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Background: Benign fasciculation syndrome (BFS) is quality. Patients with BFS tended to be male and in
characterized by persistent spontaneous contractions of their 30s or 40s. There was an overrepresentation of
muscle fibers in the absence of a pathological cause. clinicians. Anxiety symptoms were common and
Patients with BFS often have concerns around having frequently coexisted alongside fasciculations. Health
motor neuron disease, in some cases fulfilling the criteria anxiety was overwhelmingly focused around motor
for health anxiety disorder. Research on how BFS and neuron disease. Conclusion: A proportion of individuals
health anxiety relate to one another and how they should with BFS experience anxiety around having motor
be optimally managed together is sparse. Objective: We neuron disease—to the point of developing health
report two cases of BFS associated with health anxiety. anxiety disorder. A bidirectional relationship may exist
We also review the literature on the association between between BFS and health anxiety disorder. Clinicians
BFS and health anxiety. Methods: We systematically should be alert to the possibility of health anxiety
reviewed the literature using MEDLINE, Embase, disorder in patients with BFS and have a low
PsycINFO, and OpenGrey for studies investigating threshold to refer for psychiatric assessment. There
benign fasciculations and anxiety up to August 2018. is support for the role of psychological therapy,
Results: Both cases were successfully treated for health especially CBT, as well as pharmacotherapy, in the
anxiety disorder with cognitive-behavioral therapy form of antidepressant medication. In severe or
(CBT) and antidepressant medication. We identified treatment-refractive cases, combined treatment
eight studies that met the inclusion criteria, describing a may be indicated.
total of 384 patients. Most studies were of moderate (Psychosomatics 2019; -:-–-)
INTRODUCTION Received January 25, 2019; revised April 3, 2019; accepted April 4, 2019.
Institute of Psychiatry, Psychology and Neuroscience, King’s College
London (G.B., A.S.D.), South London and Maudsley NHS Foundation
Fasciculations are spontaneous, fast contractions of fine Trust (G.B., F.R.), King’s College London School of Medical Education
muscle fibers, which can be perceived as brief muscle (Y.C., H.M.), Motor Nerve Clinic, King’s College Hospital (C.M.E.,
J.B.), UCL Institute of Mental Health (A.S.D.), London, UK. Send
“twitches.”1 They are experienced by up to 70% of the correspondence and reprint requests to Graham Blackman, MRCPsych,
healthy population2 and are associated with increased Department of Psychosis Studies, Institute of Psychiatry, Psychology and
age, strenuous physical exercise, stress, and fatigue.3 Neuroscience, King’s College London, 16 De Crespigny Park, Cam-
berwell, London SE5 8AF, UK; e-mail: graham.blackman@kcl.ac.uk
Fasciculations can be benign or associated with ª 2019 Academy of Consultation-Liaison Psychiatry. Published
neurological disorders, such as radiculopathy, by Elsevier Inc. All rights reserved.
peripheral nerve injury,4 autoimmunity,5 and peripheral slight residual contrast agent in the pharynx, and
nerve hyperexcitability syndromes.6 Fasciculations can functional endoscopy was normal. He was referred to a
also be the presenting symptom in motor neuron disease tertiary neurology center where initial neurological ex-
(MND), a progressive neurodegenerative disorder.7 amination revealed fasciculations of his left gastrocne-
When fasciculations are persistent, but no patho- mius and normal magnetic resonance imaging (MRI) of
logical cause is identified, patients may be diagnosed the head and cervical spine. Nerve conduction and
with benign fasciculation syndrome (BFS). Diagnosis EMG studies were also normal. He was diagnosed with
typically requires a normal neurological examination BFS and health anxiety and referred to a neuropsy-
and electromyography (EMG), excluding fasciculation chiatry clinic for assessment.
potentials. Since the first description of BFS, an asso- At the peak of his health anxiety symptoms, three
ciation with anxiety around MND has been noted,8 months from symptom onset, he was unable to work, his
particularly in doctors and other health care pro- relationship with his partner was severely strained, and he
fessionals.9 In some cases, it is sufficiently severe to suffered with dysphoria and insomnia. He held intrusive
fulfill the criteria for health anxiety disorder. This is a overvalued ideas around having MND with 90% cer-
psychiatric disorder characterized by a persistent pre- tainty. There was evidence of catastrophizing and rumi-
occupation with having a serious physical illness lead- nation, as well as an excessive interest in news stories
ing to marked emotional distress, despite negative related to MND, and he regularly consulted online BFS
findings and reassurance.1 Alongside these cognitive forums. Reassurance-seeking from medical pro-
and affective components, safety behaviors, such as fessionals, self-testing of muscular strength, and inspec-
excessive checking and reassurance-seeking, are also tion of his body for evidence of muscle wasting were also
defining features. The disorder is synonymous with the prominent. He also switched to eating semi-solid foods
International Statistical Classification of Diseases and and reduced exercise to avoid stimulating fasciculations.
Related Health Problems 10th Revision (ICD-10) He was initially trialled on fluoxetine 20 mg before
diagnosis of hypochondriacal disorder, and the Diag- being switched to mirtazapine 30 mg. This transiently
nostic and Statistical Manual of Mental Disorders lead to insomnia but was otherwise well tolerated. He
(DSM-5) diagnoses of somatic symptom disorder underwent a course of cognitive-behavioral therapy
(where marked somatic symptoms are present) and (CBT) for health anxiety following principles previously
illness anxiety disorder (where somatic symptoms are described10 and focusing around a shared understanding
mild, or absent). Research on how health anxiety and of his condition, cognitive restructuring, dropping of
BFS relate to one another—and how patients suffering safety behaviors, and exposure-response prevention. By
from both disorders should be managed—is sparse. We the seventh session, in conjunction with mirtazapine, his
systematically reviewed the literature and report two health anxiety had fully remitted, and he returned to
cases successfully treated with psychological and work. There was also a subjective reduction of fascicu-
pharmacological interventions. lation symptoms by approximately 50%. At follow-up,
eight months after the completion of CBT, he remained
Case Presentations in remission of health anxiety and related disorders.
Mr. B, a 41-year-old male, worked in finance as a
Mr. A, a 38-year-old male clinical neuroscientist with manager. He had a medical history of irritable bowel
no significant medical history, spontaneously developed syndrome and had no prior contact with mental health
twitching in his left calf, spreading to other parts of his services. Health concerns emerged after an elaborate
leg over two days. He became convinced that he was trick was played on him by colleagues to suggest that he
suffering from MND after noticing subtle muscle had been infected with the Ebola virus. One month later,
asymmetries. Approximately two weeks later, he he became convinced that he was suffering from lung
developed difficulties with swallowing and chewing, cancer, despite normal investigations, and was briefly
and twitching spread to his right leg, chest, abdomen, voluntarily admitted to a psychiatric ward due to health
and face. Approximately four weeks later, he developed anxiety. Approximately one year later, he noticed
brief involuntary muscle spasms around his neck and twitching in his right calf. After researching his symp-
upper limbs before falling asleep, consistent with hypnic toms on the internet, he became concerned that he had
jerks. Barium swallow was normal, except for some MND. An initial neurological examination and EMG
study were normal, with no evidence of denervation or it was interpreted as persistent postural perceptual
fasciculation potentials, and he was diagnosed with BFS. dizziness.11 He developed a new concern that he had
He was trialled on alprazolam, which temporarily contracted Lyme disease and consulted several private
reduced his muscle twitching. Although briefly reas- clinicians, despite advice to limit this, resulting in
sured, he subsequently sought multiple neurological further investigation and offers of various treatments,
opinions regarding the possibility of MND. including antibiotics. We recommenced escitalopram
A trial of duloxetine was ineffective, and sertraline 10 mg for exacerbation of health anxiety. By the end of
was discontinued due to a rash. He had two further CBT and having been established on escitalopram, his
voluntary psychiatric admissions over a 1-year period conviction that he had MND had reduced by over 95%,
due to increasing distress around having MND, and he and his functional status had normalized with a
openly contemplated euthanasia and assisted suicide. Modified Rankin Scale (mRS) score of zero. In addi-
During the third admission, he was treated with escitalo- tion to subjective reports of fewer fasciculations, serial
pram 10 mg and CBT, which reduced his conviction that surface EMG measurements demonstrated a reduction
he had MND from approximately 95% to 50%. He dis- in fasciculation potentials (Figure 1). At follow-up,
continued medication after nine months and was subse- eight months after the completion of CBT, he
quently referred to a specialist neuropsychiatry clinic. remained in remission of health anxiety and related
On assessment, his main concern was a subjective disorders, and dizziness and fasciculation symptoms
experience of slurred speech and spasms of the tongue, had also largely resolved.
suggesting bulbar-onset MND to him. Safety behaviors
included seeking reassurance from family and medical Systematic Review
professionals, strength testing, visiting BFS websites
and online forums, and visually inspecting his body We reviewed the primary literature of BFS and anxiety
dozens of times daily. Neurological examination was symptoms and followed the Preferred Reporting Items
normal; however, he was able to provide video evidence for Systematic Reviews and Meta-Analyses (PRISMA)
of intermittent fasciculations. He commenced a 12- guidelines12 and registered the study on the International
week course of CBT for health anxiety, which was Prospective Register of Systematic Reviews (PROS-
extended to 18 weeks due to the treatment-refractory PERO) (CRD42018107211). We searched MEDLINE
nature of his symptoms. Medically-unexplained sub- (PubMed), Embase, PsycINFO, and OpenGrey to iden-
jective dizziness, especially upon standing, emerged and tify articles and abstracts in all years, up to August 2018,
FIGURE 1. Timeline of treatment, health anxiety symptoms and fasciculations. Treatment focused on CBT, which was later augmented with
escitalopram 5–10 mg. Scores of the Health Anxiety Inventory (right y-axis) demonstrated a reduction in symptom severity over time.
Surface EMG measurements over the right gastrocnemius also reduced (left y-axis).
using the following search terms: “anxiety” OR “hypo- commonly an anxiety disorder. The presence of anxiety
chondria*” AND “fasciculation*” OR “twitch”. Studies symptoms, including somatic complaints, coexisting
were included if patients were assessed for benign fascic- alongside fasciculation symptoms was reported in seven
ulations and neurological examination and EMG had studies. Their prevalence ranged between 29%1 and
ruled out pathological causes. Patients were excluded if 39%.15 In three studies, patients’ concern specifically
their diagnosis was subsequently revised. In addition, around MND was reported, ranging between 14%18 and
studies must have included an assessment of current or 100%.18 Two studies reported anxiety measures,16,19 and
past psychiatric symptoms. Two authors (H.M. and fasciculation potentials were found to correlate posi-
Y.C.) independently screened the titles and abstracts of all tively with patient-rated anxiety levels in one study.16
identified articles. Full articles of the remaining studies One study clinically assessed patients for health anxiety
were reviewed to confirm eligibility. Any dispute over the disorder, which was endorsed in all cases.1 An associa-
inclusion of an article was resolved by a third author tion between anxiety and fasciculations symptoms was
(G.B.). Where two articles reported the same data set, the noted in all five studies that reported factors related to
more comprehensive one was selected. Data extracted exacerbation of fasciculations.1,9,15,16,18
included psychiatric and neurological characteristics, in-
vestigations, treatment, and outcome. Each publication Treatment and Outcome
was assessed independently for quality by two researchers
(H.M. and Y.C.), with any discrepancies resolved by a Four studies reported an intervention for BFS or health
third author (G.B.) using a quality appraisal tool.13 anxiety, consisting of counseling,1 reassurance alone,17
or reassurance plus antidepressant medication.14,15
Follow-up was assessed in six studies, ranging be-
RESULTS tween 6 months19 and 7 years.1 An improvement, or
resolution of BFS symptoms, was reported in 11%19 to
The search of electronic databases yielded a total of 362 50%18 of patients at follow-up. Counseling was asso-
articles (Figure 2). After screening titles and abstracts, ciated with an improvement in health anxiety symp-
20 were reviewed in full. Two additional studies were toms.1 Reassurance and antidepressant medication
included by reviewing the references of included studies. were associated with an improvement in fasciculation
Our search yielded eight eligible articles1,2,14–19 with a symptoms15 though reassurance alone was not.17
total of 384 patients (see Supplementary Data S1 for
table of included studies). Sample size ranged between 9
and 122 patients. All the studies were observational, DISCUSSION
with one using a cross-sectional design16 and the
remaining studies using a case series design.1,9,14,16–19 Through a systematic review of the literature and two
Quality appraisal identified six studies as being of me- case reports, we explored the association between
dium quality and two of low quality (see Supplementary benign fasciculations and health anxiety. A consistent
Data S2 for details of quality appraisal). finding was the tendency for benign fasciculations to
affect men in their 30s or 40s. Also notable was the
Clinical Features association between being a clinician with benign fas-
ciculations and having concerns around having MND,
The mean age of included patients across the studies a finding highlighted as far back as 1951.9 Only one
ranged from 3920 to 4717 years. In every study reporting study specifically enrolled patients with BFS and health
gender,1,15–19 there was a male predominance, and anxiety, and notably, health anxiety focused exclusively
where occupational status was recorded,1,9,17–19 there around MND.1
was an overrepresentation of clinicians. After fascicu- Health anxiety has a prevalence of between 0.8 and
lations, the most common neurological symptoms were 4.5% in primary care.21,22 Recognized risk factors
numbness or paraesthesia, cramps or spasms, and fa- include increasing age,23 previous illness, and traumatic
tigue or subjective weakness, suggesting a somatization childhood experiences,24 and it often coexists with other
propensity in this patient population. mental disorders, particularly anxiety disorders.20 In
Six studies reported previous psychiatric diagnoses, our review, patients with BFS who developed health
which varied between 0%1 and 80%,9 and this was most anxiety symptoms did so at a younger age than the age
Idenficaon
Relevant arcles
from references
(n=2) Full-text arcles excluded
Eligibility
of onset normally seen in health anxiety. A plausible sympathetic nervous system during periods of
explanation for this is that health anxiety is temporally emotional arousal (leading to hyperventilation and
linked to the emergence of fasciculations. motor axon excitability).26
Patients with BFS have been found to have similar
BFS and Health Anxiety levels of concurrent depressive and anxiety symptoms
compared with patients with MND. However, they
Whilst apparent that fasciculations may elicit concern have also been found to have a higher rate of psy-
in some individuals, conversely anxiety symptoms chosomatic symptoms, stress, and previous psychiatric
have been postulated to play a causal role in the illness, leading some researchers to suggest that BFS
emergence of benign fasciculations.1,2,16,18 Several may be a form of somatization disorder.15 Mr. B was
mechanisms have been proposed, such as a heightened notable for his propensity toward somatization,
sensitivity to bodily sensations (driven by greater including gastrointestinal, neurological, and vestibular
internally focused attention)25 and activation of the symptoms. Also noteworthy was the co-occurrence of
Supplementary Data S3 for an example “vicious patients who met the diagnostic criteria for health
flower” model). For example, in Mr. A, avoidance of anxiety disorder. Further research in a representative
exercise was an important safety behavior and patient group using a prospective design (incorporating
therefore identified as a “petal” to be addressed. standardized instruments to assess neurological and
During therapy, exercise was actively encouraged, psychiatric features) is warranted. A randomized
resulting in a transient increase in fasciculations. controlled trial would be particularly informative to
Making a causal link between exercise and fascicu- address the optimal treatment in this patient group.
lations (rather than between MND and fascicula-
CONCLUSION
tions) over time led to desensitization and a sense of
mastery over fasciculation symptoms. Mr. B was
Health anxiety regarding MND in patients with BFS is
notable in the degree of internal self-monitoring that
an underreported phenomenon, to which clinicians
inadvertently maintained health anxiety. This was
appear particularly prone. The relationship between
addressed in therapy through behavioral experiments,
BFS and health anxiety is complex however, a
in which systematic shifting of attentional focus could
bidirectional relationship likely contributes toward
be demonstrated to modulate the severity of symp-
maintaining both sets of symptoms. Neurologists
toms, such as dizziness. Thus, in both cases, identi-
should be alert to the presence of health anxiety coex-
fying and addressing each “petal” ultimately led to a
isting with BFS and resist carrying out unnecessary
reduction in symptom levels.
investigations and consider referral for psychiatric
In the absence of a specific biomarker to exclude
assessment where reassurance is ineffective. From the
MND, a concern for neurologists in the assessment of
available evidence, there is support for the role of
new-onset fasciculations is misdiagnosing a patient
psychological therapy, especially CBT, as well as
for BFS when they have MND.40 However, evidence
pharmacotherapy, in the form of antidepressant medi-
suggests this is uncommon,18 and where it does occur,
cation. Furthermore, in severe or treatment-refractive
patients typically have additional neurological fea-
cases, combined treatment may be indicated.
tures such as cramps41–43 or weakness.1 It is notable
that in both cases, patients sought several neurolog- Conflicts of Interests: The authors declare that they
ical opinions and repeat investigations. Significant have no conflict of interest.
improvements in health anxiety symptoms were only Funding: This article represents independent
demonstrated when these safety behaviors were research part funded by the National Institute for Health
addressed, highlighting the importance of continuity Research (NIHR) Biomedical Research Centre at South
of care and avoiding overinvestigation. After the London and Maudsley NHS Foundation Trust and
completion of a surveillance period to exclude MND, King’s College London. A.S.D. is supported by the
we suggest that patients who do not respond to NIHR Biomedical Research Centre at UCLH. The
views expressed are those of the authors and not neces-
simple reassurance should be considered for a psy-
sarily those of the NHS, the NIHR, or the Department
chiatric review to assess for the presence of health
of Health and Social Care.
anxiety. Patient Consent: Written patient consent was ob-
Limitations and Recommendations tained for both case reports.
for Future Research Acknowledgments: The authors thank the two pa-
tients for their permission to publish their cases. The
While providing a valuable contribution to the field, it authors also thank Ken McKeown for his involvement in
is important to acknowledge some limitations. The case 1 and Prof David Veale for his involvement in
majority of studies were of medium quality, and in case 2.
some cases, low. There was also a high degree of het-
erogeneity with regard to the design, patient popula- SUPPLEMENTARY DATA
tion, and recorded variables. Only one study exclusively
included patients with BFS and health anxiety,1 and no Supplementary data to this article can be
study reported a clinical assessment by a psychiatrist. found online at https://doi.org/10.1016/j.psym.2019.
As such, it is difficult to ascertain the proportion of 04.001.
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