Journal of the Neurological Sciences 435 (2022) 120204

Contents lists available at ScienceDirect

Journal of the Neurological Sciences

journal homepage: www.elsevier.com/locate/jns

Review Article

Problems and controversies in tremor classification

Elan D. Louis a, *, Peter G. Bain b
a
Department of Neurology, University of Texas Southwestern Medical Center, Dallas, TX, USA
b
Department of Neurosciences, Division of Brain Sciences, Charing Cross Hospital Campus, Imperial College London, UK

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

Keywords: The Consensus Statement on the Classification of Tremors, from the Task Force of the International Parkinson’s
Tremor classification and Movement Disorder Society was published in 2018 in response to advances in the understanding of essential
Internal tremor tremor (ET), tremor associated with dystonia and other monosymptomatic and indeterminate tremors. This was a
Essential tremor
revision of the previous 1998 Consensus Statement. A number of problems arise from this classification and, in
Essential tremor plus
this manuscript, we highlight several of these. These problems relate to the definition of tremor, the definition of
Orthostatic tremor
ET, the proposal of the term ‘essential tremor plus’, issues related to orthostatic tremor (OT) including the
proposal of the term ‘orthostatic tremor plus’, and the absence of a hierarchy of terms used to classify tremor.
Among the proposals we make is that the definition of tremor should acknowledge that tremor is a symptom, ET
should be redefined, severity is an important characteristic for classifying and diagnosing tremor, the terms
‘essential tremor plus’ and ‘orthostatic tremor plus’ should be abandoned, and classification of tremor requires
rules for creating a hierarchy of terminology.
"This article is part of the Special Issue "Tremor" edited by Daniel D. Truong, Mark Hallett, and Aasef
Shaikh".

1. Introduction presumed anatomical origin, such as cerebellar tremor pertaining to

Humans may exhibit a broad array of tremors under a range of
different circumstances. Indeed, tremor disorders are among the most
common neurological disorders. As with most disorders, there is a need
for classification and, given this broad variety, this can pose challenges.
These challenges afford opportunities to reconsider these disorders as
well as advance our understanding of them. Several classification
schema have been published over the years as our understanding has
advanced. In 2018, the Consensus Statement on the Classification of
Tremors, from the Task Force of the International Parkinson’s and
Movement Disorder Society was published. A number of scholars,
including both of the authors of the current manuscript, engaged in the
discussions and participated in the writing of the manuscript. This
classification was deemed necessary following advances in the under­
standing of essential tremor (ET), tremor associated with dystonia and
other monosymptomatic and indeterminate tremors; this was a revision
of the previous 1998 Consensus Statement [1,2]. Furthermore, the
previous consensus statement (1998) was criticized for not using a
consistent approach to tremor classification. In some instances in the
1998 Consensus Statement, a tremor was defined according to its
intention tremor. In other instances, tremor was defined according to a
presumed underlying pathophysiology, for example, parkinsonian
tremor and tremors associated with neuropathies. Classification of other
tremors was based purely on clinical phenomenology, as in primary
writing tremor, orthostatic tremor, and isolated voice tremor. None of
these approaches was considered adequate, notably when a particular
type of tremor had multiple causes [1,2].
In the 2018 Consensus Statement (CS 2018), the proposed classifi­
cation had two main axes: clinical features (Axis 1) and etiology (Axis 2)
(see Fig. 1 in CS 2018). This approach to disease classification is com­
mon in epidemiological studies in which clinical features of a disease are
used to define a clinical syndrome, and studies of this syndrome ulti­
mately lead to the discovery of one or more etiologies. It was empha­
sized in the CS 2018 that a syndrome may have multiple etiologies, and a
particular etiology may produce multiple clinical syndromes [3,4]. This
two-axis approach is similar to the new classification scheme for dys­
tonia and was designed to facilitate the collection of clinically important
information from tremor patients and to serve as a tool for clinical
diagnosis and research [5]. Although the newest classification is of
value, on further inspection, a number of problems arise as well. In this

* Corresponding author.
E-mail address: elan.louis@utsouthwestern.edu (E.D. Louis).

https://doi.org/10.1016/j.jns.2022.120204
Received 5 August 2021; Received in revised form 24 September 2021; Accepted 17 February 2022
Available online 19 February 2022
0022-510X/© 2022 Elsevier B.V. All rights reserved.
E.D. Louis and P.G. Bain Journal of the Neurological Sciences 435 (2022) 120204

manuscript, we highlight several of these. 2.3. The definition of ET

2. Problems and controversies arising from the Consensus
Statement on the Classification of Tremors 2018
2.1. The definition of tremor
The definition of tremor itself can be quite challenging and this
serves as a good starting point for our discussion. In the Consensus
Statement on the Classification of Tremors 2018 (CS-2018) tremor was
defined as follows: ‘tremor is an involuntary, rhythmic, oscillatory move­
ment of a body part’. This definition needs modification, as we explain
below.
First, the CS 2018 definition should specify that tremor can be a
symptom and/or a sign. Tremor can also be a symptom only, for example
in one survey, the sensation of ‘internal tremor’ was reported by 32.6%
of 89 consecutive patients with Parkinson’s disease, 36% of 70 with
multiple sclerosis and 55% of 6 patients with ET [6]. Tremor is often also
a symptom even when there is an associated sign (tremor). The re­
lationships between the signs and the symptoms of tremor are complex.
It is often the symptom of tremor (i.e., the patient’s experience of
shaking) that drives the clinician’s treatment response rather than the
degree of objective tremor on examination.
A second modification is as follows. Further clarity is required on the
issue of whether tremor must be involuntary, for example does a
research control subject deliberately producing a rhythmic oscillatory
movement of the hand for a functional imaging study exhibit tremor?
Even if this is deemed ‘feigned-tremor’ or ‘mimicked-tremor’ the terms
contain the word tremor, so that if these are not tremors what are they?
Similarly some patients deliberately produce or feign tremor for medico-
legal reasons or in order to obtain social security benefits.
Third, how does one classify an involuntary movement that is
oscillatory but not always strictly rhythmic? In dystonic tremor, for
example tremulous cervical dystonia, there may be tonic contractions
with sustained EMG activity breaking up the oscillatory behaviour, so
that the overall movement is not rhythmic.
2.2. Isolated tremor syndromes
“Isolated tremor syndrome” is a phrase that has been used to denote a
syndrome in which tremor is the only symptom or sign. In the CS 2018
the use of the phrase ‘isolated tremor syndrome’ is redundant, for
example, in the definition of ET given below:
Essential tremor:
1) Isolated tremor syndrome of bilateral upper limb action tremor
2) at least 3 years’ duration
3) with or without tremor in other locations (e.g., head, voice, or lower limbs)
4) absence of other neurological signs, such as dystonia, ataxia, or parkinsonism.
ET is the most prevalent tremor disorder and, as a result, its defini­
tion is particularly important. Increasing understanding of heterogene­
ity in ET patient populations led to considerable discussions in the
drafting of the CS 2018 definition. That definition was of value; how­
ever, we propose the following modifications to enhance the definition:
Essential tremor (revised):
1) An upper limb action tremor that has a predominantly kinetic component and is
usually bilateral but may start in one arm and can occasionally remain unilateral.
2) Tremor may be apparent in other parts of the body.
3) A rest tremor component may be present but:
a. is usually less severe than the action tremor components.
b. should not be anatomically separated from the action tremor components.
4) Should not be diagnosed in the presence of another diagnosis that is likely to be
responsible for the tremor.
In our revised definition of ET no arbitrary duration of tremor is
specified and there is an emphasis on the kinetic component of the ac­
tion tremor because studies indicate that the kinetic component is pre­
dominant to the postural tremor [7,8].
2.4. Problems in the consensus statement 2018 reflected in Fig. 3
It is always a challenge to organize the full spectrum of heterogeneity
into a single, neat scheme.
Fig. 3 of the CS 2018 the tremor syndromes are organized under six
headings; and the scheme creates several contentious issues. It is
worthwhile reviewing these and we do so below.
First, in column 5: Intention tremor is included under ‘tremor with
prominent additional signs’ but is not included in column 1 under: ‘ac­
tion and rest tremor’. However, intention tremor can occur in isolation
or together with other forms of action tremor, for example in some pa­
tients with ET. It can also be seen with additional neurological signs for
example in patients with multiple sclerosis.
Second, isolated rest tremor is included in column 1 but should also
be included in column 2 ‘focal tremor’ as it usually starts as focal tremor
of the fingers.
Third, orthostatic tremor plus was omitted from Fig. 3. This is pre­
sumably because it would be included in column 5 under ‘tremor with
prominent other signs’ which would then disassociate orthostatic tremor
from orthostatic tremor in column 4. The merits or otherwise of dis­
tinguishing orthostatic tremor plus from orthostatic tremor is discussed
further in section 2.9 below.
2.5. Tremor severity is important in the classification of a tremor’s
characteristics

Tremors are generally classified based on their speed (i.e., fre­

The word isolated in ‘isolated tremor syndrome’ is unnecessary as the
quency), severity (i.e., amplitude) as well as a number of other char­
observation of a bilateral upper limb action tremor (point 1 in the
acteristics including bodily location and activation conditions. Thus,
definition above) in the absence of other neurological signs (point 4
severity is an important feature to consider. Additionally, the relative
above) indicates that it is just a type of tremor. The use of the word
severities of different types of tremors within a different disorder are of
‘isolated’ is also ambiguous as other types of tremor (e.g., head, voice, or
value in arriving at the correct diagnosis. In Fig. 1a of the CS 2018
lower limbs) are permitted in ET. So the phrase ‘Isolated head tremor’
tremor severity is not included under ‘tremors characteristics’ nor
could mean that there are no other neurological signs (i.e. only tremor is
included at all in the Consensus Statement, which would appear to be an
present) or that there are no other tremors (i.e. there is just a head
important oversight because:
tremor and no other types of tremor present), which is confusing.
To avoid future ambiguities and to aid in precision, we propose that:
1. It is part of the characterization of tremor.
The concept of ‘Isolated tremor syndromes’ be replaced by ‘types of
2. It has a major influence on patient’s’ function and thus therapy.
tremor’ that are classified simply by their characteristics (e.g. anatom­
3. It is helpful for the diagnostic process, for example:
ical distribution, activation states and severity).
i. Normal physiological tremor is not severe.
ii. The comparative severity of the rest, postural and intention tremor
components of a patient’s tremor is useful diagnostically. For

2
E.D. Louis and P.G. Bain Journal of the Neurological Sciences 435 (2022) 120204

example, compare patients A and B below (The tremor components 2.5 above), as the rest tremor component is usually less severe than
are scored on the Bain & Findley 1993 rating scale from 0 to 10) [9]: the action tremor components. Furthermore, the distribution of the
rest tremor: for example the onset of a rest tremor in an ET patient’s
right hand and right foot would strongly indicate the development of
Right Left Parkinsonism.
Patient A:
2. The term ‘essential tremor plus’ is unhelpful because it creates
Rest 1/10 1/10 another problem, which is how to interpret the classifier ‘of uncer­
Post 6/10 6/10 tain significance’? What does this mean? Does it include minor
Int 5/10 5/10 neurological signs such as carpal tunnel syndrome, absent vibration
sense on the toes, a diminished tendon reflex or minimal cognitive
Patient B: impairment or tandem gait abnormality? ET will co-exist with a
Rest 6/10 5/10 plethora of other medical and neurological conditions for example
Post 5/10 3/10
Int 1/10 1/10
diabetes mellitus and cerebrovascular vascular disease and is most
prevalent in persons of advanced age. The application of the term
essential tremor plus also means that the majority of ET patients,
In this example Patient A has a marked intention tremor component particularly the elderly would have to be relabelled Essential tremor
with minimal rest tremor whereas patient B has prominent rest tremor plus.
with a minimal intention tremor component. Patient B is much more 3. If a neurological sign is of uncertain significance, then it is not a sign
likely to have parkinsonism than patient A because the tremor severity but just an observed phenomenon, until its sensitivity and specificity
gradient is RT > PT> > IT whereas patient A has a severity gradient of have been defined and ideally its receiver operating characteristics
RT < <PT>IT indicating a predominant action tremor, the commonest documented. Thus, such phenomena (‘soft signs’) should not be used
cause of which is ET. to alter a clinical diagnosis from essential tremor to essential tremor
plus.

2.6. The site of onset of tremor in ET

2.8. Primary orthostatic tremor

ET is not a static condition and the phenotype at onset is not often the
Aside from ET, the Consensus statement had to contend with the
phenotype that is seen many years later. The site of onset of ET is an
definition of the full panoply of tremor disorders. In the Consensus
important consideration. It was recorded in the CS 2018 that a discus­
Statement on the Classification of Tremor (2018), Primary orthostatic
sion occurred about whether to include the onset of tremor in the upper
tremor is defined as follows:
limbs as a further criterion for the definition of ET. However, it was
Primary orthostatic tremor: is a generalized high frequency (13-18 Hz) isolated tremor
concluded that there were no convincing data to support this criterion. syndrome that occurs when standing. Confirmation of the tremor frequency is needed,
It is accepted that ET typically begins in the hands. However, we typically with an electromyography (EMG).
propose that should a patient develop a head tremor initially and sub­
sequently develop an action tremor of the hands that would fulfil the
criteria for ET, it would be helpful to label these patients as ‘essential Let us consider a number of issues. First, it is notable that this defi­
tremor with onset as head tremor’, as careful follow up of these patients nition does not specify that primary orthostatic tremor (POT) should
would determine whether their subsequent clinical course is that of ET affect a leg or legs. This appears to be an oversight that requires
patients with the typical onset in their hands or whether other features, correction. Second it is also now accepted that patients with POT may
for example dystonia develop. In this respect patients with fleeting mild have various types of tremor affecting other parts of the body as shown
head tremor with no abnormal hand tremor have been detected in in Tables 1 and 2:
families with ET [10]. These patients would be diagnosed as having
‘head tremor’ but it is possible that they may go on to develop an action 2.9. Orthostatic tremor plus
tremor of the hands so that they would then fulfil criteria for ET. We
highlight this issue as an important area for further study. ‘Orthostatic tremor plus’ is the term recommended by the Interna­
tional Parkinson’s and Movement Disorder Society Task force on tremor
to describe 13-18 Hz orthostatic tremor found in combination with other
2.7. Essential tremor plus
neurological conditions.2 These include various forms of Parkinsonism
(Parkinson’s disease, cerebrovascular parkinsonism, primary gait igni­
In the Consensus Statement on the Classification of Tremor (2018), a
tion failure, progressive supranuclear palsy), cerebellar degeneration,
new term was proposed to deal with the clinical heterogeneity that is
dementia with Lewy bodies, and restless legs syndrome [13–16].
seen in patients with ET. In general, new terms should be given special
consideration to ensure that they are optimal. ‘Essential tremor plus’
was defined as: ‘Tremor with the characteristics of ET and additional Table 1
Parts of the body reported to be affected by tremor by patients with primary
neurological signs of uncertain significance such as impaired tandem
orthostatic tremor [11].
gait, questionable dystonic posturing, memory impairment, or other
mild neurologic signs of unknown significance that do not suffice to Type of Tremor Number (/56) Percentage (%)
make an additional syndrome classification or diagnosis. ET with tremor RLL tremor 55 98.2*
at rest should be classified here’. LLL Tremor 55 98.2*
There are three main issues about Essential tremor plus that we RUL Tremor 28 50.0
LUL Tremor 25 44.6
would like to consider: Head tremor 11 19.6
Trunk tremor 9 16
1. Including the presence of tremor at rest within ‘ET plus’ is ques­ Voice Tremor 4 7.1
tionable because in patients with ET rest tremor components may Tongue Tremor 4 7.1
occur in the hands and are not infrequent, particularly in those with Key: RLL: right lower limb, LLL: left lower limb, RUL: right upper limb, LLL: left
long standing tremor. However, the relative severities of the rest and lower limb *Note that two patients were aware of orthostatic tremor in only one
action components of the tremor need careful assessment (see point leg (i.e. symptomatically unilateral POT but it was present in both legs on EMG).

3
E.D. Louis and P.G. Bain Journal of the Neurological Sciences 435 (2022) 120204

Table 2 currently, there appear to be none in practice. For example:

Prevalence of the rest, postural (arms held outstretched) and intention tremor of
the upper limbs detected on examination in patients with primary orthostatic 1. Parkinsonian tremor or Tremulous Parkinsonism
tremor [12]. 2. Parkinsonism with primary orthostatic tremor or primary orthostatic
Study Number in Rest tremor Postural Intention tremor plus
study in arms N tremor in tremor in arms 3. Dystonic tremor or tremulous dystonia
(%) arms N (%) N (%)

Hassan et al. 184 5 (2.7%) 42 (22.8%) NS For example, if a patient has a severe bilateral action tremor of the
(2016) arms and trivial asymptomatic dystonia of the hands, is it appropriate
Ganos et al. 59* NS 30 (50.8%) NS
(2016)
that the weight of the terminology should reflect the dystonia, so that
Bain & Jones 56 2 (3.6%) 50 (89.3%) 33 (58.9%) the patient is then classified as having ‘dystonic tremor’? The definition of
(2010) dystonic tremor in the MDS Consensus Statement on Tremor (2018) is
Yaltho & Ondo 40* NS 30 (75.0%) NS rather loose: ‘Tremor in a body part affected by dystonia is labelled as
(2014)
dystonic tremor’. Thus, as some of the patients have tremor of the hands
Mestre et al. 24 NS NS NS
(2012) and if dystonia of the hands is deemed to be present this would then
Gerschlager et al. 31* NS 24 (77.4%) NS constitute ‘dystonic tremor’, which would seem particularly unbalanced
(2004) and misleading if the tremor is the major issue (severe and symptomatic)
Piboolnurak et al. 26 NS NS NS and the dystonic signs are trivial (minimal and asymptomatic). There is
(2005)
the additional issue of whether or not the phrase ‘tremor in a body part
McManis & 30 NS 9 (30%) NS
Sharborough affected by dystonia’ is a sufficiently accurate definition of ‘dystonic
(1993) tremor’, which is widely used to describe the more refined situation
Range in above 2.7%–3.6% 22.8% - 33.0% where tremor and dystonia are occurring in the same muscle group.
studies 89.3%

KEY: *Including POT (± upper limb tremor) only. NS: not specified. 3. Conclusions

This use of the ‘plus’ in orthostatic tremor plus in the Consensus
Statement on the Classification of Tremors (2018) differs from that used
for essential tremor plus:
In essential tremor plus it indicates ‘additional neurological signs of
uncertain significance’ whilst in orthostatic tremor plus it indicates ‘other
neurological conditions’ (i.e. established neurological diagnoses). This
inconsistency of terminology is incongruous and thus confusing.
It also leaves the presence of orthostatic tremor resulting from
medical (rather than neurological) conditions, for example Graves dis­
ease, or reversible orthostatic tremor secondary to dopamine blocking
agents, outside the definition of orthostatic tremor plus [17–20].
In this respect it is noteworthy that Mestre and colleagues. (2012)
found no difference in any of the demographic variables, the clinical
features, neurophysiology and patients’ responses to treatment between
patients with orthostatic tremor and orthostatic tremor plus, which raises
the question as to whether this distinction has any value [13].
The use of the term orthostatic tremor plus also raises another
complexity, which is: if a patient has another neurological condition, for
example Parkinson’s should the diagnosis be:
a. Parkinson’s with orthostatic tremor, or
b. Orthostatic tremor plus
Should the terminology used reflect the relative importance of the
two conditions? If so, Parkinson’s with orthostatic tremor would be more
appropriate than orthostatic tremor plus. The latter also obfuscates a
plethora of other neurological conditions as orthostatic tremor plus does
not reveal what the ‘plus’ indicates. For example, 13-18 Hz orthostatic
tremor has been reported in patients with spinocerebellar ataxia type 2,
stiff person syndrome, mitochondrial C10orf2 TWINKLE mutation, a
female carrier of a REEP 1 mutation (spastic paraparesis SPG31) focal
dystonia, oro-facial dyskinesia, (minor) head injury, a pontine tuber­
culoma and a midbrain lesion [13,21–25].
Given the high prevalence as well as broad variety of tremor disor­
ders, attempts at classification are likely to meet special challenges.
These challenges serve as useful opportunities to re-evaluate our un­
derstanding of these conditions. Discussion of these issues can thus be
particularly fruitful. While the 2018 consensus statement on the classi­
fication of tremor provided value, a number of problems arise as dis­
cussed elsewhere and here [26], and it requires modification for the
reasons stated above. Many of the problems are caused by the use of
syndromes to classify tremor rather than viewing tremors as signs and
symptoms that can occur alone or in the context of a plethora of medical
and neurological diseases. The attempt to put tremors into syndromes
creates unnecessary dichotomies when instead the phenomena can more
accurately be simply described.
In our view, rules governing the hierarchy of terms used in tremor
classification need to be created to improve the current situation. For
example, in the terms Parkinsonian tremor or tremulous Parkinsonism the
order of the two conditions are transposed, or in Parkinsonism and
orthostatic tremor and orthostatic tremor plus completely different termi­
nology can be deployed to describe the same situation.
Finally, the MDS Consensus Statement on the Classification of
Tremor (2018) classifies tremor by their clinical features (Axis 1) and
their etiology (Axis 2). For example, in Table 2 in the paper (Etiological
causes of tremor), PD (Parkinson’s disease) is given as an etiology.
However, some patients with PD have tremor but others do not, indi­
cating differences in the pathophysiology between PD patients with and
without tremor. Thus future classifications need to embrace the patho­
physiological mechanisms causing tremor within the same etiology, in
this case Parkinson’s disease where tremor may or may not be present.
Relevant conflicts of interest/financial disclosures
Nothing to declare.
Funding agencies

2.10. Absence of a hierarchy of terms used to classify tremor No funding was involved in this work.

The language one uses to classify medical conditions is of impor­ Ethical approval and consent to participate
tance. Part of this involves the ordering of words in multi-word terms.
Stated in another way, what rules decide the hierarchy of terms used to Ethical approval was not required as no patient participation or
describe tremulous conditions? There were none in the CS 2018 and novel data was involved in this work.

4
E.D. Louis and P.G. Bain
Availability of supporting data
All relevant supporting data are referenced.
Author’s contributions
Elan D Louis and Peter G Bain created the paper. We have
acknowledged Madelein van der Stouwe for her helpful comments,
which were incorporated into the manuscript.
Declaration of Competing Interest
PGB None to declare. EDL None to declare.
Acknowledgement
We would like to thank Dr. Madelein van der Stouwe for her con­
tributions to this article.
References
[1] G. Deuschl, P.G. Bain, M. Brin, an Ad Hoc Scientific Committee, Consensus
statement of the movement disorder society on tremor, in: International Congress:
Tremor: Basic Mechanisms and Clinical Aspects, Kiel, Germany 1997. Movement
Disorders 13, 1998, pp. 2–23, suppl 3.
[2] K.P. Bhatia, P.G. Bain, N. Bajaj, R.J. Elble, M. Hallett, E.D. Louis, J. Raethjen,
M. Stamelou, C.M. Testa, Deuschl G and the tremor task force of the International
Parkinson and Movement Disorder Society, Mov. Disord. 33 (1) (2018) 75–87.
[3] V.J. Schoenbach, W.D. Rosamond, The Phonomenon of Disease. Understanding the
Fundamentals of Epidemiology—an Evolving Text. http://www.epidemiolog.
net/evolving/PhenomenonofDisease.pdf.
[4] B. MacMahon, T.F. Pugh, Causes and entities of disease, in: D.W. Clark,
B. MacMahon (Eds.), Preventive Medicine, Little, Brown, Boston, MA, 1967,
pp. 26–33.
[5] A. Albanese, K. Bhatia, S.B. Bressman, et al., Phenomenology and classification of
dystonia: a consensus update, Mov. Disord. 28 (2013) 863–873.
[6] G.D. Cochrane, S. Rizvi, A. Abrantes, B. Crabtree, J. Cahill, J.H. Friedman, Internal
tremor in Parkinson’s disease, multiple sclerosis and essential tremor,
Parkinsonism Relat. Disord. 21 (10) (2015) 1145–1147, https://doi.org/10.1016/j.
parkreldis.2015.07.014.
[7] E.D. Louis, The primary type of tremor in essential tremor is kinetic rather than
postural: cross-sectional observation of tremor phenomenology in 369 cases, Eur. J.
Neurol. 20 (4) (2013) 725–727, https://doi.org/10.1111/j.1468-
1331.2012.03855.x. Apr. (Epub 2012 Aug 27.PMID: 22925197).
[8] K.C. Brennan, E.C. Jurewicz, B. Ford, S.L. Pullman, E.D. Louis, Is essential tremor
predominantly a kinetic or a postural tremor? A clinical and electrophysiological
study, Mov. Disord. 17 (2) (2002) 313–316, https://doi.org/10.1002/mds.10003.
Journal of the Neurological Sciences 435 (2022) 120204
[9] P.G. Bain, L.J. Findley, Assessing Tremor Severity: A Clinical Handbook, Smith-
Gordon, London, 1993 (ISBN: 1 85463 099 7).
[10] E.D. Louis, J.H. Meyers, A.D. Cristal, A. Patel, S. Tinaz, S.L. Pullman, L.N. Clark,
R. Ottman, P. Factor-Litvak, Transient, isolated head tremor in “unaffected”
individuals: is essential tremor an even more prevalent disease than we suppose?
Front. Neurol. 9 (2018 Jul 13) 570, https://doi.org/10.3389/fneur.2018.00570
(eCollection 2018).
[11] L. Jones, P.G. Bain, Characterising orthostatic tremor: a study of 56 patients,
J. Neurol. Neurosurg. Psychiatry 81 (11) (2010) E22.
[12] P.G. Bain, Orthostatic tremor, in: D. Haubenberger, C. Testa (Eds.), Tremor, Oxford
University Press, 2021 in press.
[13] T.A. Mestre, A.E. Lang, J.J. Ferreira, V. Almeida, M. de Carvalho, J. Miyasaki, et
al., Associated movement disorders in orthostatic tremor, J. Neurol. Neurosurg.
Psychiatry 83 (2012) 725–729, https://doi.org/10.1136/jnnp-2012-302436.
[14] M.U. Manto, F. Setta, B. Legros, J. Jacquy, E. Godaux, Resetting of orthostatic
tremor associated with cerebellar cortical atrophy by transcranial magnetic
stimulation, Arch. Neurol. 56 (1999) 1497–1500, https://doi.org/10.1001/
archneur.56.12.1497.
[15] F. Setta, J. Jacquy, J. Hildebrand, M.U. Manto, Orthostatic tremor associated with
cerebellar ataxia, J. Neurol. 245 (1998) 299–302, https://doi.org/10.1007/
s004150050222.
[16] H. Sarva, W.L. Severt, N. Jacoby, S.L. Pullman, R. Saunders-Pullman, Secondary
orthostatic tremor in the setting of cerebellar degeneration, J. Clin. Neurosci. 27
(2016) 173–178, https://doi.org/10.1016/j.jocn.2015.10.027.
[17] E.K. Tan, Y.L. Lo, L.L. Chan, Graves disease and isolated orthostatic tremor,
Neurology 70 (2008) 1497–1498, https://doi.org/10.1212/01.wnl.0000310405.
36026.92.
[18] O. Stich, C. Fritzsch, B. Heimbach, M. Rijntjes, Orthostatic tremor associated with
biclonal IgG and IgA lambda gammopathy of undetermined significance, Mov.
Disord. 24 (2009) 154–155, https://doi.org/10.1002/mds.22200.
[19] J. Benito-Leo’n, J. Porta-Etessam, Shaky-leg syndrome and vitamin B12 deficiency,
N. Engl. J. Med. 342 (2000) 981, https://doi.org/10.1056/
NEJM200003303421318.
[20] H. Alonso-Navarro, M. Orti-Pareja, F.J. Jiménez-Jiménez, J.M. Zurdo-Hernández,
M. de Toledo, I. Puertas-Muñoz, Orthostatic tremor induced by pharmaceuticals,
Rev. Neurol. 39 (2004) 834–836.
[21] J. Benito-Leon, J. Rodriguez, M. Orti-Pareja, L. Ayuso-Peralta, F.J. Jimenez-
Jimenez, J.A. Molina, Symptomatic orthostatic tremor in pontine lesions,
Neurology 49 (1997) 1439–1441, https://doi.org/10.1212/WNL.49.5.1439.
[22] M. Milone, B.T. Klassen, M.L. Landsverk, R.H. Haas, L.J. Wong, Orthostatic tremor,
progressive external ophthalmoplegia, and Twinkle, JAMA Neurol. 70 (2013)
1429–1431, https://doi.org/10.1001/jamaneurol.2013.3521.
[23] S.S. Sanitate, J.R. Meerschaert, Orthostatic tremor: delayed onset following head
trauma, Arch. Phys. Med. Rehabil. 74 (1993) 886–889, https://doi.org/10.1016/
0003-9993(93)90017-5.
[24] R. Vetrugno, R. D’Angelo, M. Alessandria, M. Mascalchi, P. Montagna, Orthostatic
tremor in a left midbrain lesion, Mov. Disord. 25 (2010) 793–795, https://doi.org/
10.1002/mds.23018.
[25] R. Erro, C. Cordivari, K.P. Bhatia, SPG31 presenting with orthostatic tremor, Eur. J.
Neurol. 21 (2014) e34–e35, https://doi.org/10.1111/ene.12360.
[26] E.D. Louis, M. Bares, J. Benito-Leon, S. Fahn, S.J. Frucht, J. Jankovic, W.G. Ondo,
P.K. Pal, E.K. Tan, Essential tremor-plus: a controversial new concept, Lancet
Neurol. 19 (3) (2020) 266–270, https://doi.org/10.1016/S1474-4422(19)30398-
9.