Abstracts
Psychogenic nonepileptic seizures (PNES) and psychogenic movement disorders (PMD) are commonly seen in Neurology practice and are categorized in the DSM-5 as functional neurological disorders/conversion disorders. This review encompasses historical and epidemiological data, clinical aspects, diagnostic criteria, treatment and prognosis of these rather challenging and often neglected patients. As a group they have puzzled generations of neurologists and psychiatrists and in some ways continue to do so, perhaps embodying and justifying the ultimate and necessary link between these specialties.
psychogenic nonepileptic seizures; psychogenic movement disorders; conversion disorder; hysteria
Crises não-epilépticas psicogênicas (CNEP) e distúrbios do movimento psicogênicos (DMP) são comuns na prática e na atualidade são melhor categorizados no DSM-V como distúrbios neurológicos funcionais/desordens de conversão. Esta revisão enfatiza os principais dados históricos, epidemiológicos, clínicos, critérios diagnósticos, tratamento e o prognóstico destes pacientes, frequentemente negligenciados e desafiadores, os quais, como um grupo, tem intrigado gerações de neurologistas e psiquiatras, caracterizando, de forma justificada o elo definitivo entre estas especialidades.
crises não-epilépticas psicogênicas; distúrbios do movimento psicogênicos; distúrbio conversivo; histeria
In the medical sciences, a diagnosis is established when a level of certainty regarding
the nature of a condition is achieved. Psychogenic nonepileptic seizures (PNES) and
psychogenic movement disorders (PMD) share a slightly less linear diagnostic route.
Historically, making a conversion diagnosis required ruling out the originally
considered medical diagnosis, i.e., it is neither epilepsy, nor a neurogenic movement
disorder. Only then a non-neurologic (in the sense of
not resulting from epileptogenic or known motor circuitry
imbalance) condition is suspected and raised to the level of diagnosis11 . Edwards M. Historical keyword: hysteria [editorial].
Lancet 2009(9702);374:1669.
http://dx.doi.org/10.1016/S0140-6736(09)61979-6
https://doi.org/10.1016/S0140-6736(09)61...
. This kind of diagnosis has intrigued
generations of physicians and spared no culture, gender or age. A diversity of symptoms,
including motor, sensory, cognitive, and/or behavior symptoms are present in these
presentations. Patients with PNES or PMD frequently are misdiagnosed as having their
initial suspected neurological diseases, and ultimately pose a diagnostic and treatment
challenge to many clinicians. Interestingly, it was perhaps this varied presentation
that has captured the attention of many professionals through the centuries and lead to
the construction and recognition of a very particular diagnosis. Two rather advanced
historic civilizations, Egyptians first, and then the Greeks, posited that a displaced
hystera (womb), and the consequent suffocation that it produced,
was the source of choking, mutism, paralysis and fainting11 . Edwards M. Historical keyword: hysteria [editorial].
Lancet 2009(9702);374:1669.
http://dx.doi.org/10.1016/S0140-6736(09)61979-6
https://doi.org/10.1016/S0140-6736(09)61...
. Hysteria, a natural development on this concept, was
claimed as an explanation for similar phenomena in women and reached almost epidemic
proportions in the late 19th century. By then, French and British women
displayed a rather elevated prevalence of hysteria, which was associated with the work
of Jean Martin Charcot (Figure 1). His harshest
critics proposed that he created this disease and gained profit from it22 . Goetz CG, Bonduelle M, Gelfand T. Hysteria. In: Goetz CG,
Bonduelle M, Gelfand T. Charcot. Constructing neurology. New York: Oxford
University Press; 1995. p. 172-216.. From a contemporary perspective, it
may be tempting to scrutinize the dramatic performances of some of Charcot’s
young female patients, who may have been prone to suggestibility of the famous
professor’s hypnosis techniques22 . Goetz CG, Bonduelle M, Gelfand T. Hysteria. In: Goetz CG,
Bonduelle M, Gelfand T. Charcot. Constructing neurology. New York: Oxford
University Press; 1995. p. 172-216.. It was due to Charcot’s astute clinical observations,
however, that hysteria moved from an almost gynecological disease to a neurologically
conceived condition. Regarding male hysteria, Charcot followed after Galen (who
suggested that retained sperm could lead to male hysteria) and later Charles LePois,
Briquet and Savill, to offer a full description of hysteria in men (Charcot even
mentioned epidemiological data, suggesting that 5% of his hysterical patients were
male)22 . Goetz CG, Bonduelle M, Gelfand T. Hysteria. In: Goetz CG,
Bonduelle M, Gelfand T. Charcot. Constructing neurology. New York: Oxford
University Press; 1995. p. 172-216.. The
“shell-shock” syndrome described in the World War I trenches
consubstantiated the concept of “male hysteria”. Hysteria, in all
fairness, respects no gender, i.e. somatoform symptoms are present in women and men33 . Linden SC, Jones E, Lees AJ. Shell shock at queen square: Lewis
Yealland 100 years on. Brain. 2013;136(Pt 6):1976-88.
http://dx.doi.org/10.1093/brain/aws331
https://doi.org/10.1093/brain/aws331...
. In spite of the controversy produced
by historical reflections and opinions on Charcot’s motives and deep involvement
with the field of hysteria, these patients finally came to light in a somewhat
“medically organized” fashion and certainly inspired very prestigious
minds eager to take this diagnosis to a different level of understanding22 . Goetz CG, Bonduelle M, Gelfand T. Hysteria. In: Goetz CG,
Bonduelle M, Gelfand T. Charcot. Constructing neurology. New York: Oxford
University Press; 1995. p. 172-216.. Sigmund Freud spent four months at the
Salpêtrière between October, 1885 and February, 1886 and his enthusiasm for
neurosis and psychology was certainly the product of this stay22 . Goetz CG, Bonduelle M, Gelfand T. Hysteria. In: Goetz CG,
Bonduelle M, Gelfand T. Charcot. Constructing neurology. New York: Oxford
University Press; 1995. p. 172-216.. His attention is observed in that over 100 out of the
3000 books in Freud’s private library were dedicated to hysteria and hypnosis,
and all were authored by either Charcot himself or one of the French professor’s
direct pupils22 . Goetz CG, Bonduelle M, Gelfand T. Hysteria. In: Goetz CG,
Bonduelle M, Gelfand T. Charcot. Constructing neurology. New York: Oxford
University Press; 1995. p. 172-216.. Just prior to
Charcot’s death, Charcot acknowledged that he had been mistaken and that hysteria
was truthfully a psychiatric disease. By then, Alienism was in the process of becoming
the modern psychiatry22 . Goetz CG, Bonduelle M, Gelfand T. Hysteria. In: Goetz CG,
Bonduelle M, Gelfand T. Charcot. Constructing neurology. New York: Oxford
University Press; 1995. p. 172-216.. Hysteria fell
into academic disgrace, raising derogatory connotations and the word itself was removed
to the lay domain. The condition was renamed by Babinski to pithiatism; the number of
publications on hysteria decreased significantly and at a given point it has been
considered an excuse for lack of medical knowledge or poor diagnostic skills. Renowned
publications such as the British Medical Journal released papers in the 60’s
reflecting the thoughts of authors calling hysteria the “disguise for ignorance
and fertile source for clinical errors”44 . Stone J, Warlow C, Carson A, Sharpe M. Eliot Slater’s Myth
of the non-existence of hysteria. J R Soc Med. 2006;98(12):547-8.
http://dx.doi.org/10.1258/jrsm.98.12.547
https://doi.org/10.1258/jrsm.98.12.547...
. This assumption of diagnostic ignorance was proven
incorrect with the advent of advanced diagnostic techniques. In the 1970’s and
1980’s, the birth of epilepsy monitoring units and the technology to document
patients and their events for long periods of time surfaced literally all sorts of
epilepsy “imitators”, including PNES55 . Rowan AJ. An introduction to current practice in the diagnosis of
non-epileptic seizures. In: Gates JR, Rowan AJ. Non-epileptic seizures. Boston:
Butteworth-Heinemann; 1993. p. 1-8.. Some of them, such as paroxysmal, transitory, tremor-like
or tic-like episodes without loss of consciousness can be interpreted as either PNES or
PMD, depending on the setting and context that they are disclosed. What was
“hysteria” in the past, today has different nomenclatures (e.g. medically
unexplained symptoms, somatoform disorders, etc.). As any practicing clinician knows,
the disorder clearly still presents itself in the 21st century in the
symptoms and semiology of a substantial percentage of patients in both epilepsy and
movement disorders clinics. Conversion symptoms very infrequently can be a sole
phenomenon, however, the vast majority of patients have a psychiatric comorbidity and/or
a significant stressor, either recently or remotely. In this paper, the authors intend
to review and explore both commonalities, as well as, unique expressions that are
present in PNES and PMD. We will systematically discuss PNES and PMD as independent
conditions that might share similar natures and etiologies, and hopefully, offer a
systematic approach to general management and treatment.
FUNCTIONAL NEUROLOGICAL DISORDERS (CONVERSION DISORDERS)
Psychogenic symptoms, or, functional symptoms, are commonly seen in clinical practice
and are estimated to represent 10% of all medical complaints66 . Hinson VK, Haren WB. Psychogenic movement disorders. Lancet
Neurol. 2006;5(8):695-700.
http://dx.doi.org/10.1016/S1474-4422(06)70523-3
https://doi.org/10.1016/S1474-4422(06)70...
,77 . Edwards MJ, Bhatia KP. Functional (psychogenic movement disorders:
merging mind and brain. Lancet Neurol. 2012;11(3):250-60.
http://dx.doi.org/10.1016/S1474-4422(11)70310-6
https://doi.org/10.1016/S1474-4422(11)70...
,88 . Lempert T, Dietrich M, Huppert D, Brandt T. Psychogenic disorders
in neurology: frequency and clinical spectrum. Acta Neurol Scand.
1990;82(5):335-40.
http://dx.doi.org/10.1111/j.1600-0404.1990.tb03312.x
https://doi.org/10.1111/j.1600-0404.1990...
,99 . Stone J, LaFrance Jr WC, Levenson JL, Sharpe M. Issues for DSM-V:
conversion disorder. Am J Psychiatry. 2010;167(6):626-7.
http://dx.doi.org/10.1176/appi.ajp.2010.09101440
https://doi.org/10.1176/appi.ajp.2010.09...
. They are common in neurology, accounting for 10 to 33% of
patient visits to a neurologist and 9% of inpatient neurology admissions66 . Hinson VK, Haren WB. Psychogenic movement disorders. Lancet
Neurol. 2006;5(8):695-700.
http://dx.doi.org/10.1016/S1474-4422(06)70523-3
https://doi.org/10.1016/S1474-4422(06)70...
7 . Edwards MJ, Bhatia KP. Functional (psychogenic movement disorders:
merging mind and brain. Lancet Neurol. 2012;11(3):250-60.
http://dx.doi.org/10.1016/S1474-4422(11)70310-6
https://doi.org/10.1016/S1474-4422(11)70...
-88 . Lempert T, Dietrich M, Huppert D, Brandt T. Psychogenic disorders
in neurology: frequency and clinical spectrum. Acta Neurol Scand.
1990;82(5):335-40.
http://dx.doi.org/10.1111/j.1600-0404.1990.tb03312.x
https://doi.org/10.1111/j.1600-0404.1990...
. The importance of the exam in conversion disorder
(CD) has been underscored with the inclusion of the presence of these signs in the
diagnostic criteria of CD in DSM-51010 . American Psychiatric Association. Diagnostic and statistical
manual of mental disorders. 5th ed. (DSM-V). Arlington: American Psychiatry
Publishing; 2013.. Several neurological symptoms can have psychogenic
mechanisms underlying their presentation, including paralysis, sensory loss,
blindness, astasia-abasia, amnesia, PNES and PMD66 . Hinson VK, Haren WB. Psychogenic movement disorders. Lancet
Neurol. 2006;5(8):695-700.
http://dx.doi.org/10.1016/S1474-4422(06)70523-3
https://doi.org/10.1016/S1474-4422(06)70...
7 . Edwards MJ, Bhatia KP. Functional (psychogenic movement disorders:
merging mind and brain. Lancet Neurol. 2012;11(3):250-60.
http://dx.doi.org/10.1016/S1474-4422(11)70310-6
https://doi.org/10.1016/S1474-4422(11)70...
8 . Lempert T, Dietrich M, Huppert D, Brandt T. Psychogenic disorders
in neurology: frequency and clinical spectrum. Acta Neurol Scand.
1990;82(5):335-40.
http://dx.doi.org/10.1111/j.1600-0404.1990.tb03312.x
https://doi.org/10.1111/j.1600-0404.1990...
-99 . Stone J, LaFrance Jr WC, Levenson JL, Sharpe M. Issues for DSM-V:
conversion disorder. Am J Psychiatry. 2010;167(6):626-7.
http://dx.doi.org/10.1176/appi.ajp.2010.09101440
https://doi.org/10.1176/appi.ajp.2010.09...
. Daum et al. performed a systematic and narrative review
about the value of “positive” clinical signs for weakness, sensory and
gait disorders in conversion disorder1111 . Daum C, Hubschmid M, Aybek S. The value of
“positive” clinical signs for weakness, sensory and gait disorders
in conversion disorder: a systematic and narrative review. J Neurol Neurosurg
Psychiatry. 2014;85(2):180-90.
http://dx.doi.org/10.1136/jnnp-2012-304607
https://doi.org/10.1136/jnnp-2012-304607...
. The authors did not study signs for PNES. They concluded
that clinical signs for motor, sensory and gait functional neurological symptoms are
numerous, and 14 have been validated (7 motor, 5 sensory, and 2 gait related)1111 . Daum C, Hubschmid M, Aybek S. The value of
“positive” clinical signs for weakness, sensory and gait disorders
in conversion disorder: a systematic and narrative review. J Neurol Neurosurg
Psychiatry. 2014;85(2):180-90.
http://dx.doi.org/10.1136/jnnp-2012-304607
https://doi.org/10.1136/jnnp-2012-304607...
. Among positive signs of
functional motor, sensory and gait disorders, Hoover sign, abductor sign, abductor
finger sign, co-contraction, midline splitting, non-anatomical sensory loss,
dragging monoplegic gait, and chair test have been validated1111 . Daum C, Hubschmid M, Aybek S. The value of
“positive” clinical signs for weakness, sensory and gait disorders
in conversion disorder: a systematic and narrative review. J Neurol Neurosurg
Psychiatry. 2014;85(2):180-90.
http://dx.doi.org/10.1136/jnnp-2012-304607
https://doi.org/10.1136/jnnp-2012-304607...
. Daum et al. also reviewed non-validated,
positive signs of functional disorders, which included non-pyramidal weakness,
absent pronator drift, arm drop test, Barré test, platysme sign, Babinski
trunk-thigh test, Bowlus-Currier test, excessive slowness, fluctuation, psychogenic
Romberg test, walking on ice, sudden knee buckling, astasia-abasia, and expressive
behavior1111 . Daum C, Hubschmid M, Aybek S. The value of
“positive” clinical signs for weakness, sensory and gait disorders
in conversion disorder: a systematic and narrative review. J Neurol Neurosurg
Psychiatry. 2014;85(2):180-90.
http://dx.doi.org/10.1136/jnnp-2012-304607
https://doi.org/10.1136/jnnp-2012-304607...
. Lombardi et al.
published on a sign to detect unilateral upper extremity non-organic paresis, the
elbow flex-ex1212 . Lombardi TL, Barton E, Wang J, Eliashiv DS, Chung JM, Muthukumaran
A et al. The elbow flex-ex: a new sign to detect unilateral upper extremity
non-organic paresis. J Neurol Neurosurg Psychiatry. 2014;85(2):165-7.
http://dx.doi.org/10.1136/jnnp-2012-304607
https://doi.org/10.1136/jnnp-2012-304607...
. This test is
useful in differentiating between functional and non-neurologic arm paresis1212 . Lombardi TL, Barton E, Wang J, Eliashiv DS, Chung JM, Muthukumaran
A et al. The elbow flex-ex: a new sign to detect unilateral upper extremity
non-organic paresis. J Neurol Neurosurg Psychiatry. 2014;85(2):165-7.
http://dx.doi.org/10.1136/jnnp-2012-304607
https://doi.org/10.1136/jnnp-2012-304607...
.
Functional (psychogenic) neurological disorders/CD have been described as hysteria,
somatization disorder, non-organic disorders and medically unexplained symptoms77 . Edwards MJ, Bhatia KP. Functional (psychogenic movement disorders:
merging mind and brain. Lancet Neurol. 2012;11(3):250-60.
http://dx.doi.org/10.1016/S1474-4422(11)70310-6
https://doi.org/10.1016/S1474-4422(11)70...
,1111 . Daum C, Hubschmid M, Aybek S. The value of
“positive” clinical signs for weakness, sensory and gait disorders
in conversion disorder: a systematic and narrative review. J Neurol Neurosurg
Psychiatry. 2014;85(2):180-90.
http://dx.doi.org/10.1136/jnnp-2012-304607
https://doi.org/10.1136/jnnp-2012-304607...
,1313 . Rowe JB. Conversion disorder: understanding the pathogenic links
between emotion and motor systems in the brain. Brain. 2010;133(Pt 5):1295-9.
http://dx.doi.org/10.1093/brain/awq096
https://doi.org/10.1093/brain/awq096...
. Stone et al., evaluated 1144 new neurology
outpatients with symptoms “unexplained by organic disease”1414 . Stone J, Carson A, Duncan R, Coleman R, Roberts R, Warlow C et al.
Symptoms “unexplained by organic disease” in 1144 new neurology
out-patients: how often does the diagnosis change at follow-up? Brain.
2009;132(Pt 10):2878-88. http://dx.doi.org/10.1093/brain/awp220
https://doi.org/10.1093/brain/awp220...
. The most common diagnoses were
neurological disease but with symptoms unexplained by this condition (26%), headache
disorders (26%) and conversion symptoms (motor, sensory or non-epileptic seizures)
(18%)1414 . Stone J, Carson A, Duncan R, Coleman R, Roberts R, Warlow C et al.
Symptoms “unexplained by organic disease” in 1144 new neurology
out-patients: how often does the diagnosis change at follow-up? Brain.
2009;132(Pt 10):2878-88. http://dx.doi.org/10.1093/brain/awp220
https://doi.org/10.1093/brain/awp220...
. At follow-up, only
0.4% of 1030 patients had an organic disease diagnosis confirmed. The authors
concluded that one-third of new neurology outpatients were diagnosed as having
symptoms “unexplained by organic disease”1414 . Stone J, Carson A, Duncan R, Coleman R, Roberts R, Warlow C et al.
Symptoms “unexplained by organic disease” in 1144 new neurology
out-patients: how often does the diagnosis change at follow-up? Brain.
2009;132(Pt 10):2878-88. http://dx.doi.org/10.1093/brain/awp220
https://doi.org/10.1093/brain/awp220...
.
Structural neuroimaging does not reveal a “conversion lesion”.
Unexplained neurological symptoms, however, are being studied using functional MRI.
In general, studies using this technique have demonstrated that active inhibition in
the orbitofrontal cortex and cingulate gyrus may be implicated in functional
disorders1515 . Hurwitz TA, Prichard JW. Conversion disorder and fMRI. Neurology.
2006;67(11):1914-815.
http://dx.doi.org/10.1212/01.wnl.0000249127.69348.a4
https://doi.org/10.1212/01.wnl.000024912...
. Different
subtypes of FNDs are shown in Figure 2. The
most prevalent psychogenic conditions in practical clinical neurology are generally
PNES and PMD66 . Hinson VK, Haren WB. Psychogenic movement disorders. Lancet
Neurol. 2006;5(8):695-700.
http://dx.doi.org/10.1016/S1474-4422(06)70523-3
https://doi.org/10.1016/S1474-4422(06)70...
,88 . Lempert T, Dietrich M, Huppert D, Brandt T. Psychogenic disorders
in neurology: frequency and clinical spectrum. Acta Neurol Scand.
1990;82(5):335-40.
http://dx.doi.org/10.1111/j.1600-0404.1990.tb03312.x
https://doi.org/10.1111/j.1600-0404.1990...
.
PSYCHOGENIC NON-EPILEPTIC SEIZURES
Non-epileptic seizures (NES) are characterized by paroxysmal, involuntary, usually
time-limited alterations in motor and/or sensory function, level of consciousness
and behavior that may resemble epileptic seizures (ES), but are not caused by
epileptic discharges1616 . Gates JR. Epidemiology and classification of non-epileptic events.
In: Gates JR, Rowan AJ, editors. Non-epileptic seizures. 2th ed. Boston:
Butterworth-Heinemann; 2000. p. 3-14.. They can
be of physiologic or psychogenic (PNES) origin. The most common causes of
physiologic non-epileptic events, such as syncope1616 . Gates JR. Epidemiology and classification of non-epileptic events.
In: Gates JR, Rowan AJ, editors. Non-epileptic seizures. 2th ed. Boston:
Butterworth-Heinemann; 2000. p. 3-14., do not pose a significant problem to differential
diagnosis when video-electroencephalography (VEEG) monitoring is used1616 . Gates JR. Epidemiology and classification of non-epileptic events.
In: Gates JR, Rowan AJ, editors. Non-epileptic seizures. 2th ed. Boston:
Butterworth-Heinemann; 2000. p. 3-14.. A long list of mental disorders
may present as PNES1616 . Gates JR. Epidemiology and classification of non-epileptic events.
In: Gates JR, Rowan AJ, editors. Non-epileptic seizures. 2th ed. Boston:
Butterworth-Heinemann; 2000. p. 3-14.. Patients
typically present with episodes characterized by disrupted consciousness or
motor/sensory manifestations. Although deficits and symptoms can be produced
voluntarily, as in factitious disorders and malingering (found in a small percentage
of individuals with neurologic presentations), psychogenic disorder presentations
are involuntary (unconsciously produced), as in patients with somatoform disorders
and dissociative disorders1616 . Gates JR. Epidemiology and classification of non-epileptic events.
In: Gates JR, Rowan AJ, editors. Non-epileptic seizures. 2th ed. Boston:
Butterworth-Heinemann; 2000. p. 3-14..
Several putative psychological mechanisms of PNES have been conceptualized and
described1717 . LaFrance Jr, WC, Bjornaes, H. Designing treatment plans based on
etiology of psychogenic nonepileptic seizures. In: Schachter, SC, LaFrance Jr,
WC, editors. Gates & Rowan’s Nonepileptic seizures. 3 rd ed. New
York: Cambridge University Press; 2010. p. 266-80.. Some of the
mechanisms include a psychodynamic model of primary gain (intended to solve a
dilemma, escape an intolerable situation or reduce anxiety), secondary gain
(directed to obtain affective or social benefits), a behavioral model of
reinforcement of illness behavior (by attention given to symptoms, for instance),
and psychosocial model of maladaptive coping responses to stress (due to
deficiencies of coping style, for example)1717 . LaFrance Jr, WC, Bjornaes, H. Designing treatment plans based on
etiology of psychogenic nonepileptic seizures. In: Schachter, SC, LaFrance Jr,
WC, editors. Gates & Rowan’s Nonepileptic seizures. 3 rd ed. New
York: Cambridge University Press; 2010. p. 266-80.. Dissociation, suggestion and hypnotizability
mechanisms also act anchored in pathogenic beliefs that suggest to the patient a
severe disease. Suggestion and dissociation also is found in the unconscious
modeling of symptoms by the patient, based in his personal past experience or in his
conceptualization of the medical problem1818 . Marchetti RL, Kurcgant D, Gallucci Neto J, Bismark MA, Marchetti
LB, Fiore LA. Psychiatric diagnoses of patients with psychogenic non-epileptic
seizures. Seizure. 2008;17(3):247-53.
http://dx.doi.org/10.1016/j.seizure.2007.07.006
https://doi.org/10.1016/j.seizure.2007.0...
.
EPIDEMIOLOGY OF PNES
Studies on the prevalence of PNES show variable but clinically significant results,
from five to 33% of outpatients receiving treatment for epilepsy, and from 10 to 58%
of inpatients treated for refractory epilepsy present PNES1919 . Benbadis SR, Hauser AW. An estimate of the prevalence of
psychogenic non-epileptic seizures. Seizures 2000;9(4):280-1.
http://dx.doi.org/10.1053/seiz.2000.0409
https://doi.org/10.1053/seiz.2000.0409...
. According to Gates1616 . Gates JR. Epidemiology and classification of non-epileptic events.
In: Gates JR, Rowan AJ, editors. Non-epileptic seizures. 2th ed. Boston:
Butterworth-Heinemann; 2000. p. 3-14. such a significant difference in results may be
explained by differences in diagnostic criteria for PNES. A female preponderance of
up to 80% has been observed in studies of patients with PNES55 . Rowan AJ. An introduction to current practice in the diagnosis of
non-epileptic seizures. In: Gates JR, Rowan AJ. Non-epileptic seizures. Boston:
Butteworth-Heinemann; 1993. p. 1-8.,1616 . Gates JR. Epidemiology and classification of non-epileptic events.
In: Gates JR, Rowan AJ, editors. Non-epileptic seizures. 2th ed. Boston:
Butterworth-Heinemann; 2000. p. 3-14.,1717 . LaFrance Jr, WC, Bjornaes, H. Designing treatment plans based on
etiology of psychogenic nonepileptic seizures. In: Schachter, SC, LaFrance Jr,
WC, editors. Gates & Rowan’s Nonepileptic seizures. 3 rd ed. New
York: Cambridge University Press; 2010. p. 266-80.,1919 . Benbadis SR, Hauser AW. An estimate of the prevalence of
psychogenic non-epileptic seizures. Seizures 2000;9(4):280-1.
http://dx.doi.org/10.1053/seiz.2000.0409
https://doi.org/10.1053/seiz.2000.0409...
. PNES is present in children and elderly people, but
many patients’ age range between the 20’s and 30’s2020 . Reuber M, Pukrop R, Bauer J, Helmstaedter C, Tessendorf N, Elger
CE. Outcome in psychogenic nonepileptic seizures: 1 to 10-year follow-up in 164
patients. Ann Neurol. 2003;53(3):305-11.
http://dx.doi.org/10.1002/ana.3000
https://doi.org/10.1002/ana.3000...
.
IMPACT OF PNES
PNES is as disabling as epilepsyand may lead to severe social and psychological
impairments2020 . Reuber M, Pukrop R, Bauer J, Helmstaedter C, Tessendorf N, Elger
CE. Outcome in psychogenic nonepileptic seizures: 1 to 10-year follow-up in 164
patients. Ann Neurol. 2003;53(3):305-11.
http://dx.doi.org/10.1002/ana.3000
https://doi.org/10.1002/ana.3000...
. Patients are
also exposed to iatrogenic procedures, such as high doses of AEDs, intravenous AED
use, and orotracheal intubation2121 . Brown RJ, Syed TU, Benbadis S, LaFrance WC Jr, Reuber M.
Psychogenic nonepileptic seizures. Epilepsy Behav. 2011;22(1):85-93.
http://dx.doi.org/10.1016/j.yebeh.2011.02.016
https://doi.org/10.1016/j.yebeh.2011.02....
.
Quality of life (QoL) measures in patients with PNES reveal that QoL is worse than
that of patients with refractory epilepsyand that QoL is related to symptoms and
depression2121 . Brown RJ, Syed TU, Benbadis S, LaFrance WC Jr, Reuber M.
Psychogenic nonepileptic seizures. Epilepsy Behav. 2011;22(1):85-93.
http://dx.doi.org/10.1016/j.yebeh.2011.02.016
https://doi.org/10.1016/j.yebeh.2011.02....
. The diagnostic
delay in PNES patients has been reported in the past an average of 7.2 years,
however, with increasing awareness of PNES by providers and patients with seizures,
and the increased access to seizure monitoring units, the time to correct diagnosis
is decreasing, considerably2121 . Brown RJ, Syed TU, Benbadis S, LaFrance WC Jr, Reuber M.
Psychogenic nonepileptic seizures. Epilepsy Behav. 2011;22(1):85-93.
http://dx.doi.org/10.1016/j.yebeh.2011.02.016
https://doi.org/10.1016/j.yebeh.2011.02....
.
CLINICAL PATH TO DIAGNOSIS OF PNES
The possibility of PNES being present is usually considered when there is a complete
absence of therapeutic response to AED, loss of response (therapeutic failure), or
paradoxical responses (worsening or spontaneous and unexpected remissions).
Likewise, PNES may be considered because of atypical, multiple, inconsistent or
changing seizure patterns, or when the seizures are provoked by evident and specific
emotional stress, with a narrow temporal relation to seizure occurrence2121 . Brown RJ, Syed TU, Benbadis S, LaFrance WC Jr, Reuber M.
Psychogenic nonepileptic seizures. Epilepsy Behav. 2011;22(1):85-93.
http://dx.doi.org/10.1016/j.yebeh.2011.02.016
https://doi.org/10.1016/j.yebeh.2011.02....
,2222 . Avbersek A, Sisodiya S. Does the primary literature provide
support for clinical signs used to distinguish psychogenic nonepileptic seizures
from epileptic seizures? J Neurol Neurosurg. 2010;81(7):719-25.
http://dx.doi.org/10.1136/jnnp.2009.197996
https://doi.org/10.1136/jnnp.2009.197996...
. Table 1
summarizes important clinical semiologic features of PNES that help distinguish it
from epileptic seizures. These elements are considered particularly when the patient
demonstrates normal ancillary exams, (interictal routine EEGs, and neuroimaging
studies, such as brain CT, MRI and SPECT)2222 . Avbersek A, Sisodiya S. Does the primary literature provide
support for clinical signs used to distinguish psychogenic nonepileptic seizures
from epileptic seizures? J Neurol Neurosurg. 2010;81(7):719-25.
http://dx.doi.org/10.1136/jnnp.2009.197996
https://doi.org/10.1136/jnnp.2009.197996...
. The suspected diagnosis raised by a neurologist has a
positive predictive value of 84.6% for PNES2222 . Avbersek A, Sisodiya S. Does the primary literature provide
support for clinical signs used to distinguish psychogenic nonepileptic seizures
from epileptic seizures? J Neurol Neurosurg. 2010;81(7):719-25.
http://dx.doi.org/10.1136/jnnp.2009.197996
https://doi.org/10.1136/jnnp.2009.197996...
. Patients with PNES tend to have a greater frequency
of seizures than do epilepsy patients2121 . Brown RJ, Syed TU, Benbadis S, LaFrance WC Jr, Reuber M.
Psychogenic nonepileptic seizures. Epilepsy Behav. 2011;22(1):85-93.
http://dx.doi.org/10.1016/j.yebeh.2011.02.016
https://doi.org/10.1016/j.yebeh.2011.02....
,2222 . Avbersek A, Sisodiya S. Does the primary literature provide
support for clinical signs used to distinguish psychogenic nonepileptic seizures
from epileptic seizures? J Neurol Neurosurg. 2010;81(7):719-25.
http://dx.doi.org/10.1136/jnnp.2009.197996
https://doi.org/10.1136/jnnp.2009.197996...
. They also have a greater frequency of hospital admissions
due to prolonged seizures, or nonepileptic status2121 . Brown RJ, Syed TU, Benbadis S, LaFrance WC Jr, Reuber M.
Psychogenic nonepileptic seizures. Epilepsy Behav. 2011;22(1):85-93.
http://dx.doi.org/10.1016/j.yebeh.2011.02.016
https://doi.org/10.1016/j.yebeh.2011.02....
. The occurrence of seizures in the physician’s
office or in the waiting room is very suggestive of PNES, as well as a history of
unexplained “chronic pain” or “fibromyalgia”2121 . Brown RJ, Syed TU, Benbadis S, LaFrance WC Jr, Reuber M.
Psychogenic nonepileptic seizures. Epilepsy Behav. 2011;22(1):85-93.
http://dx.doi.org/10.1016/j.yebeh.2011.02.016
https://doi.org/10.1016/j.yebeh.2011.02....
. Antecedent trauma is reported by
up to 70% of PNES patients, and sexual abuse in 40%, which may be an
underestimate2121 . Brown RJ, Syed TU, Benbadis S, LaFrance WC Jr, Reuber M.
Psychogenic nonepileptic seizures. Epilepsy Behav. 2011;22(1):85-93.
http://dx.doi.org/10.1016/j.yebeh.2011.02.016
https://doi.org/10.1016/j.yebeh.2011.02....
,2323 . Proença IC, Castro LH, Jorge CL, Marchetti RL. Emotional
trauma and abuse in patients with psychogenic nonepileptic seizures. Epilepsy
Behav. 2011;20(2):331-3.
http://dx.doi.org/10.1016/j.yebeh.2010.11.015
https://doi.org/10.1016/j.yebeh.2010.11....
. They also have other psychogenic disorders in
approximately 70% of cases2121 . Brown RJ, Syed TU, Benbadis S, LaFrance WC Jr, Reuber M.
Psychogenic nonepileptic seizures. Epilepsy Behav. 2011;22(1):85-93.
http://dx.doi.org/10.1016/j.yebeh.2011.02.016
https://doi.org/10.1016/j.yebeh.2011.02....
,2323 . Proença IC, Castro LH, Jorge CL, Marchetti RL. Emotional
trauma and abuse in patients with psychogenic nonepileptic seizures. Epilepsy
Behav. 2011;20(2):331-3.
http://dx.doi.org/10.1016/j.yebeh.2010.11.015
https://doi.org/10.1016/j.yebeh.2010.11....
.
DIAGNOSIS DURING VIDEO EEG
Video EEG (VEEG) is the “gold standard” for proper diagnosis of
PNES2121 . Brown RJ, Syed TU, Benbadis S, LaFrance WC Jr, Reuber M.
Psychogenic nonepileptic seizures. Epilepsy Behav. 2011;22(1):85-93.
http://dx.doi.org/10.1016/j.yebeh.2011.02.016
https://doi.org/10.1016/j.yebeh.2011.02....
,2424 . LaFrance Jr WC, Devinsky O. The treatment of psychogenic
nonepileptic seizures: historical perspectives and future directions. Epilepsia.
2004;45(Suppl. s2):15-21.
http://dx.doi.org/10.1111/j.0013-9580.2004.452002.x
https://doi.org/10.1111/j.0013-9580.2004...
. During VEEG monitoring, behavior
and electroencephalographic activity are simultaneously registered. A spontaneous or
elicited event is defined as a PNES when there is no ictal EEG evidence of epileptic
discharges before, during or after the ictus, and semiology is consistent with PNES
and not epilepsy. The event must be critically investigated in the context of
clinical data because simple partial epileptic seizures, parietal lobe and
hypermotor complex partial frontal lobe epileptic seizures may occur without evident
epileptic discharges2121 . Brown RJ, Syed TU, Benbadis S, LaFrance WC Jr, Reuber M.
Psychogenic nonepileptic seizures. Epilepsy Behav. 2011;22(1):85-93.
http://dx.doi.org/10.1016/j.yebeh.2011.02.016
https://doi.org/10.1016/j.yebeh.2011.02....
,2424 . LaFrance Jr WC, Devinsky O. The treatment of psychogenic
nonepileptic seizures: historical perspectives and future directions. Epilepsia.
2004;45(Suppl. s2):15-21.
http://dx.doi.org/10.1111/j.0013-9580.2004.452002.x
https://doi.org/10.1111/j.0013-9580.2004...
. The International League Against Epilepsy (ILAE) NES Task
Force published minimal requirements for NES diagnosis, which utilizes history,
semiology and EEG (preferably video) to establish diagnosis2525 . LaFrance Jr WC, Baker GA, Duncan R, Goldstein LH, Reuber M.
Minimum requirements for the diagnosis of psychogenic nonepileptic seizures: a
staged approach: A report from the International League Against Epilepsy
Nonepileptic Seizures Task Force. Epilepsia. 2013;54(11):2005-18.
http://dx.doi.org/10.1111/epi.12356
https://doi.org/10.1111/epi.12356...
. As is done with PMD diagnosis, levels of
diagnostic certainty are ranked based on what data are available from history,
witnessed event and diagnostic testing, with levels of Possible, Probable,
Clinically Established, and Documented diagnosis (see Table 2).
Provocative procedures, such as saline provocation, hypnosis, simple suggestions,
suggestive interview or a mixture of them have been used to obtain a typical event,
however, the ethics of provocative procedures has been raised2424 . LaFrance Jr WC, Devinsky O. The treatment of psychogenic
nonepileptic seizures: historical perspectives and future directions. Epilepsia.
2004;45(Suppl. s2):15-21.
http://dx.doi.org/10.1111/j.0013-9580.2004.452002.x
https://doi.org/10.1111/j.0013-9580.2004...
,2525 . LaFrance Jr WC, Baker GA, Duncan R, Goldstein LH, Reuber M.
Minimum requirements for the diagnosis of psychogenic nonepileptic seizures: a
staged approach: A report from the International League Against Epilepsy
Nonepileptic Seizures Task Force. Epilepsia. 2013;54(11):2005-18.
http://dx.doi.org/10.1111/epi.12356
https://doi.org/10.1111/epi.12356...
. The average sensitivity of saline provocation across
studies with VEEG monitoring is approximately 74%2424 . LaFrance Jr WC, Devinsky O. The treatment of psychogenic
nonepileptic seizures: historical perspectives and future directions. Epilepsia.
2004;45(Suppl. s2):15-21.
http://dx.doi.org/10.1111/j.0013-9580.2004.452002.x
https://doi.org/10.1111/j.0013-9580.2004...
. Routine activation procedures (hyperventilation and
photic stimulation) can be used during EEG for seizure induction and do not pose the
risk of compromising the physician-patient alliance. Approximately 10% of patients
with PNES have epilepsy, when studied using the most stringent criteria2626 . Marchetti RL, Kurcgant D, Gallucci Neto J, Von Bismark MA, Fiore
LA. Epilepsy in patients with psychogenic non-epileptic seizures. Arq
Neuropsiquiatr. 2010;68:168-73.
http://dx.doi.org/10.1590/s0004-282x2010000200003
https://doi.org/10.1590/s0004-282x201000...
. Other reports show a range from
5.3 to 73% of patients with PNES with mixed epilepsy55 . Rowan AJ. An introduction to current practice in the diagnosis of
non-epileptic seizures. In: Gates JR, Rowan AJ. Non-epileptic seizures. Boston:
Butteworth-Heinemann; 1993. p. 1-8.,1616 . Gates JR. Epidemiology and classification of non-epileptic events.
In: Gates JR, Rowan AJ, editors. Non-epileptic seizures. 2th ed. Boston:
Butterworth-Heinemann; 2000. p. 3-14.,1717 . LaFrance Jr, WC, Bjornaes, H. Designing treatment plans based on
etiology of psychogenic nonepileptic seizures. In: Schachter, SC, LaFrance Jr,
WC, editors. Gates & Rowan’s Nonepileptic seizures. 3 rd ed. New
York: Cambridge University Press; 2010. p. 266-80.,1818 . Marchetti RL, Kurcgant D, Gallucci Neto J, Bismark MA, Marchetti
LB, Fiore LA. Psychiatric diagnoses of patients with psychogenic non-epileptic
seizures. Seizure. 2008;17(3):247-53.
http://dx.doi.org/10.1016/j.seizure.2007.07.006
https://doi.org/10.1016/j.seizure.2007.0...
,1919 . Benbadis SR, Hauser AW. An estimate of the prevalence of
psychogenic non-epileptic seizures. Seizures 2000;9(4):280-1.
http://dx.doi.org/10.1053/seiz.2000.0409
https://doi.org/10.1053/seiz.2000.0409...
,2525 . LaFrance Jr WC, Baker GA, Duncan R, Goldstein LH, Reuber M.
Minimum requirements for the diagnosis of psychogenic nonepileptic seizures: a
staged approach: A report from the International League Against Epilepsy
Nonepileptic Seizures Task Force. Epilepsia. 2013;54(11):2005-18.
http://dx.doi.org/10.1111/epi.12356
https://doi.org/10.1111/epi.12356...
,2626 . Marchetti RL, Kurcgant D, Gallucci Neto J, Von Bismark MA, Fiore
LA. Epilepsy in patients with psychogenic non-epileptic seizures. Arq
Neuropsiquiatr. 2010;68:168-73.
http://dx.doi.org/10.1590/s0004-282x2010000200003
https://doi.org/10.1590/s0004-282x201000...
. This variability in different studies may reflect
several methodological aspects. A study carried in a Brazilian tertiary center found
this association occurring in 50% of PNES patients2626 . Marchetti RL, Kurcgant D, Gallucci Neto J, Von Bismark MA, Fiore
LA. Epilepsy in patients with psychogenic non-epileptic seizures. Arq
Neuropsiquiatr. 2010;68:168-73.
http://dx.doi.org/10.1590/s0004-282x2010000200003
https://doi.org/10.1590/s0004-282x201000...
, a higher association level than found in studies in
the US. Comorbid psychiatric disorders are the rule in patients with PNES, however,
brief psychiatric consults do not always find clear abnormalities during the mental
status examination that are found with more comprehensive anamnesis. Comorbidity
with depressive, anxiety and borderline and obsessive compulsive personality
disorders is high1616 . Gates JR. Epidemiology and classification of non-epileptic events.
In: Gates JR, Rowan AJ, editors. Non-epileptic seizures. 2th ed. Boston:
Butterworth-Heinemann; 2000. p. 3-14.,2323 . Proença IC, Castro LH, Jorge CL, Marchetti RL. Emotional
trauma and abuse in patients with psychogenic nonepileptic seizures. Epilepsy
Behav. 2011;20(2):331-3.
http://dx.doi.org/10.1016/j.yebeh.2010.11.015
https://doi.org/10.1016/j.yebeh.2010.11....
,2727 . Martin R, Burneo JG, Prasad A, Powell T, Faught E, Knowlton R et
al. Frequency of epilepsy in patients with psychogenic seizures monitored by
video-EEG. Neurology. 2003;61(12):1791-2.
http://dx.doi.org/10.1212/01.wnl.0000098890.13946.f5
https://doi.org/10.1212/01.wnl.000009889...
. Patients who receive no feedback or intervention after
VEEG have no improvement or worsening of PNES2828 . Fiszman A, Kanner AM. Comorbidities in psychogenic nonepileptic
seizures: Depressive, anxiety, and personality disorders. In: Schacter S,
LaFrance Jr WC, editors. Gates and Rowan’s Nonepileptic seizures. 3rd ed.
Cambridge: Cambridge University Press; 2010. p. 225-34.. In contrast, a correct diagnosis or a therapeutic
communication can reduce or even abolish PNES in a significant amount of patients,
at least temporally2828 . Fiszman A, Kanner AM. Comorbidities in psychogenic nonepileptic
seizures: Depressive, anxiety, and personality disorders. In: Schacter S,
LaFrance Jr WC, editors. Gates and Rowan’s Nonepileptic seizures. 3rd ed.
Cambridge: Cambridge University Press; 2010. p. 225-34., although if
no treatment is proposed after communication, seizures may return, or even the
appearance of new medically unexplained symptoms2020 . Reuber M, Pukrop R, Bauer J, Helmstaedter C, Tessendorf N, Elger
CE. Outcome in psychogenic nonepileptic seizures: 1 to 10-year follow-up in 164
patients. Ann Neurol. 2003;53(3):305-11.
http://dx.doi.org/10.1002/ana.3000
https://doi.org/10.1002/ana.3000...
,2828 . Fiszman A, Kanner AM. Comorbidities in psychogenic nonepileptic
seizures: Depressive, anxiety, and personality disorders. In: Schacter S,
LaFrance Jr WC, editors. Gates and Rowan’s Nonepileptic seizures. 3rd ed.
Cambridge: Cambridge University Press; 2010. p. 225-34..
PROGNOSIS OF PNES
Adults with PNES having risk factors that include comorbid depression, a personality
disorder and a history of abuse were more likely to have persistence of seizures,
compared to those without these comorbidities2828 . Fiszman A, Kanner AM. Comorbidities in psychogenic nonepileptic
seizures: Depressive, anxiety, and personality disorders. In: Schacter S,
LaFrance Jr WC, editors. Gates and Rowan’s Nonepileptic seizures. 3rd ed.
Cambridge: Cambridge University Press; 2010. p. 225-34.. Several studies show that early and correct diagnosis
of PNES, followed by adequate treatment, could lead either to remission in 19 to
25%, or to improvement in 75 to 95%. Therefore, correct diagnosis and treatment
might result in a significant reduction of utilization and cost of health
programs2020 . Reuber M, Pukrop R, Bauer J, Helmstaedter C, Tessendorf N, Elger
CE. Outcome in psychogenic nonepileptic seizures: 1 to 10-year follow-up in 164
patients. Ann Neurol. 2003;53(3):305-11.
http://dx.doi.org/10.1002/ana.3000
https://doi.org/10.1002/ana.3000...
,2323 . Proença IC, Castro LH, Jorge CL, Marchetti RL. Emotional
trauma and abuse in patients with psychogenic nonepileptic seizures. Epilepsy
Behav. 2011;20(2):331-3.
http://dx.doi.org/10.1016/j.yebeh.2010.11.015
https://doi.org/10.1016/j.yebeh.2010.11....
,2424 . LaFrance Jr WC, Devinsky O. The treatment of psychogenic
nonepileptic seizures: historical perspectives and future directions. Epilepsia.
2004;45(Suppl. s2):15-21.
http://dx.doi.org/10.1111/j.0013-9580.2004.452002.x
https://doi.org/10.1111/j.0013-9580.2004...
,2828 . Fiszman A, Kanner AM. Comorbidities in psychogenic nonepileptic
seizures: Depressive, anxiety, and personality disorders. In: Schacter S,
LaFrance Jr WC, editors. Gates and Rowan’s Nonepileptic seizures. 3rd ed.
Cambridge: Cambridge University Press; 2010. p. 225-34..
PSYCHOGENIC MOVEMENT DISORDERS
Identifying psychogenic movement disorders can be a challenging task in neurological
practice. Historically, Charcot was known for his great interest in psychogenic
cases and for paying special attention in its definition and treatment22 . Goetz CG, Bonduelle M, Gelfand T. Hysteria. In: Goetz CG,
Bonduelle M, Gelfand T. Charcot. Constructing neurology. New York: Oxford
University Press; 1995. p. 172-216.. Charcot considered that besides
women, men employed in labor, effeminate men and children were prone to the
development of hysterical neurological deficits, including PNES (then referred to as
hysteroepilepsy) and PMD22 . Goetz CG, Bonduelle M, Gelfand T. Hysteria. In: Goetz CG,
Bonduelle M, Gelfand T. Charcot. Constructing neurology. New York: Oxford
University Press; 1995. p. 172-216.,2929 . Goetz CG. Charcot and psychogenic movement disorders. In: Hallet
M, Fahn S, Jankovic J, Lang AE, Cloninger CR, Yudofsky SC. Psychogenic movement
disorders: neurology and neuropsichiatry. Philadelphia: Lippincot Williams &
Wilkins; 2006. p. 3-13.,3030 . Munhoz RP, Zavala JA, Becker N, Teive HAG. Cross-cultural
influences on psychogenic movement disorders: a comparative review with a
Brazilian series of 83 cases. Clinical Neurol Neurosurg. 2011;113(2):115-8.
http://dx.doi.org/10.1016/j.clineuro.2010.10.004
https://doi.org/10.1016/j.clineuro.2010....
. More recent epidemiological data show that PMD represents
2% to 3% of all cases seen in movement disorders clinics and occur more commonly in
women (ratio of men to women about 1:5) between 37 and 50 years of age3030 . Munhoz RP, Zavala JA, Becker N, Teive HAG. Cross-cultural
influences on psychogenic movement disorders: a comparative review with a
Brazilian series of 83 cases. Clinical Neurol Neurosurg. 2011;113(2):115-8.
http://dx.doi.org/10.1016/j.clineuro.2010.10.004
https://doi.org/10.1016/j.clineuro.2010....
,3131 . Factor SA, Podskalny GD, Molho ES. Psychogenic movement disorders
frequency, clinical profile and characteristics. J Neurol Neurosurg Psychiatry.
1995;59(4):406-12. http://dx.doi.org/10.1136/jnnp.59.4.406
https://doi.org/10.1136/jnnp.59.4.406...
,3232 . Galvéz-Jimenez N, Lang AE. Psychogenic movement disorders.
In: Watts RL, Koller WC. Movement disorders: neurologic principles and practice.
New York: McGraw-Hill; 1997. p. 715-32.,3333 . Lang AE. General overview of psychogenic movement disorders:
epidemiology, diagnosis, and prognosis. In: Hallet M, Fahn S, Jankovic J, Lang
AE, Cloninger CR, Yudofsky SC. Psychogenic movement disorders: neurology and
neuropsichiatry. Philadelphia: Lippincot Williams & Wilkins; 2006. p.
35-41.. Like PNES, comorbidities are common in PMD. Ten to
15% of cases with PMD have additional neurological disorders, and 80% have
psychiatric comorbidities66 . Hinson VK, Haren WB. Psychogenic movement disorders. Lancet
Neurol. 2006;5(8):695-700.
http://dx.doi.org/10.1016/S1474-4422(06)70523-3
https://doi.org/10.1016/S1474-4422(06)70...
,77 . Edwards MJ, Bhatia KP. Functional (psychogenic movement disorders:
merging mind and brain. Lancet Neurol. 2012;11(3):250-60.
http://dx.doi.org/10.1016/S1474-4422(11)70310-6
https://doi.org/10.1016/S1474-4422(11)70...
,3131 . Factor SA, Podskalny GD, Molho ES. Psychogenic movement disorders
frequency, clinical profile and characteristics. J Neurol Neurosurg Psychiatry.
1995;59(4):406-12. http://dx.doi.org/10.1136/jnnp.59.4.406
https://doi.org/10.1136/jnnp.59.4.406...
,3232 . Galvéz-Jimenez N, Lang AE. Psychogenic movement disorders.
In: Watts RL, Koller WC. Movement disorders: neurologic principles and practice.
New York: McGraw-Hill; 1997. p. 715-32.,3333 . Lang AE. General overview of psychogenic movement disorders:
epidemiology, diagnosis, and prognosis. In: Hallet M, Fahn S, Jankovic J, Lang
AE, Cloninger CR, Yudofsky SC. Psychogenic movement disorders: neurology and
neuropsichiatry. Philadelphia: Lippincot Williams & Wilkins; 2006. p.
35-41.,3434 . Williams DT, Ford B, Fahn S. Phenomenology and psychopathology
related to psychogenic movement disorders. Adv Neurol. 1995;65:231-57.
http://dx.doi.org/10.1002/9781444346183.ch24
https://doi.org/10.1002/9781444346183.ch...
,3535 . Gupta A, Lang AE. Psychogenic movement disorders. Curr Opin
Neurol. 2009;22(4):430-6.
http://dx.doi.org/10.1097/WCO.0b013e32832dc169
https://doi.org/10.1097/WCO.0b013e32832d...
,3635 . Gupta A, Lang AE. Psychogenic movement disorders. Curr Opin
Neurol. 2009;22(4):430-6.
http://dx.doi.org/10.1097/WCO.0b013e32832dc169
https://doi.org/10.1097/WCO.0b013e32832d...
. Other risk factors for PMD include a history of sexual
abuse, surgery, trauma and major emotional stress3131 . Factor SA, Podskalny GD, Molho ES. Psychogenic movement disorders
frequency, clinical profile and characteristics. J Neurol Neurosurg Psychiatry.
1995;59(4):406-12. http://dx.doi.org/10.1136/jnnp.59.4.406
https://doi.org/10.1136/jnnp.59.4.406...
,3333 . Lang AE. General overview of psychogenic movement disorders:
epidemiology, diagnosis, and prognosis. In: Hallet M, Fahn S, Jankovic J, Lang
AE, Cloninger CR, Yudofsky SC. Psychogenic movement disorders: neurology and
neuropsichiatry. Philadelphia: Lippincot Williams & Wilkins; 2006. p.
35-41.,3434 . Williams DT, Ford B, Fahn S. Phenomenology and psychopathology
related to psychogenic movement disorders. Adv Neurol. 1995;65:231-57.
http://dx.doi.org/10.1002/9781444346183.ch24
https://doi.org/10.1002/9781444346183.ch...
. Somatoform disorders are very common in PMD,
including somatization disorder, conversion disorder, and hypochondriasis, followed
by depression, anxiety and personality disorders2929 . Goetz CG. Charcot and psychogenic movement disorders. In: Hallet
M, Fahn S, Jankovic J, Lang AE, Cloninger CR, Yudofsky SC. Psychogenic movement
disorders: neurology and neuropsichiatry. Philadelphia: Lippincot Williams &
Wilkins; 2006. p. 3-13.,3131 . Factor SA, Podskalny GD, Molho ES. Psychogenic movement disorders
frequency, clinical profile and characteristics. J Neurol Neurosurg Psychiatry.
1995;59(4):406-12. http://dx.doi.org/10.1136/jnnp.59.4.406
https://doi.org/10.1136/jnnp.59.4.406...
,3333 . Lang AE. General overview of psychogenic movement disorders:
epidemiology, diagnosis, and prognosis. In: Hallet M, Fahn S, Jankovic J, Lang
AE, Cloninger CR, Yudofsky SC. Psychogenic movement disorders: neurology and
neuropsichiatry. Philadelphia: Lippincot Williams & Wilkins; 2006. p.
35-41.,3434 . Williams DT, Ford B, Fahn S. Phenomenology and psychopathology
related to psychogenic movement disorders. Adv Neurol. 1995;65:231-57.
http://dx.doi.org/10.1002/9781444346183.ch24
https://doi.org/10.1002/9781444346183.ch...
.
Diagnosis OF PMD
The semiology and characteristics of PMD are important for the diagnosis. The onset
of PMD is usually abrupt and occurs in the context of a precipitating event.
Disabilities may be selective, and signs may improve with distraction3131 . Factor SA, Podskalny GD, Molho ES. Psychogenic movement disorders
frequency, clinical profile and characteristics. J Neurol Neurosurg Psychiatry.
1995;59(4):406-12. http://dx.doi.org/10.1136/jnnp.59.4.406
https://doi.org/10.1136/jnnp.59.4.406...
,3333 . Lang AE. General overview of psychogenic movement disorders:
epidemiology, diagnosis, and prognosis. In: Hallet M, Fahn S, Jankovic J, Lang
AE, Cloninger CR, Yudofsky SC. Psychogenic movement disorders: neurology and
neuropsichiatry. Philadelphia: Lippincot Williams & Wilkins; 2006. p.
35-41.. In general, PMD are not consistent over time and
are not congruent with the classical definitions of neurological movement disorders.
The association of changes in abnormal movements with distractibility is very common
and is an important observation used during examination. Fatigability, particularly
in patients presenting with tremor, is also common3333 . Lang AE. General overview of psychogenic movement disorders:
epidemiology, diagnosis, and prognosis. In: Hallet M, Fahn S, Jankovic J, Lang
AE, Cloninger CR, Yudofsky SC. Psychogenic movement disorders: neurology and
neuropsichiatry. Philadelphia: Lippincot Williams & Wilkins; 2006. p.
35-41.,3535 . Gupta A, Lang AE. Psychogenic movement disorders. Curr Opin
Neurol. 2009;22(4):430-6.
http://dx.doi.org/10.1097/WCO.0b013e32832dc169
https://doi.org/10.1097/WCO.0b013e32832d...
. In some cases, abnormal movements stop when fatigue
appears. PMD can present with a wide variety of manifestations, including tremor,
dystonia, chorea, gait disorders, parkinsonism, tics and ataxia, usually in
combination and affecting multiple body parts. The diagnosis of PMD has been
classically viewed as one of exclusion3232 . Galvéz-Jimenez N, Lang AE. Psychogenic movement disorders.
In: Watts RL, Koller WC. Movement disorders: neurologic principles and practice.
New York: McGraw-Hill; 1997. p. 715-32.,3333 . Lang AE. General overview of psychogenic movement disorders:
epidemiology, diagnosis, and prognosis. In: Hallet M, Fahn S, Jankovic J, Lang
AE, Cloninger CR, Yudofsky SC. Psychogenic movement disorders: neurology and
neuropsichiatry. Philadelphia: Lippincot Williams & Wilkins; 2006. p.
35-41.,3434 . Williams DT, Ford B, Fahn S. Phenomenology and psychopathology
related to psychogenic movement disorders. Adv Neurol. 1995;65:231-57.
http://dx.doi.org/10.1002/9781444346183.ch24
https://doi.org/10.1002/9781444346183.ch...
,4545 . Grimald I, Dubuc M, Kahane P, Bougerol T, Vercueil L. Anxiety and
depression in psychogenic movement disorder and non-epileptic seizures: a
prospective comparative study. Revue Neurol (Paris). 2010;166(5):515-22.
http://dx.doi.org/10.1016/j.neurol.2009.10.016
https://doi.org/10.1016/j.neurol.2009.10...
. However, recent clinical advances and advances in
neurophysiological examinations have led to the establishment of diagnostic criteria
that help clinicians make a more accurate diagnosis and manage the disease
better3131 . Factor SA, Podskalny GD, Molho ES. Psychogenic movement disorders
frequency, clinical profile and characteristics. J Neurol Neurosurg Psychiatry.
1995;59(4):406-12. http://dx.doi.org/10.1136/jnnp.59.4.406
https://doi.org/10.1136/jnnp.59.4.406...
. There are some
clinical characteristics of PMD that are more common in clinical neurological
practice. Table 3 summarizes the signs and
characteristics associated with PMD. PMD are traditionally classified into the
following four categories according to basic definitions proposed originally by Fahn
and Williams in 19883636 . Fahn S, Williams D. Psychogenic dystonia. Adv Neurol.
1988;50:431-55. and
subsequently modified by Fahn in 19943737 . Bhatia KP, Schneider SA. Psychogenic tremor and related disorders.
J Neurol. 2007;254(5):569-74.
http://dx.doi.org/10.1007/s00415-006-0348-z
https://doi.org/10.1007/s00415-006-0348-...
. The same diagnostic level category names are used for
PNES, however, the criteria for the diagnostic designation differ significantly for
PMD:
-
Documented PMD: Documented PMD includes those patients who have complete resolution of PMD following psychotherapy, psychological suggestion by the physician, physiotherapy or administration of a placebo with suggestion or are witnessed as being free of symptoms when left alone and supposedly unobserved;
-
Clinically established PMD: Clinically established PMD is inconsistent over time or incongruent with the typical presentation of a classical movement disorder. In the presence of either of the above, the patient must have any of the additional manifestations, including other neurological signs, multiple somatizations, obvious psychiatric disturbance, disappearance of the PMD with distraction and excessive (almost deliberate) slowing;
-
Probable PMD: Probable PMD includes patients with incongruous and inconsistent movements in the absence of any of the other features listed in category 2 (Clinically established) to support the diagnosis of PMD and patients with a movement disorder that is consistent and congruent with a classical neurological movement disorder but who have other features, such as disappearance of the movement with distraction or other psychogenic neurologic disorders and multiple somatizations;
-
Possible PMD: Possible PMD is characterized by clinical features of PMD occurring in the presence of an emotional disturbance3636 . Fahn S, Williams D. Psychogenic dystonia. Adv Neurol. 1988;50:431-55.,3737 . Bhatia KP, Schneider SA. Psychogenic tremor and related disorders. J Neurol. 2007;254(5):569-74. http://dx.doi.org/10.1007/s00415-006-0348-z
https://doi.org/10.1007/s00415-006-0348-... .
Most of the information used to delineate the currently available diagnostic criteria for PMD come from case series. The three most significant of these series are those described by Lang3333 . Lang AE. General overview of psychogenic movement disorders: epidemiology, diagnosis, and prognosis. In: Hallet M, Fahn S, Jankovic J, Lang AE, Cloninger CR, Yudofsky SC. Psychogenic movement disorders: neurology and neuropsichiatry. Philadelphia: Lippincot Williams & Wilkins; 2006. p. 35-41., including the case series of PMD evaluated by Fahn et al., Jankovic et al., and Lang et al. in which tremor, dystonia, myoclonus and mixed (often bizarre) movement disorders were the most common PMD.
Additionally, in a Brazilian series published in 2010 (Table 4), tremor was the most frequent PMD (55.6%)3030 . Munhoz RP, Zavala JA, Becker N, Teive HAG. Cross-cultural
influences on psychogenic movement disorders: a comparative review with a
Brazilian series of 83 cases. Clinical Neurol Neurosurg. 2011;113(2):115-8.
http://dx.doi.org/10.1016/j.clineuro.2010.10.004
https://doi.org/10.1016/j.clineuro.2010....
. The second most common PMD
semiology was pure dystonia (33.3%), agreeing with data in the medical literature in
which the frequency of this presentation ranges from 15% to 53%3030 . Munhoz RP, Zavala JA, Becker N, Teive HAG. Cross-cultural
influences on psychogenic movement disorders: a comparative review with a
Brazilian series of 83 cases. Clinical Neurol Neurosurg. 2011;113(2):115-8.
http://dx.doi.org/10.1016/j.clineuro.2010.10.004
https://doi.org/10.1016/j.clineuro.2010....
,3131 . Factor SA, Podskalny GD, Molho ES. Psychogenic movement disorders
frequency, clinical profile and characteristics. J Neurol Neurosurg Psychiatry.
1995;59(4):406-12. http://dx.doi.org/10.1136/jnnp.59.4.406
https://doi.org/10.1136/jnnp.59.4.406...
. Parkinsonism and myoclonus PMD were uncommon, each
occurring in four patients3030 . Munhoz RP, Zavala JA, Becker N, Teive HAG. Cross-cultural
influences on psychogenic movement disorders: a comparative review with a
Brazilian series of 83 cases. Clinical Neurol Neurosurg. 2011;113(2):115-8.
http://dx.doi.org/10.1016/j.clineuro.2010.10.004
https://doi.org/10.1016/j.clineuro.2010....
.
Factor et al. studied 28 patients with PMD and found that the most common semiology
was tremor (50%), followed by dystonia, myoclonus and parkinsonism3131 . Factor SA, Podskalny GD, Molho ES. Psychogenic movement disorders
frequency, clinical profile and characteristics. J Neurol Neurosurg Psychiatry.
1995;59(4):406-12. http://dx.doi.org/10.1136/jnnp.59.4.406
https://doi.org/10.1136/jnnp.59.4.406...
. Distractibility (86%) and abrupt
onset (54%) were the most common clinical characteristics in this series of
patients. Twenty-five percent presented with combined PMD and neurological movement
disorder3131 . Factor SA, Podskalny GD, Molho ES. Psychogenic movement disorders
frequency, clinical profile and characteristics. J Neurol Neurosurg Psychiatry.
1995;59(4):406-12. http://dx.doi.org/10.1136/jnnp.59.4.406
https://doi.org/10.1136/jnnp.59.4.406...
.
In general psychogenic or functional tremor (PT) is the most common psychogenic
movement disorder3737 . Bhatia KP, Schneider SA. Psychogenic tremor and related disorders.
J Neurol. 2007;254(5):569-74.
http://dx.doi.org/10.1007/s00415-006-0348-z
https://doi.org/10.1007/s00415-006-0348-...
. It can occur
in any body part, although the hands and arms are the most frequently involved. More
rarely, PT can occur in the head and legs77 . Edwards MJ, Bhatia KP. Functional (psychogenic movement disorders:
merging mind and brain. Lancet Neurol. 2012;11(3):250-60.
http://dx.doi.org/10.1016/S1474-4422(11)70310-6
https://doi.org/10.1016/S1474-4422(11)70...
,3737 . Bhatia KP, Schneider SA. Psychogenic tremor and related disorders.
J Neurol. 2007;254(5):569-74.
http://dx.doi.org/10.1007/s00415-006-0348-z
https://doi.org/10.1007/s00415-006-0348-...
. Of the different clues suggesting tremor may be
psychogenic, the most common are distractibility, entrainment (a change in the
original tremor frequency to match the frequency of a repetitive task performed in
another limb) and the presence of coactivation (the co-contraction sign)3737 . Bhatia KP, Schneider SA. Psychogenic tremor and related disorders.
J Neurol. 2007;254(5):569-74.
http://dx.doi.org/10.1007/s00415-006-0348-z
https://doi.org/10.1007/s00415-006-0348-...
. In general, PT is thought never
to affect the fingers, tongue or face3737 . Bhatia KP, Schneider SA. Psychogenic tremor and related disorders.
J Neurol. 2007;254(5):569-74.
http://dx.doi.org/10.1007/s00415-006-0348-z
https://doi.org/10.1007/s00415-006-0348-...
. The dystonia semiology represents the second most common
form of PMD3838 . Lang AE. Psychogenic dystonia: a review of 18 cases. Can J Neurol
Sci. 1995;22(2):136-43..
In neurological clinical practice the border between functional or psychogenic,
dystonia (FD) and neurological dystonia is not clear-cut77 . Edwards MJ, Bhatia KP. Functional (psychogenic movement disorders:
merging mind and brain. Lancet Neurol. 2012;11(3):250-60.
http://dx.doi.org/10.1016/S1474-4422(11)70310-6
https://doi.org/10.1016/S1474-4422(11)70...
,88 . Lempert T, Dietrich M, Huppert D, Brandt T. Psychogenic disorders
in neurology: frequency and clinical spectrum. Acta Neurol Scand.
1990;82(5):335-40.
http://dx.doi.org/10.1111/j.1600-0404.1990.tb03312.x
https://doi.org/10.1111/j.1600-0404.1990...
,3737 . Bhatia KP, Schneider SA. Psychogenic tremor and related disorders.
J Neurol. 2007;254(5):569-74.
http://dx.doi.org/10.1007/s00415-006-0348-z
https://doi.org/10.1007/s00415-006-0348-...
. Historically, several forms of dystonia have been
considered to be of psychogenic origin, such as blepharospasm and writer’s
cramp77 . Edwards MJ, Bhatia KP. Functional (psychogenic movement disorders:
merging mind and brain. Lancet Neurol. 2012;11(3):250-60.
http://dx.doi.org/10.1016/S1474-4422(11)70310-6
https://doi.org/10.1016/S1474-4422(11)70...
,88 . Lempert T, Dietrich M, Huppert D, Brandt T. Psychogenic disorders
in neurology: frequency and clinical spectrum. Acta Neurol Scand.
1990;82(5):335-40.
http://dx.doi.org/10.1111/j.1600-0404.1990.tb03312.x
https://doi.org/10.1111/j.1600-0404.1990...
,3737 . Bhatia KP, Schneider SA. Psychogenic tremor and related disorders.
J Neurol. 2007;254(5):569-74.
http://dx.doi.org/10.1007/s00415-006-0348-z
https://doi.org/10.1007/s00415-006-0348-...
. In the last 30 years, however, with the great
advances in genetics, neurophysiology and neuroimaging, the phenotypes of primary
idiopathic dystonia have been clearly defined. Some forms of fixed dystonia
associated with previous peripheral trauma and with pain similar to chronic regional
pain syndrome (“causalgia-dystonia”), have been considered FD77 . Edwards MJ, Bhatia KP. Functional (psychogenic movement disorders:
merging mind and brain. Lancet Neurol. 2012;11(3):250-60.
http://dx.doi.org/10.1016/S1474-4422(11)70310-6
https://doi.org/10.1016/S1474-4422(11)70...
,88 . Lempert T, Dietrich M, Huppert D, Brandt T. Psychogenic disorders
in neurology: frequency and clinical spectrum. Acta Neurol Scand.
1990;82(5):335-40.
http://dx.doi.org/10.1111/j.1600-0404.1990.tb03312.x
https://doi.org/10.1111/j.1600-0404.1990...
. The most common forms of FD are blepharospasm,
limb focal dystonia and abductor laryngeal dystonia or paradoxical vocal cord
dysfunction3333 . Lang AE. General overview of psychogenic movement disorders:
epidemiology, diagnosis, and prognosis. In: Hallet M, Fahn S, Jankovic J, Lang
AE, Cloninger CR, Yudofsky SC. Psychogenic movement disorders: neurology and
neuropsichiatry. Philadelphia: Lippincot Williams & Wilkins; 2006. p.
35-41.,3535 . Gupta A, Lang AE. Psychogenic movement disorders. Curr Opin
Neurol. 2009;22(4):430-6.
http://dx.doi.org/10.1097/WCO.0b013e32832dc169
https://doi.org/10.1097/WCO.0b013e32832d...
,3636 . Fahn S, Williams D. Psychogenic dystonia. Adv Neurol.
1988;50:431-55.,3737 . Bhatia KP, Schneider SA. Psychogenic tremor and related disorders.
J Neurol. 2007;254(5):569-74.
http://dx.doi.org/10.1007/s00415-006-0348-z
https://doi.org/10.1007/s00415-006-0348-...
,3838 . Lang AE. Psychogenic dystonia: a review of 18 cases. Can J Neurol
Sci. 1995;22(2):136-43..
The next most common form of PMD is myoclonus, now defined as functional myoclonus
(FM)3939 . Monday K, Jankovic J. Psychogenic myoclonus. Neurology.
1993;43(2):349-52. http://dx.doi.org/10.1212/wnl.43.2.349
https://doi.org/10.1212/wnl.43.2.349...
. In this setting,
electrophysiological tests are needed to ensure correct diagnosis, particularly
those using electroencephalogram-electromyography back-averaging3939 . Monday K, Jankovic J. Psychogenic myoclonus. Neurology.
1993;43(2):349-52. http://dx.doi.org/10.1212/wnl.43.2.349
https://doi.org/10.1212/wnl.43.2.349...
. Functional parkinsonism, chorea,
tics and ataxia are rarely reported4040 . Lang AE, Koller WC, Fahn S. Psychogenic parkinsonism. Arch Neurol.
1995;52(8):802-10.
http://dx.doi.org/10.1001/archneur.1995.00540320078015
https://doi.org/10.1001/archneur.1995.00...
.
Prognosis OF PMD
Patients with PMD usually have a poor prognosis4141 . Lang AE. Long term follow-up of psychogenic movement disorders.
Neurology. 2000;54(Suppl 3):A50-1.,4242 . Thomas M, Vuong KD, Jankovic J. Long-term prognosis with
psychogenic movement disorders. Parkinsonism Related Disord. 2006;12(6):382-7.
http://dx.doi.org/10.1016/j.parkreldis.2006.03.005
https://doi.org/10.1016/j.parkreldis.200...
. Cases with at least six months of symptoms at the
time of diagnosis had little or no response to proposed treatments4141 . Lang AE. Long term follow-up of psychogenic movement disorders.
Neurology. 2000;54(Suppl 3):A50-1.,4242 . Thomas M, Vuong KD, Jankovic J. Long-term prognosis with
psychogenic movement disorders. Parkinsonism Related Disord. 2006;12(6):382-7.
http://dx.doi.org/10.1016/j.parkreldis.2006.03.005
https://doi.org/10.1016/j.parkreldis.200...
. Feinstein et al.4343 . Feinstein A, Stergiopoulos V, Fine J, Lang AE. Psychiatric outcome
in patients with a psychogenic movement disorder: a prospective study.
Neuropsychiatry Neuropsychol Behav Neurol. 2001;14(3):169-76., after evaluating psychiatric
outcomes of their patients with a mean follow up of 3.2 years, showed that PMD
persisted in more than 90% of them, especially in cases with comorbid major
depression, anxiety and personality disorders4343 . Feinstein A, Stergiopoulos V, Fine J, Lang AE. Psychiatric outcome
in patients with a psychogenic movement disorder: a prospective study.
Neuropsychiatry Neuropsychol Behav Neurol. 2001;14(3):169-76.. In contrast, Thomas et al., evaluated 228
patients with PMD with a mean follow-up of 3.4 years (6 months to 12 years), and
concluded that symptoms improved in 56.6% of patients, worsened in 22.1% and
remained the same in 21.3%4242 . Thomas M, Vuong KD, Jankovic J. Long-term prognosis with
psychogenic movement disorders. Parkinsonism Related Disord. 2006;12(6):382-7.
http://dx.doi.org/10.1016/j.parkreldis.2006.03.005
https://doi.org/10.1016/j.parkreldis.200...
.
Ertan et al. studied 49 patients with PMD in a tertiary clinic in Turkey and
concluded that the response to treatment was poor, with a high rate of drop out
of these patients in the follow-up4444 . Ertan S, Uluduz D, Ozekmekçi S, Kiziltan G, Ertan T,
Yalçinkaya C et al. Clinical characteristics of 49 patients with
psychogenic movement disorders in a tertiary clinic in Turkey. Mov Disord.
2009;24(5):759-82. http://dx.doi.org/10.1002/mds.22114
https://doi.org/10.1002/mds.22114...
.
PSYCHOGENIC MOVEMENT DISORDERS AND NON-EPILEPTIC SEIZURES
An intriguing question is whether PMD and PNES share the same psychopathological
comorbidities, particularly anxiety, depression and other conditions, such as
personality disorders and trauma/abuse histories4545 . Grimald I, Dubuc M, Kahane P, Bougerol T, Vercueil L. Anxiety and
depression in psychogenic movement disorder and non-epileptic seizures: a
prospective comparative study. Revue Neurol (Paris). 2010;166(5):515-22.
http://dx.doi.org/10.1016/j.neurol.2009.10.016
https://doi.org/10.1016/j.neurol.2009.10...
,4646 . Driver-Dunckley E, Stonnington CM, Locke DEC, Noe K. Comparison of
psychogenic movement disorders and psychogenic nonepileptic seizures: Is
phenotype clinically important? Psychosomatics. 2011;52(4):337-45.
http://dx.doi.org/10.1016/j.psym.2011.01.008
https://doi.org/10.1016/j.psym.2011.01.0...
,4747 . Hopp JL, Anderson KE, Krumholz A, Gruber-Baldini AL, Shulman LM.
Psychogenic seizures and psychogenic movement disorders: are they the same
patients? Epilepsy Behav. 2012;25(4):666-9.
http://dx.doi.org/10.1016/j.yebeh.2012.10.007
https://doi.org/10.1016/j.yebeh.2012.10....
,4848 . Mula M. Are psychogenic non-epileptic seizures and psychogenic
movement disorders two different entities? When even neurologists stop talking
to each other. Epilepsy Behav. 2013;26(1):100-1.
http://dx.doi.org/10.1016/j.yebeh.2012.07.024
https://doi.org/10.1016/j.yebeh.2012.07....
. Against this background, Grimaldi et al. performed a
prospective comparative study in which they investigated the presence of anxiety and
depression in 17 patients, nine of whom had PMD and eight PNES4545 . Grimald I, Dubuc M, Kahane P, Bougerol T, Vercueil L. Anxiety and
depression in psychogenic movement disorder and non-epileptic seizures: a
prospective comparative study. Revue Neurol (Paris). 2010;166(5):515-22.
http://dx.doi.org/10.1016/j.neurol.2009.10.016
https://doi.org/10.1016/j.neurol.2009.10...
. They concluded that all the patients had the
same demographic and psychopathological profile, although in the group with PNES
there was a greater incidence of anxiety disorders and a family history of
epilepsy4545 . Grimald I, Dubuc M, Kahane P, Bougerol T, Vercueil L. Anxiety and
depression in psychogenic movement disorder and non-epileptic seizures: a
prospective comparative study. Revue Neurol (Paris). 2010;166(5):515-22.
http://dx.doi.org/10.1016/j.neurol.2009.10.016
https://doi.org/10.1016/j.neurol.2009.10...
. Driver-Dunckley et
al. studied 172 patients in a retrospective chart review, comparing 116 patients
with PNES with 56 with PMD4646 . Driver-Dunckley E, Stonnington CM, Locke DEC, Noe K. Comparison of
psychogenic movement disorders and psychogenic nonepileptic seizures: Is
phenotype clinically important? Psychosomatics. 2011;52(4):337-45.
http://dx.doi.org/10.1016/j.psym.2011.01.008
https://doi.org/10.1016/j.psym.2011.01.0...
. They
found that 82% of the patients were female and that 70% had chronic pain, 55%
subjective cognitive complaints, 47% fatigue and 45% a history of childhood abuse.
The patients with PNES had coexisting epilepsy in 17% of cases, and those with PMD
had coexisting neurologic movement disorders in 9%. The authors concluded that PNES
and PMD had the same psychopathology, with more similarities than differences4646 . Driver-Dunckley E, Stonnington CM, Locke DEC, Noe K. Comparison of
psychogenic movement disorders and psychogenic nonepileptic seizures: Is
phenotype clinically important? Psychosomatics. 2011;52(4):337-45.
http://dx.doi.org/10.1016/j.psym.2011.01.008
https://doi.org/10.1016/j.psym.2011.01.0...
. Hopp et al., studied 104
patients with PMD and 35 with PNES using different cognitive, psychological and
social function measures4747 . Hopp JL, Anderson KE, Krumholz A, Gruber-Baldini AL, Shulman LM.
Psychogenic seizures and psychogenic movement disorders: are they the same
patients? Epilepsy Behav. 2012;25(4):666-9.
http://dx.doi.org/10.1016/j.yebeh.2012.10.007
https://doi.org/10.1016/j.yebeh.2012.10....
. They
demonstrated that patients with PNES and PMD, despite differences in their
phenomenology and demographics, shared the same psychiatric symptoms, suggesting
that PNES and PMD represent different presentations of a single disorder4747 . Hopp JL, Anderson KE, Krumholz A, Gruber-Baldini AL, Shulman LM.
Psychogenic seizures and psychogenic movement disorders: are they the same
patients? Epilepsy Behav. 2012;25(4):666-9.
http://dx.doi.org/10.1016/j.yebeh.2012.10.007
https://doi.org/10.1016/j.yebeh.2012.10....
. Mula published an editorial
about PNES and PMD discussing the commonalities between these two conditions4848 . Mula M. Are psychogenic non-epileptic seizures and psychogenic
movement disorders two different entities? When even neurologists stop talking
to each other. Epilepsy Behav. 2013;26(1):100-1.
http://dx.doi.org/10.1016/j.yebeh.2012.07.024
https://doi.org/10.1016/j.yebeh.2012.07....
. He suggested that these
disorders occupy a gray area between neurology and psychiatry and commented on the
poor level of integration between neurologists and psychiatrists4848 . Mula M. Are psychogenic non-epileptic seizures and psychogenic
movement disorders two different entities? When even neurologists stop talking
to each other. Epilepsy Behav. 2013;26(1):100-1.
http://dx.doi.org/10.1016/j.yebeh.2012.07.024
https://doi.org/10.1016/j.yebeh.2012.07....
.
CONCLUDING REMARKS
Moving the field forward for better understanding of PMD, PNES and other somatoform disorders will require increased collaboration between neurology and psychiatry. A renaissance of neuropsychiatry is being driven by advances in functional neuroimaging, neuroscience and the treatment needs of the aging population with neurodegenerative neuropsychiatric disorders. Viewing somatoform/conversion disorders from a combined lens of neurology/psychiatry provides a comprehensive assessment approach and opens avenues of collaborative management.
“Lumpers or Splitters”
From prior research the question arises, should PMD and PNES be “lumped or split”, that is, are they “variations on a theme”, or independent populations? Larger samples for fully powered designed studies may require incorporating both PMD and PNES. On the other hand, discrete samples may be needed to identify regions of interest and putative networks in biomarker focused studies. Also of note is that a number of patients with PNES also have other movement symptoms without change in level of consciousness, apart from their ictus, possibly generating a third group of “mixed PNES/PMD”. These issues can be addressed by linking research centers to reach target sample sizes.
Treatment of PNES and PMD
Great strides have been made in validating treatment for patients with PNES in
the past decade. Prior to the NINDS/NIMH/AES supported NES Workshop in 2005,
only class III and IV level treatment data existed2424 . LaFrance Jr WC, Devinsky O. The treatment of psychogenic
nonepileptic seizures: historical perspectives and future directions. Epilepsia.
2004;45(Suppl. s2):15-21.
http://dx.doi.org/10.1111/j.0013-9580.2004.452002.x
https://doi.org/10.1111/j.0013-9580.2004...
,4949 . LaFrance WC Jr, Alper K, Babcock D, Barry JJ, Benbadis S, Caplan R
et al. Nonepileptic seizures treatment workshop summary. Epilepsy Behav.
2006;8(3):451-61. http://dx.doi.org/10.1016/j.yebeh.2006.02.004
https://doi.org/10.1016/j.yebeh.2006.02....
. The NES workshop set the NES research benchmarks,
and PNES treatment is a target of the NINDS Epilepsy Research Benchmarks4949 . LaFrance WC Jr, Alper K, Babcock D, Barry JJ, Benbadis S, Caplan R
et al. Nonepileptic seizures treatment workshop summary. Epilepsy Behav.
2006;8(3):451-61. http://dx.doi.org/10.1016/j.yebeh.2006.02.004
https://doi.org/10.1016/j.yebeh.2006.02....
. Our group and others have
been systematically developing treatments for patients with PNES and studying
biomarkers of PNES. This is being accomplished through multi-disciplinary,
multi-modal, multi-site work. Examples of advances in PNES treatment over the
last decade include examining clinical trial methodology in PNES in an open
label study of sertraline for PNES4949 . LaFrance WC Jr, Alper K, Babcock D, Barry JJ, Benbadis S, Caplan R
et al. Nonepileptic seizures treatment workshop summary. Epilepsy Behav.
2006;8(3):451-61. http://dx.doi.org/10.1016/j.yebeh.2006.02.004
https://doi.org/10.1016/j.yebeh.2006.02....
, followed by a pilot placebo-controlled RCT with
sertraline4949 . LaFrance WC Jr, Alper K, Babcock D, Barry JJ, Benbadis S, Caplan R
et al. Nonepileptic seizures treatment workshop summary. Epilepsy Behav.
2006;8(3):451-61. http://dx.doi.org/10.1016/j.yebeh.2006.02.004
https://doi.org/10.1016/j.yebeh.2006.02....
. Standard
medical care (SMC) or treatment as usual (TAU) is in the US,and in a
cross-cultural comparison with Chile has been described5050 . LaFrance WC Jr, Marinis AJ, Webb AF, Machan JT, Rusch MD, Kanner
AM. Comparing standard medical care for nonepileptic seizures in Chile and the
United States. Epilepsy Behav. 2012;25(2):224-29.
http://dx.doi.org/10.1016/j.yebeh.2012.07.028
https://doi.org/10.1016/j.yebeh.2012.07....
. A psychotherapy initially used in epilepsy
has been modified for PNES and was used to conduct an open label trial for
PNES5151 . LaFrance WC Jr, Miller IW, Ryan CE, Blum AS, Solomon DA, Kelley JE
et al. Cognitive behavioral therapy for psychogenic nonepileptic seizures.
Epilepsy Behav. 2009;14(4):591-6.
http://dx.doi.org/10.1016/j.yebeh.2009.02.016
https://doi.org/10.1016/j.yebeh.2009.02....
. With
pharmacologic, psychotherapeutic and SMC data, a multi-centered, RCT comparing
PNES-CBT, sertraline, PNES-CBT and sertraline, and SMC/TAU was conducted5252 . LaFrance WC Jr, Baird GB, Barry JJ, Blum AS, Webb FA, Keitner GL
et al. Multicenter pilot preatment trial for psychogenic nonepileptic seizures.
JAMA Psychiatry. 2014;71(9):997-1005.
http://dx.doi.org/10.1001/jamapsychiatry.2014.817
https://doi.org/10.1001/jamapsychiatry.2...
. Based on the successful
reduction in seizures, improvement in comorbidities, QoL and functioning in the
two groups treated with the manualized therapy, the treatment workbook5353 . Reiter J, Andrews D, Reiter C, LaFrance WC Jr. Taking control of
your seizures: a Workbook. New York: Oxford University Press;
2014. is being published along with
a therapist’s guide5454 . LaFrance WC, Wincze J. Taking control of your seizures: therapist
guide. New York: Oxford University Press; 2014..
Providers at sites across the US are being trained in administering the
manualized treatment. These and other modalities studied in PNES treatments are
reviewed in the ILAE NES management article5555 . LaFrance WC, Reuber M, Goldstein LH. Management of psychogenic
nonepileptic seizures. Epilepsia. 2013;54 Suppl 1:S53-67.
http://dx.doi.org/10.1111/epi.12106
https://doi.org/10.1111/epi.12106...
. Advances in PMD treatment include an open label
trial of sertraline for PMD5656 . Hinson VK, Weinstein S, Bernard B, Leurgans SE, Goetz CG.
Single-blind clinical trial of psychotherapy for treatment of psychogenic
movement disorders. Parkinsonism Relat Disord. 2006;1(3)2:177-80.
http://dx.doi.org/10.1016/j.parkreldis.2005.10.006
https://doi.org/10.1016/j.parkreldis.200...
.
In another study, psychodynamic psychotherapy was used in a single-blind
treatment trial for patients with PMD5656 . Hinson VK, Weinstein S, Bernard B, Leurgans SE, Goetz CG.
Single-blind clinical trial of psychotherapy for treatment of psychogenic
movement disorders. Parkinsonism Relat Disord. 2006;1(3)2:177-80.
http://dx.doi.org/10.1016/j.parkreldis.2005.10.006
https://doi.org/10.1016/j.parkreldis.200...
. Non-psychotherapy modalities including
physiotherapy also have been used to treat patients with PMD5757 . Czarnecki K, Thompson JM, Seime R, Geda YE, Duffy JR, Ahlskog JE.
Functional movement disorders: successful treatment with a physical therapy
rehabilitation protocol. Parkinsonism Relat Disord. 2012;18:247-51.
http://dx.doi.org/10.1016/j.parkreldis.2011.10.011
https://doi.org/10.1016/j.parkreldis.201...
. A summary of cognitive
behavioral approaches used in patients with funtional neurololgical disorders
provides a review of studies in a variety of somatoform presentations5858 . Hopp JL, LaFrance WC. Cognitive behavioral therapy for psychogenic
neurological disorders. Neurologist. 2012;18(6):364-72.
http://dx.doi.org/10.1097/NRL.0b013e31826e8ff5
https://doi.org/10.1097/NRL.0b013e31826e...
.
With the momentum of recognition by the Movement Disorders Society (MDS) and the
International League Against Epilepsy (ILAE)designating these
populations’ symptoms as significant disorders found in patients with
movement disorders and seizures, task forces are setting standards for diagnosis
and treatment. Examples include the ILAE NES Task Force establishing the minimum
requirements standards for making the diagnosis of PNES2525 . LaFrance Jr WC, Baker GA, Duncan R, Goldstein LH, Reuber M.
Minimum requirements for the diagnosis of psychogenic nonepileptic seizures: a
staged approach: A report from the International League Against Epilepsy
Nonepileptic Seizures Task Force. Epilepsia. 2013;54(11):2005-18.
http://dx.doi.org/10.1111/epi.12356
https://doi.org/10.1111/epi.12356...
, and summarized the literature on PNES
management5555 . LaFrance WC, Reuber M, Goldstein LH. Management of psychogenic
nonepileptic seizures. Epilepsia. 2013;54 Suppl 1:S53-67.
http://dx.doi.org/10.1111/epi.12106
https://doi.org/10.1111/epi.12106...
. Text books
are published summarizing the NES5959 . Schachter SC, LaFrance WC Jr, editors. Gates and Rowan’s
Nonepileptic seizures. 3rd ed. Cambridge: Cambridge University Press;
2010., and PMD literature6060 . Hallett M, Lang AE, Jankovic J, et al. Psychogenic movement
disorders and other conversion disorders. 2nd ed. Cambridge: Cambridge
University Press; 2011.. National research funding sources (Institutes,
Medical Societies and voluntaries) now are providing support for studies
examining treatment and mechanisms for patients with conversion disorders.With
greater identification of the somatoform disorders by neurologists,
psychiatrists, primary care physicians, opportunities for treatment are opened.
With a neuropsychiatric conceptualization and formulation and with more
management options now available, this population is being demystified and
providers are empowered to effectively treat patients. Cross-disciplinary and
cross-cultural collaboration will continue to facilitate advances in this common
and challenging neuropsychiatric disorder.
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» https://doi.org/10.1016/j.parkreldis.2005.10.006 -
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» https://doi.org/10.1016/j.parkreldis.2011.10.011 -
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59Schachter SC, LaFrance WC Jr, editors. Gates and Rowan’s Nonepileptic seizures. 3rd ed. Cambridge: Cambridge University Press; 2010.
-
60Hallett M, Lang AE, Jankovic J, et al. Psychogenic movement disorders and other conversion disorders. 2nd ed. Cambridge: Cambridge University Press; 2011.
Publication Dates
-
Publication in this collection
Oct 2014
History
-
Received
01 June 2014 -
Accepted
25 June 2014