Schizophrenia Research 263 (2024) 45–54

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Schizophrenia Research
journal homepage: www.elsevier.com/locate/schres

Parakinesia: A Delphi consensus report

Jack R. Foucher a, b, *, Andreas J. Bartsch c, Olivier Mainberger a, b, Laurent Vercueil d,
Clément C. de Billy a, b, Alexandre Obrecht a, b, Hippolyte Arcay a, Fabrice Berna e, f,
Julie M.E. Clauss e, g, Sébastien Weibel e, f, Markus Hanke h, Julien Elowe i, Benoit Schorr e, f,
Efflam Bregeon j, Birgit Braun k, Marcelo Cetkovich l, m, Burkhard E. Jabs n,
Thomas Dorfmeister o, Gabor S. Ungvari p, q, Ludovic C. Dormegny-Jeanjean a, b,
Bruno Pfuhlmann n
a
ICube - CNRS UMR 7357, Neurophysiology, FMTS, University of Strasbourg, France
b
CEMNIS - Noninvasive Neuromodulation Center, University Hospital Strasbourg, France
c
Department of Neuroradiology, University of Heidelberg, Heidelberg, Germany
d
Clinical Neurophysiology Unit, Univ. Grenoble Alpes, INSERM U1216, CHU Grenoble Alpes, Grenoble Institut Neurosciences, Grenoble, France
e
Pôle de Psychiatrie, Santé Mentale et Addictologie, University Hospital Strasbourg, France
f
Physiopathologie et Psychopathologie Cognitive de la Schizophrénie – INSERM 1114, FMTS, University of Strasbourg, France
g
SAGE – CNRS UMR 7363, FMTS, University of Strasbourg, France
h
University Hospital of Child and Adolescent Psychiatry and Psychotherapy, University of Bern, Switzerland
i
Department of Psychiatry, Prangins Psychiatric Hospital (CHUV), Prangins, Switzerland
j
Pôle de Psychiatrie, University Hospital Angers, France
k
Abteilung für Psychosomatische Medizin und Psychotherapie, Universitätsklinikum Regensburg, Germany
l
Institute of Translational and Cognitive Neuroscience (INCyT), INECO Foundation, Favaloro University, Buenos Aires, Argentina
m
National Scientific and Technical Research Council (CONICET), Buenos Aires, Argentina
n
Klinik für Psychiatrie & Psychotherapie, Städtisches Klinikum Dresden, Dresden, Germany
o
Abteilung für Psychiatrie und psychotherapeutische Medizin, Landesklinikum Neunkirchen, Austria
p
Section of Psychiatry, University Notre Dame, Fremantle, Australia
q
Division of Psychiatry, School of Medicine, University of Western Australia, Crawley, WA, Australia

A R T I C L E I N F O A B S T R A C T

Keywords: Abnormal movements are intrinsic to some forms of endogenous psychoses. Spontaneous dyskinesias are
Parakinesia observed in drug-naïve first-episode patients and at-risk subjects. However, recent descriptions of spontaneous
Grimacing dyskinesias may actually represent the rediscovery of a more complex phenomenon, ‘parakinesia’ which was
Mannerism
described and documented in extensive cinematographic recordings and long-term observations by German and
Periodic catatonia
Tardive dyskinesia
French neuropsychiatrists decades before the introduction of antipsychotics. With the emergence of drug induced
Endogenous psychoses movement disorders, the description of parakinesia has been refined to emphasize the features enabling dif­
Psychomotor phenomena ferential diagnosis with tardive dyskinesia. Unfortunately, parakinesia was largely neglected by mainstream
Schizophrenia psychiatry to the point of being almost absent from the English-language literature. With the renewed interest in
Wernicke–Kleist–Leonhard school motor phenomena intrinsic to SSD, it was timely not only to raise awareness of parakinesia, but also to propose a
Neuropsychiatry scientifically usable definition for this phenomenon. Therefore, we conducted a Delphi consensus exercise with
Schizophrenia spectrum disorders clinicians familiar with the concept of parakinesia.
The original concept was separated into hyperkinetic parakinesia (HPk) as dyskinetic-like expressive move­
ments and parakinetic psychomotricity (PPM), i.e., patient's departing from the patient's normal motion style.
HPk prevails on the upper part of the face and body, resembling expressive and reactive gestures that not only
occur inappropriately but also appear distorted. Abnormal movements vary in intensity depending on the level of
psychomotor arousal and are thus abated by antipsychotics. HPk frequently co-occurs with PPM, in which
gestures and mimics lose their naturalness and become awkward, disharmonious, stiff, mannered, and bizarre.
Patients are never spontaneously aware of HPk or PPM, and the movements are never experienced as self-

* Corresponding author at: CEMNIS (UF 4768) - Centre de NeuroModulation Non Invasive de Strasbourg, 1 place de l'Hôpital, BP 426, 67 091 Strasbourg Cedex,
France.
E-mail address: [email protected] (J.R. Foucher).

https://doi.org/10.1016/j.schres.2022.09.024
Received 21 July 2022; Received in revised form 22 September 2022; Accepted 22 September 2022
Available online 7 November 2022
0920-9964/© 2022 Published by Elsevier B.V.
J.R. Foucher et al. Schizophrenia Research 263 (2024) 45–54

dystonic or self-alien. HPk and PPM are highly specific to endogenous psychoses, in which they are acquired and
progressive, giving them prognostic value. Their differential diagnoses and correspondences with current in­
ternational concepts are discussed.

1. Introduction Applying ICD-10 diagnostic criteria, 82 % of PPC were diagnosed with

SSDs, but only 20 % had one catatonic episode (Krause, 2012). PPC
Abnormal movements have long been described as intrinsic to some attracted attention of DSM-5 Task-Force and the World Federation of
schizophrenia spectrum disorders (SSDs). They were even at the very Societies of Biological Psychiatry (Kupfer et al., 2002; Stöber et al.,
core of the concept of ‘catatonia’ for Karl Kahlbaum who described them 2009) because of its high heritability and the replication of a suscepti­
as muscle twitches; pseudo-epileptic, choreatic, cramp-like movements; bility locus in 15q (OMIM 605419) (Stöber and Reis, 2009). The sig­
and spasms affecting any part of the body (§1 - in Supplementary ma­ nificance of PPC was further supported by the discovery of its brain
terial no. 1) (Kahlbaum, 1874). At the beginning of the 20th century, imaging biomarker (de Billy, 2020; Foucher et al., 2020a). Unfortu­
Kraepelin and Bleuler abandoned the conception of catatonia as a nately, PPC is only known and diagnosed by clinicians trained in the
nosological entity and blended it with other SSDs (Fink et al., 2010) thus Wernicke-Kleist-Leonhard (WKL) school of psychiatry, hindering the
diverting the interest that psychiatrists had in motor anomalies (Foucher replication of their results by other investigators. The increased
et al., 2022b). Formerly considered to be indicative of the involvement awareness of HPk and its importance in differential diagnosis from TD
of specific brain systems (Kahlbaum, 1874; Wernicke, 1900, 2015), might call the attention to the diagnosis of PPC beyond the small WKL-
movement disorders were relegated to the status of secondary mani­ community, since in acutely hospitalized SSDs >85 % of HPks are
festations and nearly disappeared from psychiatric training and recog­ accounted for by PPC patients (Foucher et al. in preparation).
nition (Kendler, 2016). This neglect became nearly complete after the The objective of this article is, therefore, to provide a sufficiently
introduction of neuroleptic drugs. First-generation antipsychotics unambiguous and distinctive definition of parakinesia that can be
attenuated intrinsic abnormal movements, while their extrapyramidal shared and investigated by the broader psychiatric research community.
side-effects became a one-fits-all explanation for motor disorders in To this end, German and French original descriptions were reviewed to
SSDs. Thus, these movement anomalies were either masked by, or lay the foundation for a consensus definition using the Delphi procedure.
wrongly attributed to antipsychotics.
However, partisans of the neuropsychiatric approach continued to 2. Methods
enrich the description of spontaneously occurring abnormal movements
in the context of SSD. In Germany, Carl Wernicke viewed them as a The Delphi procedure is a structured, systematic, interactive method
component of his ‘parakinetic’ phenomenon (Wernicke, 1900, 2015), in which a panel of experts builds a consensus (Dalkey, 1969; Eubank
but it was his follower Karl Kleist who separated parakinesia (‘Para­ et al., 2016). We applied the Delphi method as follows:
kinesen’) from other psychomotor and neurological motor disturbances
(§2,3) (Kleist, 1909, 1934). In the 1920s, patients exhibiting parakinesia 1) The first author systematically reviewed the German, French, and
were filmed and subsequently followed over several decades, well English literature on the concept of parakinesia. The manuscript was
before antipsychotics were introduced. In the French literature, the reviewed and enriched by all co-authors, including a movement
same phenomenon was described by Paul Guiraud as ‘meddling motor disorder specialist (LV). The experts first agreed on four video cases
complexes’ (‘complexes moteurs parasites’) (§5) (Guiraud, 1956), which but discarded one because some experts found it too short to confi­
were later conflated with Kleist's concept by Henri Ey, endorsing the dently diagnose the disorder. The reader can find addendum (A),
name parakinesia (§6) (Ey et al., 1989). However, it is mainly to Karl video illustrations ( ), historical quotes (§), and definitions (¤) in
Leonhard (§4), and later to the Würzburg school, that we owe not only Supplementary material no. 1.
the continuation of the teaching of parakinesia semiology but also the 2) The first series of statements (S01–52) and proposals to name the
establishment of clinical characteristics distinguishing parakinesia from concepts (N01–11) were developed from the revised version of the
tardive dyskinesia (TD) (Foucher et al., 2022a). draft manuscript by the first author and submitted to 22 potential
Although ‘spontaneous dyskinesia’ is arguably a rediscovery of hy­ experts, of whom 16 (73 %) responded (see Supplementary material
perkinetic parakinesia (HPk), it is only described in neuroleptic-naïve no. 1 for experts and further details).
first-episode psychotic patients (Ayehu et al., 2014; Fenton, 2000; 3) The manuscript was rewritten according to the experts' responses
Owens et al., 1982). Yet, with notable exceptions (e.g. Waddington and and sent together with additional statements (A01–27) for a second
Crow, 1988), the question of a possible conflation of endogenous round of voting, to which 15 of 16 (94 %) experts responded.
movement disorder (HPk) with TD remains iconoclastic as soon as the 4) The manuscript was appropriately amended and sent to the experts,
patient has been given antipsychotics. Investigations of parakinesia are who are co-authors of this paper. All authors agreed on the current,
further limited by the absence of its term and concept in the contem­ final version; no third round of voting was needed.
porary English-language literature. Since Fish's articles and textbooks in
the 1950s and 1960s (§7) (Fish, 1958, 1962, 1967), the only mentions of
3. Results
which we are aware are a review of abnormal movement in psychosis in
the pre-neuroleptic era (Waddington and Crow, 1988) and Bräunig's
3.1. Context
scale of catatonic symptoms (Bräunig et al., 2000).
We believe that the concept of parakinesia deserves to be known and
3.1.1. The experts
studied, at least for two reasons. Firstly, as an individual sign, it proposes
All experts were trained in the WKL neuropsychiatric tradition and
empirically driven clues to distinguish HPk from TD that has never been
still use and teach the concept of parakinesia (Foucher et al., 2020a,
formally tested. Secondly, as part of a symptom-complex, parakinesia is
2020b). Four rated their level of expertise as “fair,” eight as “good,” and
highly suggestive of a good candidate phenotype, to which we will refer
four as “high.”
to as progressive periodic catatonia (PPC – Addendum no. 1 in the
Supplement). The addition of the course (progressive) to Leonhard's
3.1.2. Neuropsychiatric background
label (periodic catatonia) is intended to prevent it from being misun­
Parakinesia was conceptualized within the neuropsychiatric frame­
derstood as a simple recurrence of ICD/DSM catatonic episodes.
work which, unlike current ‘atheoretical’ psychiatry, relies on a firm

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theoretical background. This may not change the phenomenon per se, 〉 HPk can be outlined as “parasitic movements that distort, overload,
but gives it a different meaning. ‘Psychomotricity’ (PM) for instance, or replace normal movements,” mixing “grimaces, [and] paradoxical
would not be described differently as a phenomenon, i.e., the whole non- mimics” (§6,8) (Ey et al., 1989).
verbal motor output, which can be interpreted as reflecting emotional 〉 PPM refers to the distortion of psychomotricity (PM): expressive and
and motivational states (Foucher et al., 2022b). But stating that ‘para­ reactive gestures appear bizarre and/or lose their expressiveness to
kinesia is a psychomotor disorder’ has very different meaning from a the point of being uninterpretable.
system-based neuropsychiatry perspective. The most important differ­
ence is that PM is supposed to be accounted for by specific (psycho) 3.1.4. Examination conditions
motor systems, which are separate from intentional motor systems. HPk, and even more so PPM are subtle phenomena that are not al­
Accordingly, intentional motor systems can be affected without ways easy to identify during an interview. Experts agree that the sys­
impairment of expressive movements, but most importantly with respect tematic use of videotaping facilitates evaluation by allowing PM to be re-
to parakinesia, expressive PM can be affected without gross motor examined a posteriori and is useful for sharing experiences (A24).
impairment. Central facial palsy is illustrative of this double dissociation
and its interpretation by two independent motor systems, volitional and 3.2. Hyperkinetic parakinesia or HPk as “psychomotor dyskinesia”
expressive, and consistently supported by evidence (Fig. 1) (Müri,
2016). Consequently, the outputs of psychomotor systems, i.e. PM, can 3.2.1. Phenomenology of HPk
be dissociated from patients' (internal) emotional or motivational states HPk comprise abnormal involuntary movements so protean that any
and can be specifically disordered as in parakinesia. attempt to describe them is doomed to be incomplete. These movements
The prefix ‘para-’ meant that it was supposed to result from aberrant are classically described as having a ‘pseudo-reactive’ (S02) but mostly
activity of psychomotor systems (Wernicke, 1900, 2015), mainly of ‘pseudo-expressive’ appearance (S06) to indicate that they are, or look
emotional motor systems (Kleist, 1909, 1934; Müri, 2016). Emotional like, distorted PM (S04). Thus, at the beginning of its evolution, HPk is
motor systems control expressive movements, that are always more or characterized by complex expressive-like movements, i.e., movements
less complex and global patterns of co-contraction (Du and Martinez, less elementary than chorea or dystonia (S01), to the point that their
2015). They are opposite to the intentional motor systems, which can appearance suggests the interpretation of a reflection of psychic activity
have the precise control of elementary movement. (S03). Though HPk are typically distorted and bizarre (S04), they are
sometimes recognizable only by their dissociation from the patient's
3.1.3. Hyperkinetic parakinesia (HPk) – parakinetic psychomotricity reported subjective experience, i.e., paramimia, paradoxical expres­
(PPM) distinction sions, discordant smiles, or inappropriate affect (Dromard, 1907; Ey
The classical WKL psychopathology does not distinguish between et al., 1989; Guiraud, 1956).
hyperkinetic parakinesia (HPk) and parakinetic psychomotricity (PPM) HPk essentially occurs on top of expressive (S02) or reactive move­
(N02, N10). However, experts agreed on describing HPk and PPM ments (S06). HPk is less perceptible or even absent during controlled
separately (A01): voluntary actions and the automatisms that accompany them (Leonhard,
1999). HPk movements are exceptionally large in amplitude and never
like ballistic movements (S27). Although HPk often has an asymmetric
aspect at a given time (S05), longitudinal observation shows that in

Fig. 1. Double dissociation between expressive and volitional facial expression. Central facial palsy only involves the lower part of the face because the upper part
receives innervation from both sides. Hence only the inferior part shows the dissociation. When the left opercular part of M1 is injured, voluntary movements of
lower right side of the face are impaired. But when telling the patient a good joke she presents a normal, symmetric smile (upper row). The opposite deficit is
observed when the rostral and the caudal cingulate motor areas (M3, M4) are injured (lower row). The patient can still contract perioral muscles when asked to show
his teeth (intentional) but has weakness of the lower right side of the face on spontaneous expressive smiling. The supplementary motor area corresponds to M2.
Inspired from Ross and Mathiesen (1998).

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SSDs, HPk mostly affects both sides (A03) (Kleist, 1934). HPk has no 3.2.1.3. HPk modulation by the level of PM excitation. The frequency and
functional impact on fine motor skills; any impairment in the manipu­ intensity of HPk increase with the level of PM arousal (S14) (Fish, 1958;
lation of objects would be an argument against the diagnosis (S39). Leonhard, 1999). In addition to the quantitative effect, PM excitation
also qualitatively modifies HPk. Pseudo-expressive and pseudo-reactive
3.2.1.1. Body location of HPk movements become larger but also more distorted sometimes to the
3.2.1.1.1. Pseudo-expressive appearance of facial HPk. HPk predom­ point of resembling grimaces; HPk becomes more impulsive and les
inantly appears on the upper part of the face (S07); the mouth and jaw complex as the level of PM excitation increases and may lose their
are rarely involved alone. HPk of forehead, eyebrows, eyelids, and expressiveness, taking on a more choreiform appearance.
orbital muscles produces a particularly pseudo-expressive appearance This PM arousal may be endogenous (e.g., hyperkinetic episodes) or
( 1,2) (Bräunig et al., 2000; Leonhard, 1999). The movements may externally driven ( 2). Thus, HPk may be absent when the patient is left
involve a single muscle or several muscles; lifting or frowning of the alone without stimulation, appearing only with the arousal and normal
eyebrow, widening or blinking of the eyes, sniffing and wrinkling of the emotional expression elicited by an interview (S13–14). Interpersonal
nose, etc., may occur together or in isolation (Fink and Taylor, 2003; interaction and perhaps the evocation of emotionally charged material is
McKenna et al., 1991). Leonhard and Guiraud pointed out that certain more sensitizing than mental calculation commonly used by movement
eyebrow movements dissociated between the right and left eyebrows are disorder specialists. Hence, HPk fluctuate in intensity—stable abnormal
difficult to imitate voluntarily, describing this asymmetry as a charac­ movements (S33) and poor reactivity to PM excitation (S34) argue
teristic of facial HPk (S05) (Guiraud, 1956; Leonhard, 1999). against the diagnosis.
Conversely, HPk never involves the tongue, pharyngeal, or laryngeal
muscles like TD (S29). 3.2.2. HPk in the context of SSDs
3.2.1.1.2. Pseudo-reactive appearance in the neck, upper trunk and All experts considered that HPk frequently co-occurs with PPM but
limbs. HPk may also involve the upper part of the body (S08). Move­ did not consider PPM to be necessary for the diagnosis of HPk, i.e., PPM
ments resemble inappropriate and distorted, expressive, startled, or is a positive criterion (S18) but not a negative one (S40). HPk also
orienting movements involving the axial and proximal musculature, frequently co-occurs with other catatonic phenomena (S19).
such as head nodding, shoulder shrugs, slight trunk twists, and brief As the illness progresses, HPk becomes increasingly distorted and
body swings. HPk can also appear as slight jerky movements of the loses its expressiveness (A05). With time, HPk often loses polymorphism
fingers, hands, arms, or shoulders (S08). Because these movements are (A06) and becomes more repetitive, uniform, and stereotyped (A07), e.
often activated with increasing PM arousal during conversation, they g. HPk may take the form of blepharospasm, simple increase in blinking
may be misinterpreted as a manifestation of discomfort or nervousness frequency (Bräunig et al., 2000), choreiform movements or appear as
or as motor tics (S03) (Leonhard, 1999). HPk movements may appear distorted and bizarre stereotypies (A05) (McKenna et al., 1991).
choreiform but lack torsion or a screw-shaped characteristic (S28). HPk should be the first hypothesis in cases of abnormal movement in
3.2.1.1.3. Occasional pseudo-dystonic appearance. HPk can also take antipsychotic-naïve patients (S15). HPk is attenuated and might even
on a pseudo-dystonic appearance (A02). A classic example is the “snout disappear under treatment with all antipsychotics except clozapine
spasm” (“Schnauzkrampf”) described by Kahlbaum (§1,5). Although the (S16) (Fish, 1964), but is unresponsive to anticholinergic drugs (S37).
movement is usually described as a simple protrusion of the lips, making
them look like an animal's snout (Rogers, 1991; Tang and Duffin, 2014), 3.2.3. Differential diagnoses
it is rarely limited to the lower part of the face and is frequently HPk can mimic many forms of abnormal movements: some are
accompanied by a wrinkling of the nose (Fish, 1962), a squinting of the abrupt and rapid like chorea, others are more sustained like dystonia,
eyes, and/or a frowning of the forehead ( 3b). HPk may also resemble and some are diagnosed as tics ( 2, §7). Yet, in our practice, the most
blepharospasm, with blinking or eye closure, or even Meige syndrome¤ important differential diagnoses are tardive dyskinesia or TD (S51) and
when associated with lip pinching (Bräunig et al., 2000). choreiform movements (Table 1). When HPk are particularly apparent,
psychiatrists and movement disorder specialists unfamiliar with the
3.2.1.2. HPk is necessarily experienced as self-syntonic. HPk is often concept of HPk may qualify the movements as ‘tics’, notwithstanding the
inconspicuous and overlooked by families (A17). Even when obvious lack of awareness and the complete absence of control (A26). For the
( 2), it is not accompanied by the subjective experience that something sake of brevity, we only elaborate on the differential diagnosis with TD
is wrong and is not experienced as alien to oneself, a feature that Kleist and refer the reader to Table 1 and Supplementary material for the
referred to as ‘self-syntonic’ (S12) (Kleist, 1934). Although the invol­ others (Addendum no. 2).
untary and bizarre nature of movement may arouse compassion, pa­
tients do not suffer from HPk. Even in severe forms, HPk does not cause 3.2.3.1. Tardive dyskinesia (TD). Because TD and HPk share common
discomfort and is not accompanied by emotional reactions or concern. features, and can coexist in the same patient, the differential diagnosis
Patients never complain about HPk, and it does not induce delusions of may be challenging (Koning et al., 2010). TD follows the prescription of
influence. a precipitating drug, usually an antipsychotic, but other drugs acting on
Patients are never spontaneously aware of HPk; it does not seem to the dopaminergic system, such as antiemetics, calcium blockers, and
be accompanied by motor awareness (S09). Patients seem unable to SSRIs, may also trigger TD (Frei, 2019). If the patient has been previ­
report HPk movements, even when the examiner has drawn their ously exposed to TD-inducing drugs (S15–16), the following criteria are
attention to them (S10). However, this is not related to a global lack of proposed for the differential diagnosis (summarized in Fig. 2):
insight (S11), and some patients accept the idea of exhibiting HPk based
on what they are told by relatives, friends, or physicians, i.e., there is no 〉 TD does not have the same complex (S01) pseudo-expressive (S02) or
anosognosia. Similarly, no anosodiaphoria¤ has been reported in pseudo-reactive (S06) aspect as HPk has and usually does not
HPk—some patients say that they understand (intellectually) that HPk resemble a meaningful gesture (S03).
can socially handicap them. Patients are unable to control HPk other 〉 TDs is more stereotyped than HPk. Unfortunately, this criterion is
than by refraining from moving or mechanically hindering themselves, probably less discriminative in the final stage of SSDs.
e.g., by crossing their arms or fingers (A04) ( 3c). Accordingly, partial 〉 TD is more regular in amplitude whereas HPk fluctuates.
awareness (S31), or self-dystonic experience (S32), and the ability to 〉 TD are mostly of bucco-linguo-masticatory kind (80 %). Hence, TD
control movements (A09) strongly argue against the diagnosis of HPk. predominates in the lower part of the face vs the upper part in HPk
(S07). TD typically involves the musculature of the lips and tongue,

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Table 1
Differences between HPk and other hyperkinetic movement disorders.
Hyperkinetic parakinesia (HPk) Tardive dyskinesia (TD) Chorea
a
Description Complex movements. Stereotyped, irregular, continuous. “Dance-like” movements, randomly flowing from one
Distorted pseudo-expressive or Oromandibular type (80 %) body part to another.
pseudo-reactive aspect. Involuntary, irregular, purposeless, non-rhythmical,
Can be interpreted as being related to 〉 Pursing, sucking, pouting, or puckering of the lips rapid, unsustained, but quite continuous.
inner psychic state. 〉 Writhing, snapping of the tongue Often co-occurs with athetosis, adding torsion-like,
Frequent association with PPM ± 〉 Chewing or lateral jaw movements screw-shape, twisting, or writhing movements.
other catatonic phenomena. 〉 Often alter speech and swallowing Motor impersistence.
No functional impact. 〉 ±Painful sensation Hypotonia (flaccidity, pendulous reflexes).
Not stereotyped (initially). ±Facial “tics,” eye blinking
Limb dyskinesia: Choreiform movements of the
extremities.
Remark: Rabbit syndrome¤ and tardive dystonia¤ have
different pathophysiology and treatments.
Course and Acquired. Acquired. Mostly acquired.
fluctuation Unrelated to previous antipsychotic Secondary to sufficient D2-blocker intake (>3–6 M). Quite stable in time.
intake. Quite stable in time and poorly sensitive to PM excitation.
Fluctuates in intensity; increases with
↗ PM arousal.
Reduced by D2-blocker.
Involved body Upper part of the face, may involve the Mouth, jaw, tongue, pharynx, larynx. Poorly localized.
parts mouth and jaw. Unlikely to affect the upper part of the face (i.e., above Likely to involve the upper part of the face (above
Upper part of the body, mostly on eyelids). eyelids).
proximal muscles. Extremities of the limbs. Proximal as well as distal parts of the limbs.
Awareness Completely absent, no motor Present in half of the cases, occasional discomfort. Almost absent at the beginning.
consciousness, self-syntonic. Frequently incomplete thereafter.
Unrelated to insight in the SSD
Control None. Partial. Partial.
a
Less elementary than the classical dyskinesia, chorea, or dystonia; resembling deformed PM gestures.

Fig. 2. Elements for the differential diagnosis between facial hyperkinetic parakinesia (HPk – left) and tardive dyskinesia (TD – right). The frequent description of TD
as ‘grimacing’ questions its differential diagnosis with catatonic grimaces. HPk offers discriminative features on appearance, face location (grey: only HPk; cross­
hatched: both HPk and TD; dotted: only TD), variability (pattern and amplitude), awareness, controllability, functional impact, and relationship to drug intake.

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J.R. Foucher et al. Schizophrenia Research 263 (2024) 45–54

often that of the jaw, and more rarely the pharyngolaryngeal HPk, PPM mostly concerns interpersonal motor automatisms, i.e.,
musculature (Hauser et al., 2020), which are almost never involved expressive and reactive movements, rather than acquired skills and
in HPk (S29). voluntary actions.
〉 TD has torsion and a screw-shaped characteristic, which is not
observed in HPk (S28). 3.3.1. Phenomenology
〉 TD can be painful, and often alter speech and swallowing whereas PPM describes the deformations and disharmonious aspects of PM:
HPk has no functional impact (S39). the gait, gestures, mimics, and manner of greeting or sitting lose their
〉 In limbs, TD mostly affects extremities, whereas HPk typically in­ natural grace ( 3c) (S41). Leonhard used the term “parakinetic motor
volves the proximal musculature (S08) and can affect the lower limbs flow” to refer to a loss of fluidity during movements. PM loses its
which is hardly ever the case in HPk. expressive character and gives an impression of weirdness: the mimics
〉 TD is not related to PPM (S18) or other catatonic manifestations are distorted, sometimes to the point of becoming indecipherable.
(S19).
〉 Continuation of antipsychotic treatment aggravates TD. Although 3.3.1.1. Body location. PPM predominantly involves the facial, axial,
increasing the dose may attenuate TD in the short term, it will and proximal muscles (S43). Deformations have an inhomogeneous
aggravate it in the medium term (Caroff, 2020). This is not the case distribution at a given moment but affect different body parts during the
for HPk, which only worsens with evolution of the pathology and in same examination (S44). PPM can also affect the intensity, tone, rhythm,
relation to PM excitation (i.e., HPk disappears under antipsychotic or prosody of verbal expression (A11). The voice may be abnormally
drug treatment without delayed worsening). loud, often high and shrill, or weak. The speech rate is irregular, with
〉 Finally, at least half of the affected patients are mindful of having TD, pauses and sudden resumptions (Fish, 1962; Leonhard, 1999). The
suffer from them, and even complain about them: they are experi­ speech may lose its prosody, expressiveness, and intonations, resulting
enced as ‘self-dystonic’. In these cases, TD may be voluntarily sup­ in monotonous expression resembling that of an automaton (McKenna
pressible and can sometimes be controlled by ‘sensory tricks’ (“geste et al., 1991). The admixture of stiff and exaggerated psychomotor
antagoniste”) (Gilbert and Waters, 2010). movements at a given time is very suggestive of the specific kind of PPM
observed in PPC (S45).
3.2.4. Proposed criteria for HPk
The criteria selected for HPk are presented in Table 2. 3.3.1.2. PPM is self-syntonic. PPM has no functional impact on fine
motor skills like writing or manipulating objects (innate automatic-
3.3. Parakinetic psychomotricity (PPM) voluntary dissociation) (A12,19). PPM is never noted by the patient
(A13) and is only exceptionally noticed by the family (A18); its delayed
PPM is a more discreet but perhaps more enduring manifestation, onset (A20), concurrent with early signs of the illness, is difficult to
although its expression depends on the level of PM arousal (S42). Like ascertain in the absence of premorbid recordings. Without video docu­
mentation, examiners cannot compare the present PM style with the
Table 2 patient's initial psychomotor style and is limited to judging PPM against
Proposed criteria for parakinetic psychomotor dyskinesia. PM: psychomotor. their own standards. It often takes the examiners time to convince
Very suggestive Incompatible
themselves of the disharmonious character of the patient's PM.

Aspect Complex movements (less Large amplitude of abnormal

elementary than classical movements (e.g., ballistic).
3.3.1.3. PPM is modulated by the level of psychomotor excitation. PPM
dyskinesias, chorea, or dystonia). Functional impact on fine motor can take on several aspects depending on the degree of psychomotor
Changing, distorted pseudo- skills (e.g., impairing excitement (S42):
expressive or pseudo-reactive manipulation of objects).
aspect. Strictly unilateral (i.e.,
〉 Hypokinetic PPM: stilted, rigid, and even stiff posture, gait, and
May look like an expression of the longitudinally).
inner psychic state. gestures. The inhomogeneous distribution of stiffness is character­
Possible pseudo-dystonic istic, appearing only on certain joints ( 3c).
appearance. 〉 Normokinetic PPM: clumsy, awkward, mannered appearance.
Isolated movements of the
〉 Hyperkinetic PPM: eccentric, affected, artificial, and/or jerky,
forehead muscles (e.g., right/left
dissociation).
impulsive, hurried, choppy gestures. Thus, hyperkinetic PPM and
Location Upper part of the face. Tongue, pharynx, larynx, and HPk appear to form a continuum.
Upper part of the body (neck, mandible. 〉 Mixed PPM: stiffness and excessive movements occurring at the same
trunk, shoulders). time on different parts of the body; this aspect is quasi-specific to
Awareness Lack of motor awareness, no Patient spontaneously aware of
PPC.
control the abnormal movement and/or
able to control it.
〉 Even when attracting patient's Self-dystonic experience, 3.3.2. Interaction with treatment
attention to it discomfort. Antipsychotics may modulate PPM by promoting its expression in
〉 Unrelated to the general level of
the hypokinetic form. In our experience, the most classic manifestation
insight
Self-syntonic experience.
is the ‘robotic gait’ (A14). It is classically interpreted as antipsychotic-
Fluctuation More frequent/pronounced with Unchanged by PM excitation/ induced parkinsonism but differs in the following respects (A15):
↗ PM excitement. inhibition.
Associated Parakinetic psychomotricity 〉 Inhomogeneous distribution: the posture is upright; the trunk and
(PPM).
the roots of the limbs move as a single block. When walking, the
Other catatonic phenomena.
arms' sway seems to have a normal amplitude but is only ensured by
the shoulder joint, while the elbows lack flexibility (or vice versa).
This dissociation is sometimes even clearer when the patient's
Orienting Incompatible
shoulders are passively swung, showing no lack of pendulousness.
Treatment Patient naïve to any treatment. Improved by anticholinergic drugs. 〉 The absence of resistance to passive mobilization, often even to
Patient under clozapine.
Other Other catatonic manifestations.
Froment's¤ maneuver.

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J.R. Foucher et al. Schizophrenia Research 263 (2024) 45–54

〉 The absence of other parkinsonian signs: no objective bradykinesia grimacing and spontaneous dyskinesia and the problem of the differ­
(low amplitude, slowness, fatigability), no resting tremor. ential diagnosis with drug-induced movement disorder.
〉 The absence of awareness. The patient never complains of subjective
slowing, denies any stiffness, and does not recognize any gait 4.1. Overlap with current catatonic manifestations
abnormality.
HPk and PPM could correspond to many items in the four major
This robotization is interpreted as an interaction between PPM and catatonic scales for adults (Bräunig et al., 2000; Bush et al., 1996;
antipsychotic treatment. Antipsychotics transform the expression of McKenna et al., 1991; Northoff et al., 1999) (Fig. 3 and Supplement).
PPM by reducing the level of psychomotor excitation (Tenback and van Bräunig's scale is the only one to use the term ‘parakinesia’ in a way that
Harten, 2011). However, the likelihood that anticholinergic medications is consistent with the original WKL understanding of the term, mixing
will improve ‘robotization’ remains debated among experts (A16). PPM with HPk (Bräunig et al., 2000).

3.3.3. Proposed criteria for PPM 4.1.1. Facial HPk and grimacing
The criteria selected for PPM are outlined in Table 3 and differential Facial HPk are likely to be identified as grimacing, i.e., “strange/
diagnoses are discussed in Supplementary material no 1 (Addendum no. bizarre facial expressions” that are brief or sustained (Bräunig et al.,
3). 2000; Bush et al., 1996; Northoff et al., 1999). Yet, the overlap between
HPk and grimacing is imperfect. For instance, HPk, which appears
3.4. The perception of HPk and PPM in contemporary psychiatry similar to an emotional expression, would be qualified as inappropriate
affect rather than grimacing, and the most widely used catatonia rating
The experts acknowledged HPk and PPM to be useful in their clinical scale only includes sustained forms of grimaces, which are not the most
practice (S47–48), and believed them to be acquired and heritable frequent forms of HPk (Bush et al., 1996).
phenomena (A20–23). Related phenomena are distinguished in some scales: (i) the Roger­
The experts observed that HPk and PPM are rarely noticed by col­ s–McKenna scale includes an item for facial dyskinesias and an item for
leagues exclusively trained in the ICD/DSM tradition (S50); and that grimacing (McKenna et al., 1991); (ii) Rogers–McKenna and Bräunig
when HPk is noticed, ICD/DSM-trained colleagues are more likely to scales include specific items for an increased blinking rate (Bräunig
interpret it as TD (S51) or tics (A26) than to interpret it as catatonic et al., 2000; McKenna et al., 1991); (iii) except for the Bush–Francis
manifestations, e.g., ‘mannerism’ (A27) or ‘grimacing’ (S52). scale, the other scales have items that could account for HPk involving
The experts also believed that ‘spontaneous dyskinesia’ in SSD re­ other parts of the body, but use distinct labels (see Fig. 3) (Bräunig et al.,
ported in the recent literature likely corresponds to HPk (S15). HPk has 2000; McKenna et al., 1991; Northoff et al., 1999).
prognostic value (S49): as spontaneous dyskinesia, HPk can precede the
first psychotic episode by several years. HPk is also regarded as pre­ 4.1.2. PPM and mannerism
dictive for the persistence of residual symptoms in the medium to long Except for the Bräunig's scale that defines ‘parakinesia’ like PPM
term, especially for progression toward a state of chronic avolition (Bräunig et al., 2000), most features of PPM would be described under
formerly referred to as ‘pure defect syndrome’ (‘reine Defektsyndrome’) the label ‘mannerism’ in other catatonia scales: movements are clumsy,
(Foucher et al., 2018; Pfuhlmann et al., 2020; Stöber, 2001). Finally, as odd, and unnatural (Bush et al., 1996; McKenna et al., 1991; Northoff
spontaneous dyskinesia, HPk and PPM are thought to be heritable et al., 1999). Northoff's definition is probably the closest to that of PPM,
because of their association to PPC (A21–23). qualifying gestures as “disharmonious,” whereas the Rogers–McKenna
and Bush–Francis scales emphasize the amped-up, exaggerated, and
4. Discussion caricatural aspects that only appear in hyperkinetic PPM.
However, if hyper- and normokinetic forms of PPM are probably well
This article proposed the first consensus on concepts designed to captured by the concept of mannerism, the hypokinetic form is not.
capture the phenomenology of HPk and PPM that are abnormal move­ Because the hypokinetic form mostly reduces expressivity, it might be
ments intrinsic to SSDs and have been described long before the anti­ more likely incorporated into the ICD/DSM-understanding of blunted/
psychotic era. We will first discuss the relationship between HPk, PPM flat affect. There is an important difference however: in ‘flat affect’, the
and classical catatonic phenomenology, i.e. grimacing and mannerism. absence of expressive motion is taken as an objective marker of the
In the second part we will focus on HPk, its concordant prevalence with absence of emotion. This is not the case of hypokinetic PPM in which the
absence of expression is precisely NOT accompanied by an absence of
Table 3 emotion. Although emotions may be dampened, they are never as
Proposed criteria for parakinetic psychomotricity. All criteria were considered blunted as the expressive PM might suggest. There is a dissociation be­
clinically important by the experts. tween subjective experience and objective expression of affects (Fig. 1.)
Main Deformation of expressive and reactive movements that
become disharmonious. 4.2. Same prevalence for spontaneous dyskinesia, grimacing and
Mostly apparent on the face, trunk, and upper limb girdle. phenotypes with HPk
Inhomogeneous distributiona of stiffness, clumsiness, or
jerkiness.
The neuropsychiatric tradition gives little value to independent
Changing aspects Hypokinesia Stiff, rigid, and stifled posture, gait, and
depending on PM gestures. phenomena and considers them to be meaningful only in association
excitation Normokinesia Clumsy, awkward, mannered with others, e.g., symptom complexes and phenotypes (Foucher et al.,
appearance. 2021). Hence, we are not aware of studies on the prevalence of HPk or
Hyperkinesia Eccentric, affected, pompous, artificial PPM in isolation. However, HPks are quite uniquely observed in PPC and
gestures and/or impulsive, hurried,
choppy, or jerky movements.
parakinetic catatonia (Addendum no. 1) the prevalence of which in
Mix Admixture of stiff and exaggerated hospitalized SSDs is of 14.5 % and 2 %, respectively. If the prevalence of
movement on different parts of the body HPk were the addition of the two, i.e. 16.5 %, it would be in line with: (i)
(quasi-specific to PPC). the prevalence of spontaneous dyskinesia in antipsychotic-naive SSDs,
a
The inhomogeneous nature of the stiffness or increase in amplitude and 15–20 % (Fenton et al., 1997; Peralta et al., 2010); and (ii) the preva­
abruptness should only be considered at a given moment. During the same lence of grimacing in acutely hospitalized SSDs, irrespective of the
interview, different parts of the body may be involved. treatment, at 17 % (Peralta and Cuesta, 2001). This reinforces experts'

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J.R. Foucher et al. Schizophrenia Research 263 (2024) 45–54

Fig. 3. Concept map of the various items from the four major catatonia scales that can be related with the concept of parakinesia (see Supplementary material no. 1;
numbers correspond to scale items). HPk: hyperkinetic parakinesia; PPM: parakinetic psychomotricity.

opinion in the connection between grimacing, spontaneous dyskinesia spontaneous dyskinesia suggest that they might refer to the same
and HPk. phenomenon.

4.3.3. A conflicting issue: PPC's HPk as ‘endogenous’ movement disorder in

4.3. HPk as part of PPC treated SSDs?
While the current concept of spontaneous dyskinesia accepts hy­
4.3.1. PPC's HPk are overlooked in clinical practice perkinetic movement disorder as intrinsic to SSDs, it looks as if it was
The view that HPk are “poorly noticed by colleagues exclusively accepted only for drug-naïve patients. Once an antipsychotic has been
trained in the ICD/DSM tradition” or “mistaken for TD”, should not be initiated, the dogma of drug-induced origin of dyskinesias becomes as
taken as an arrogant criticism. As the saying goes, “people only see what unquestionable even today as it was in the 1980s, when only a few re­
they are prepared to see”, or as in this case, what they have been trained searchers challenged this dogma (Waddington and Crow, 1988). Wad­
to see. However, the retrospective re-assessment of 262 records kept of dington et al. (1987) went further and proposed to distinguish ‘sub-
PPC-patients according to ICD-10 suggests that glossing over HPk in forms’ of TD and found the one affecting orofacial rather than upper-
clinical practice is probably less a matter of knowing than that of limb muscles to be consistently associated with a flattening of affect
attention and interpretation. HPk or grimacing were mentioned in 12 % and severe cognitive dysfunction analogous to the residual syndrome of
at first hospitalizations (Krause, 2012) whereas it is unlikely that much PPC in which they are named HPk, hypokinetic PPM, avolition and
<85 % of first episode PPC would not have HPk. However, this low alogia. The analogy could be pushed further as they reported a pedigree,
frequency is also in contradiction with the prevalence of ‘grimacing’ in line with PPC heritability, in which several members not only pre­
when estimated from scales. Accordingly, HPks are probably overlooked sented the association but also other catatonic manifestations of PPC
or simply not mentioned because they are given little importance in (Waddington and Youssef, 1986). Interestingly, spontaneous dyskinesia
psychopathology underlying the ICD/DSM diagnostic systems relative to also prevails on the face both in schizotypal personality disorder (Mittal
psychotic and affective manifestations, i.e. missing motor symptoms is a and Walker, 2007) and untreated chronic SSDs (Owens et al., 1982).
problem of ‘attention’ and not that of ‘knowing’. Even more interesting These analogies are too crude to be conclusive, but they give new
was the further decrement in subsequent hospitalizations as HPk or impetus to the direction of research pursued by this paper. It may well be
grimacing were mentioned only in 8 % of the records (Krause, 2012). that these different approaches and concepts refer to the same existing
This is likely to be related to the introduction of pharmacotherapy, HPk but overlooked motor phenomena described by French and German
being either masked by, or wrongly attributed to antipsychotics, i.e. neuropsychiatrists in the pre-neuroleptic era. We propose that the Del­
mistaken for TD. phi consensus on the phenomenology of HPk, PPM and their differential
diagnosis with TD outlined in this paper may offer new leads.
4.3.2. Heritability of HPk and spontaneous dyskinesia
PPC is highly heritable. Not only 27 % of first-degree relatives pre­
sent the (full) PPC phenotype, i.e., with frank psychotic episodes (Stöber 4.4. Strengths and limitations of the study
and Reis, 2009), but a further proportion of 14 % of first-degree relatives
present with an attenuated form, in which residual signs, such as HPk The strengths of the study include the re-discovery and re-
and/or schizotypal personality traits emerge without any previous interpretation of motor phenomena associated with psychotic disor­
psychotic episode (Foucher et al., 2020b; Pfuhlmann et al., 2020). ders described by classical French and German authors based on
Similar findings are reported for spontaneous dyskinesia: (i) it is painstaking, decade-long observations. For a combination of reasons
observed in 11–12 % of patients with schizotypal personality disorder that include cultural and language barriers and differing psychiatric
(Cassady et al., 1998; Fenton et al., 1997); (ii) and 14 % of first-degree traditions, these observations have not been incorporated in mainstream
relatives of patients with a psychotic disorder (McCreadie et al., 2003). clinical and research practice in the currently dominant English-
Moreover, as it is the case in PPC's HPk, spontaneous dyskinesia tends to language psychiatry. This study also managed to assembly a group of
progress assessed at the 1- and 2-year follow-up examinations and be clinicians/researchers who have become familiar with the classical
predictive of transition to psychosis (Mittal et al., 2008; Mittal and concepts of psychomotor disorders during their training.
Walker, 2007). The many similarities between PPC's HPk and However, the limitations of this study should also be acknowledged.

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J.R. Foucher et al. Schizophrenia Research 263 (2024) 45–54

We had to take video examples from the internet because we did not Acknowledgments
have the consent of our current patients for public release in the form of
publication. Hence, even if all experts agreed, the videos' illustrative We would like to pay our gratitude and respect to Prof. Dr. Gerald
value must be taken with caution. Firstly, except the first example taken Stöber, who passed away due to a tragic accident in June 2017. Prof. Dr.
from Kleist's original teaching films, the confidence in the last two cases Stöber was president of the Wernicke–Kleist–Leonhard International
is limited by the paucity of information about the patient and the lack of Society and a professor of psychiatry at the University of Würzburg
direct examination. Secondly, by failing to cover the patients' whole (Germany), where he made major advances in the heritability and ge­
phenomenological presentation, this limited set of examples might bias netics of relapsing-progressive periodic catatonia. This article owes
the representation of the phenomenon. We encourage anyone curious much to the finesse of his clinical skills and passionate investment in
about parakinesia to contact us. We have many videos of patients with teaching psychopathology with the greatest respect for the patients.
parakinesia, including those from our German colleagues covering Finally, the authors would like to express their deep appreciation to
several decades of the life of the same patient. Although these could not Ms. Tracy Ungvari for her help in editing the manuscript.
be used in this article, our patients consented to share the videos with a
selected professional audience. Many such videos have already been Appendix A. Supplementary data
discussed with experts in catatonia and an expert in movement disorders
who contributed to the writing of this article (LV). Any constructive Supplementary data to this article can be found online at https://doi.
discussion about them will be welcomed. org/10.1016/j.schres.2022.09.024.
Finally, the separate account of HPk and PPM does not mean that
they are separate phenomena. This distinction primarily served a prac­ References
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