I bet your doctor knows nothing solving about stroke fatigue. Mine only
suggested exercise without even testing my cardiovascular fitness which
was already at the level of an athlete.
Fatigue: the unexplained phenomenon
‘On an examination of what takes place in fatigue, two
series of phenomena demand our attention. The first is the diminution of
muscular force. The second is fatigue as a sensation. That is to say,
we have a physical fact which can be measured and compared, and a
psychic fact which eludes measurement. With regard to the feeling of
fatigue, the same thing takes place as happens in the case of every
stimulus which acts upon our nerves: we begin to perceive it only when
it has attained a certain intensity.’ Angelo Mosso, 1891
One
hundred and twenty-five years later, our understanding of fatigue,
specifically the sensation of fatigue, has progressed little. Mosso’s
description of fatigue is in the context of repetition-induced,
reversible, non-pathological, neuromuscular fatigue. However, his
observations on the sensation of fatigue are pertinent to our
understanding of pathological fatigue. First, the idea of an organic
cause for what is essentially a perceptual construct and second, the
reference to a feature within the nervous system with attributes of
intensity, as the generator to explain the subjective nature of fatigue
sensation, are both useful to progress our understanding of the feeling
of fatigue.
The aim of this essay is to critically appraise the
phenomenology of fatigue and evaluate recent findings to develop
plausible mechanistic (functional) hypothesis that may explain
pathological fatigue. To this end, I first describe the defining
features of pathological fatigue and what sets it apart from other
affective symptoms. Second, I elaborate on a potential sensorimotor
mechanism, within the framework of active inference, to explain altered
effort perception. Third, I make a case for fatigue as a baseline
perceptual state and discuss how abnormal perceptual state arises from
altered effort perception. Fourth, I discuss fatigue in the context of
higher order meta-cognitive functions and finally provide some pointers
for future research.
The classic symptoms associated with
neurological disorders can be broadly classified into motor, cognitive
deficits and affective symptoms. Our understanding of—and ability to
quantify and manipulate—motor and cognitive deficits is more developed
than our understanding of affective symptoms. However, the resolution of
affective symptoms, especially persistent chronic affective symptoms,
is a greater priority for many patients. I will assume that affect is
fundamentally a product of inference, arising from mental and physical
causes. Fatigue is a poorly understood affective symptom, associated
with both neuromuscular and cognitive states. Although the notion that
physical and mental fatigue are two separate constructs is commonplace
in literature, if fatigue in itself is an inference is must be a single
construct, irrespective of whether it is based on physical or mental
evidence. In light of this, I will use the sensorimotor system to
illustrate the basic idea, under the assumption that similar principles
can be applied in other (e.g. interoceptive) domains to explain mental
fatigue.
Within the gamut of affective symptoms, chronic fatigue
is particularly difficult to investigate due to: (i) absence of an
obvious trigger; (ii) its subjective nature with no reliable, objective,
measurable behavioural surrogate; (iii) its significant overlap with
apathy and depression; (iv) the popular belief that fatigue is a
secondary symptom, despite evidence to the contrary; (v) the lack of a
precise definition; and (vi) confusing terminology. Fatigue and
fatigability are used interchangeably, despite recent attempts to
clearly distinguish between the two. All of the above are closely
linked: absent trigger, subjectivity and overlap result in poor
definitions and a confusing terminology leading to skewed popular
beliefs. Despite the challenges of investigating fatigue in a systematic
and methodical manner, clear ways forward are now emerging.
Fatigue, apathy and depression
A starting point is to define fatigue, not from an experiential
perspective, which, because of its subjective nature, engenders as many
definitions as there are respondents, but to define fatigue from a
mechanistic perspective. We do not have a good mechanistic understanding
of fatigue; however, examining the similarities and differences between
fatigue, apathy and depression might throw some light on the nature of
fatigue. Behaviourally, all three phenomena are defined by a significant
reduction in self-initiated voluntary action. Self-initiated voluntary
action calls on two systems; the motivational system to self-initiate,
and the executive (sensorimotor) system that delivers voluntary action.
It is reasonably well established that the peripheral machinery involved
in voluntary action—and to a large extent the central systems directly
involved in driving action—are intact, which leaves us with the
motivational system. Based on established motivational theories, there
are (broadly speaking) two facets to the motivational system; neural
processes involved in directional aspects and activational aspects.
Directional systems determine if behaviour should be directed towards or
away from stimuli; for example, towards food and away from fire and
activational systems determine the ‘vigour’ of the behaviour, how
quickly to run towards food and away from fire. To summarize,
directional systems inform choice and activational systems inform action
(
Salamone et al., 2016).
In the phenomena of interest, fatigue, apathy and depression, a
reduction in self-initiated voluntary action can be attributed to
dysfunction of either directional or activational systems or both; with
subtle differences between the three reflecting a differential
dysfunction of the two systems. In apathy and depression, patients
normally are not interested in performing actions while in fatigue;
despite wanting to act, they feel unable. This subtle distinction could
suggest that in apathy and depression, dysfunctional directional systems
may play a greater role. Conversely, dysfunctional activational systems
may predominate in fatigue. There is no direct evidence to corroborate
an activational dysfunction theory of fatigue; however, later I discuss
how action cost, a component of the activational system, may be
associated with fatigue. A further point of distinction between fatigue,
apathy and depression is that fatigue is always self-reported (i.e. a
symptom), while apathy and depression can be identified and diagnosed by
an external observer (i.e. signs), suggesting fatigue is primarily a
perceptual (inference) phenomenon. Based on clinical presentation,
subtle distinctions from similar affective symptoms and our
understanding of the underlying mechanisms, a plausible definition
follows: ‘fatigue is a percept arising primarily from alterations within
the activational systems that inform voluntary action’.
Effort and fatigue
How does a percept, or subjective awareness, of a dysfunction arise?
First we must analyse how subjective awareness of a function comes
about. For purposes of illustration, let us consider the sensorimotor
system. The processes that translate thought into movement are highly
automated and for the most part, the agent is unaware of the many
processes. However, neural processes that encode information (variables)
that could potentially inform explicit choice must have specific
properties, which allow the agent to experience or become aware of the
‘information’ encoded. By definition, awareness comes about when a
threshold is crossed—or hypothesis is selected; thereby the property of
intensity is a prerequisite for any neural process that encodes
variables that are experienced by the agent. Within the sensorimotor
system, one such variable—which usefully informs both implicit and
explicit motor choice (and enters awareness)—is movement/action cost,
normally experienced and reported as ‘effort’. ‘Effort’ by definition is
an inference or perception; however, there is some confusion in the
literature and at times effort is used interchangeably with force, and
what is referred to here as effort is normally alluded to as ‘perceived
effort’. To avoid any confusion, I use ‘perceived effort’ for the rest
of the manuscript.
Perceived effort is a dynamic variable and is
heavily influenced by expectations and feedback. Experimental evidence
suggests that perceived effort for a given task correlates with
pre-movement neural activity and in models of absent afferent feedback,
intention to move relates to effort (
Lafargue and Franck, 2009).
This evidence supports the notion that perceived effort arises from
efferent commands, possibly via motor efference copy. However, other
studies suggest that perceived effort is significantly altered by
manipulating (re)afferent feedback from the muscles. We also know
afferent feedback is evaluated centrally in the context of motor
intention. Therefore, perceived effort has its origin in intentions
(efferent information) that contextualizes feedback (afferent
information). In short, ‘perceived effort’ is a perceptual inference
that integrates efferent and afferent information.
The active
inference framework of sensorimotor control provides a simple framework
that integrates efferent and afferent inputs to explain movement
initiation and motor control (
Brown et al., 2013).
Here, I consider how ‘perceived effort’ could be the perceptual
consequence of active inference. The active inference framework of
sensorimotor control postulates that the (efferent) output from
cortico-motor system is in the form of sensory (proprioceptive)
predictions and (afferent) input from the somatosensory systems is in
the form of sensory (prediction) errors (
Fig. 1).
On this view, descending predictions of proprioceptive input are
fulfilled by classical motor reflexes (or autonomic reflexes in the
context of interoceptive predictions). In other words, descending
proprioceptive predictions play the role of both motor commands and
efference copy, depending on the sensory (proprioceptive or
somatosensory) modality predicted. Crucially, to engage reflexes it is
necessary to attenuate the precision or gain of afferent or ascending
prediction errors that report the fact no movement has been elicited. In
short, to perceive the consequences of movement
post hoc,
sensory errors must be heeded to; however, to initiate movement, sensory
errors must be ignored. In other words, we have to transiently suspend
attention to sensory evidence we are not moving. By altering the
precision (or intensity) of sensory prediction errors one can either
heed or ignore sensory errors. This function of altering the precision
of sensory errors is commonly referred to as ‘sensory attenuation’.
Sensory attenuation is elegantly demonstrated in the force matching
task. When one is required to match an externally applied force with an
internally generated force, one typically overshoots and produces a
higher force (
Shergill et al., 2003).
This overshooting is a result of attenuating the intensity of the
sensory consequences of a self-generated motor act, which results in a
given force being perceived as less forceful. As a result, when one
tries to match the sensation produced by the externally generated force,
one overshoots; in other words, one underestimates the force one
produces. This is a relatively well studied phenomenon and we now know
that sensory attenuation is at its strongest at low force levels and
weakest at higher force levels (
Walsh et al., 2011).
A
schematic representation of how sensory attenuation may underpin
perceived effort during movement, using the active inference framework
of sensorimotor control. The descending commands from the brain
specify sensory predictions (Efferents) that are compared with the
incoming sensory signals (Afferents)—giving rise to sensory prediction
errors (Prediction-Sensory Input = Prediction Error). ‘To attend or not
to attend’ to the sensory prediction errors, which drive motor output
(Descending Prediction Error), depends on the precision the brain
affords them (Ascending Prediction Error). When sensory precision is
high, prediction errors are heeded to, which I hypothesize gives rise to
high perceived effort. On the contrary, when sensory precision is
attenuated, afferent sensory errors are ignored resulting in sense of
effortlessness. In conditions where sensory attenuation is impaired,
incongruent perceived effort may arise leading to perceptual feeling of
fatigue.
How might sensory attenuation underpin the
experience of movement (i.e. ‘perceived effort’)? If one takes the
example of a muscle contraction, under circumstances of normal sensory
attenuation, ascending signals (proprioceptive prediction errors) that
drive muscle contraction are suppressed (or ignored), which leads to the
inference of less or no effort. On the contrary, in the absence of, or
when sensory attenuation is poor, the same muscle contraction will be
accompanied by ascending proprioceptive prediction errors that can only
be explained (by the brain) if the movement requires more effort or work
than predicted. It is known that attenuation is stronger in low force
muscle contractions than stronger muscle contractions. Similarly,
perceived effort has a non-linear relationship with force produced;
possibly due to the stronger attenuation at play at low level forces.
Although there is no direct evidence, the similarities in
‘force-perceived effort’ and ‘attenuation-muscle contraction’
relationships, strengthens the argument that sensory attenuation
underpins perceived effort.
Indirect evidence from disease states
further endorses this idea. A classic feature of pathological fatigue is
a report of high effort and performing simple activities of daily
living. Activities of daily living generally require low levels of
muscle force and—under normal circumstances—low level muscle contraction
is associated with high sensory attenuation, rendering an ‘effortless’
inference about such activities. However, if sensory attenuation were
impaired, daily motor acts would be experienced as effortful—a classic
symptom of pathological fatigue. Here, it is useful to remember that
pathological fatigue does not correlate with muscle weakness. We know
sensory attenuation is incomplete when a system is working at its
hardest; for example, when maximal muscle contraction is required.
Muscle activity required for simple activities is (near) maximal in a
weakened muscle, in the absence of which, one might assume, high
perceived effort is a result of impoverished sensory attenuation.
Therefore, in chronic pathological fatigue, simple activities feel
effortful due to the brains inability to ignore the afferent
somatosensory consequences of movement. Prolonged, consistent experience
of high perceived effort could therefore eventually lead to the report
(or symptom) of fatigue.
Fatigue, rest and multiple pathologies
We now have a candidate mechanism that potentially explains the
predominant feature of chronic pathological fatigue; however, it is as
yet unclear how an action-related mechanism explains chronic fatigue at
rest. Rest is a word used to describe lack of explicit behaviour; which
is not to say the brain is at rest, as spontaneous neuronal firing at
‘rest’ is a well-established fact (
Fox and Raichle, 2007).
Scientists have only recently started to explore the significance of
spontaneous or endogenous neuronal firing. In terms of metabolic cost,
spontaneous neuronal firing consumes 20% of body’s energy, while
task-related energy consumption accounts for <5 a="" class="link link-ref link-reveal xref-bibr" data-open="awx153-B3">Fox and Raichle, 20075>
).
In short, ‘resting state’ is a misnomer. What does spontaneous neuronal
activity encode? The most popular, mutually non-exclusive theories
hypothesize that spontaneous synchronous activity represent a rehearsal
of previous task-related use, and the other suggests it rehearses
predicted motor scenarios.If one takes a Bayesian view of the
brain, both hypotheses are true—as prior activity informs future
predictions; therefore, the rehearsal of completed tasks influences
spontaneous neuronal firing, which then informs future actions. In the
context of pathological fatigue, a memory of effortful activities
(resulting from poor sensory attenuation) influences resting state
spontaneous neuronal firing. In a recent resting state functional MRI
investigation, pattern recognition techniques have shown that
spontaneous fluctuations in resting state neuronal firing relate to
individual differences in mood and personality traits, and predict
online, self-reported feelings such as sadness. Fatigue, another such
feeling, could also be encoded in spontaneous neuronal firing while the
brain is at rest. A perceptual state at rest may then arise from
spontaneous neuronal activity that is influenced by previous
effort-related abnormal (aberrant sensory attenuation) neuronal
activity.
Can the above proposed mechanisms of chronic fatigue
hold true across several pathological conditions? The mechanisms
proposed thus far identify brain-mediated functions that explain an
abnormal perception of effort, a salient feature of chronic fatigue.
Chronic fatigue is prevalent in a large number of pathological
conditions: neurological, autonomic, immunological, hormonal and
cardiovascular diseases not to mention a significant side effect of many
pharmacological interventions. Here, I propose sensory attenuation as a
fundamental mechanism that underpins effort perception, and aberrant
sensory attenuation as a disease-independent mediator of fatigue, which
in some cases may be the primary driver and in others, a knock-on effect
of a more proximal problem. Fundamentally, fatigue can be viewed as the
end point manifestation of a cascade of events activated by
disease-specific triggers, and—following resolution of the primary
trigger—the cascade of events reverse. However, failure to reverse
changes in sensory attenuation results in chronicity of the ensuing
symptom; namely, fatigue. The point of reversal failure determines if
impoverished sensory attenuation is the driver or a mediator of fatigue.
A key aspect of these putative failures rests on the similarity between
selective attention and sensory attenuation. In active inference, these
are both sides of the same coin corresponding to a centrally mediated
increase and decrease in precision (i.e. postsynaptic gain of neurons
encoding prediction errors at various levels of processing hierarchies).
In other words, there is an intimate relationship between attention and
attenuation; both of which have to be carefully orchestrated through
descending predictions of precision—or top-down gain control. On this
view, disruption to higher order cognitive functions such as endogenous
attention may contribute to, or be intimately conflated with, the
development of fatigue.
Fatigue and agency
The inability to suppress anticipated neuromuscular sensory
consequences of motor commands as a primary cause of pathological
fatigue is proposed here for the first time. However, poor sensory
attenuation, as demonstrated by a perceptual behavioural task, is an
established correlate of disorders of agency (
Lafargue and Franck, 2009).
How then, can one reconcile the idea of fatigue and disruption of
agency as both driven by similar mechanisms? For this, we must examine
the common denominator between the two; namely, effort. In fatigue there
is a greater sense of effort and in disorders of agency there is a lack
(external attribution) of effort. Research is at very early stages and
there is little evidence to draw further conclusions; however, one might
plausibly speculate on how effort ties together the two seemingly
unrelated phenomena. In fatigue (post-stroke), patients often report a
loss of control over their body and simple movements require high
effort. In disorders of agency, patients attribute control to an
external agent or having no control of their actions. One might
speculate that different degrees of reported loss of control could be
mapped on to different levels of sensory attenuation with: (i) normal
sensory attenuation relating to reports of full control over their
movements; (ii) in partial sensory attenuation, patients report partial
loss of control over their movements, or find it difficult to move their
bodies; and (iii) in a state where sensory attenuation fails
completely, a total loss of control over their movements is reported, or
loss of agency. Moreover, recent evidence suggests effort and sense of
agency interact (
Demanet et al., 2013).
Therefore, fatigue, a perceptual disorder of the sensorimotor system,
the system through which the agent fulfils predictions of agency, can be
placed within the spectrum of agency-related disorders.
Concluding remarks and future perspectives
Thus far, I have elaborated a framework within which one can define
and understand the symptom of fatigue from a physiological standpoint
and place it in the broader context of multiple pathologies. Recent
investigations provide indirect evidence for pathological fatigue being a
disorder of sensory attenuation in neurological conditions. Low motor
cortex resting-state excitability in the stroke affected hemisphere in
the fatigue group, despite no difference in sensorimotor impairment and
behavioural outcome (
Kuppuswamy et al., 2015b)
raises two questions, the first, ‘does low excitability give rise to
fatigue and how?’ Suppression of motor cortex excitability using
inhibitory repetitive transcranial magnetic stimulation protocols in
healthy volunteers, results in poor sensory attenuation as evidenced by
more veridical force matching in the force-matching task, hence low
excitability of stroke affected hemisphere may reflect poor sensory
attenuation. Second, if sensory attenuation is critical for motor
initiation, how might we reconcile the lack of sensorimotor impairment
with poor attenuation? In this study (
Kuppuswamy et al., 2015b)
sensorimotor impairment was measured using standard clinical tests that
are not sensitive enough to capture subtle alterations in sensorimotor
control. However, in a further study we showed that self-selected
ballistic movement speeds were indeed compromised in the affected limb
of the high fatigue group (
Kuppuswamy et al., 2015a).
Limb heaviness in stroke survivors in relation to fatigue but not
muscle weakness, indicates abnormal sensory processing of the affected
limb, possibly poor attenuation of sensory afferent information from
resting state muscle tone in the affected limb? Attention deficits have
been related to post-stroke fatigue (
Radman et al., 2012)
and attention is inextricably linked to sensory attenuation. Fatigue in
multiple sclerosis has been thought of as a disorder of movement
preparation as evidenced by altered pre-movement motor cortex
excitability and movement preparation includes predictions of sensory
consequences—a key feature of sensory attenuation. Interventions
targeted at altering motor cortex excitability have thus far shown some
positive effects on fatigue; however, direct evidence linking poor
sensory attenuation to pathological fatigue is yet to emerge. Future
investigations could investigate sensory attenuation in patient cohorts
with a wide range of fatigue levels. A simple, established, robust
behavioural paradigm that allows one to quantify sensory attenuation is
the force matching illusion; see above (
Shergill et al., 2003).
Furthermore, high frequency neuronal oscillatory activity in motor
areas that encode prediction errors and movement parameters both appear
to be causally linked to sensory attenuation and neuro-modulatory
protocols also support a functional role for motor cortex in sensory
attenuation. Therefore, using a combination of behavioural, imaging and
neuromodulation techniques, future studies can, in principle, confirm or
negate the hypothesis: pathological fatigue is a disorder of sensory
attenuation. Interestingly, an explanation based on aberrant sensory
attenuation for other affective symptoms, is starting to emerge in
computational psychiatry—with the combined use of neuroimaging and
dynamic causal modelling to measure sensory attenuation and attention in
terms of neuronal gain; i.e. the synaptic efficacy of intrinsic
neuronal connections.
Fatigue is a perceptual state that is
experienced by all humans, albeit transiently, but when fatigue is
non-transient it starts to significantly impact all aspects of the
sufferer’s lives. Such fatigue is a hallmark of many pathological
conditions and despite more than a hundred years of trying to understand
fatigue, there has been very little progress. Here, I highlight the
features of fatigue that render it difficult for scientific
investigation, propose a unifying definition from a physiological
standpoint, elaborate on a disease-independent mechanism that might
underlie fatigue, discuss evidence in support of the proposed mechanism
and suggest further experiments to verify the hypothesis. If proven to
be true, this framework may provide us with the much needed foundation
on which to build our understanding of fatigue and, more broadly, a
robust link between mind and body.
Acknowledgements
I would like to extend my sincere thanks to Profs Karl Friston and
John Rothwell for the many useful discussions and valuable input to this
manuscript. I would also like to thank Dr Sasha Ondobaka and Mr William
De Doncker for their feedback on this manuscript.
Funding
A.K. is funded by the Wellcome Trust 202346/Z/16/Z.
10 references used to produce this.
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