugs are the psychoses usually requires neuroleptics, but basis of therapy, and although atypical drugs the use of anxiolytic substances substantially appear to be especially valuable in patients controls the symptoms including the paranoid with cycloid psychosis, more studies are neces- ideas. In most cases, the hyperkinetic expressi- sary to prove their efficacy15. ve and reactive motions of the motility psycho- In the present case study, there was complete sis can be improved very fast by neuroleptics. remission of the symptoms with low doses of The treatment of akinetic states is more dif- risperidone, demonstrating the effectiveness ficult, as neuroleptics may not have benefits. of atypical antipsychotics in cycloid psychosis.

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The patient abandoned the therapeutic after Orlikov (2011)20 throws out the question: being discharged. should we reevaluate the place for cycloid psy- Additionally to Leonhard’ studies18, Beckmann chosis in modern psychiatry? et al. (1990)19 have confirmed that the diag- nosis of cycloid psychoses shows high stability Conflicting Interests / Conflitos de Interesse: with few shifts between the subforms10,12. The authors have declared no competing interests The cycloid psychosis construct appears to exist. have clinical validity and utility being easily Os autores declaram não ter nenhum conflito de differentiated from neighboring syndromes interesses relativamente ao presente artigo. on psychopathological and outcome grounds. The clinical and heuristic value of the concept Funding / Fontes de Financiamento: is irrefutable, as it defines a clinical syndrome The authors have declared no external funding with a relatively characteristic symptom pat- was received for this study. tern predicting good interepisode recovery. The Não existiram fontes externas de financiamento interest of recognizing the concept lies on a para a realização deste artigo. correct differential diagnosis between patients with cycloid psychosis or with schizophrenia, References / Bibliografia something that could have dramatic thera- 1. Leonhard K. Cycloid Psychosis: Endogenous peutic and prognostic implications. All these Psychosis Which Are Neither Schizophre- features reflect well the clinical and research nic nor Manic-Depressive. Br J Psychia- importance of a concept that deserves to be in- try.1961;107(449): 633-648. cluded, on its own, in future formal diagnostic 2. Salvatore P, Bhuvaneswar C, Ebert D, Maggi- criteria of psychotic disorders15. ni C, Baldessarini RJ. Cycloid Psychosis Re- visited: Case Reports, Literature Review, and CONCLUSIONS Commentary. Harv Rev Psychiatry.2008;16 The concept of cycloid psychosis refers to par- (3):167-180. ticular types of acute, phasic, polymorphic psy- 3. Kleist K. [Cycloid, Paranoid, and Epileptoid chotic disorders. This concept may be a valua- Psychoses and the Problem of Degenerative ble alternative for some acute, good-prognosis Psychoses]. Schweiz Arch Neurol Psychi- psychotic disorders that are now very difficult atrie.1928;23:1–35 [translated by Marshall H to classify2. in: Hirsch SR, Shepherd M, eds. Themes and The cycloid psychosis concept should be ree- Variations in European Psychiatry: An Antho- valuated both for clinical diagnosis and to logy. Charlottesville: University of Virginia provide relatively homogenous disorders for Press. 1974;297–331]. genetic, physiological, neuroimaging, thera- 4. Perris C. The Concept of Cycloid Psychotic Di- peutic, and other types of research2. sorder. Psychiatr Dev.1987;6(1):37–56.

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