Keywords:
Restless legs syndrome - Willis-Ekbom disease - diagnosis
Palavras-chave:
Síndrome das pernas inquietas - doença de Willis-Ekbom - diagnóstico
Restless legs syndrome/Willis-Ekbom disease (RLS/WED) is a highly-prevalent disease
but is still little known, both by health professionals and by the population[1],[2]. It causes a great negative impact on quality of life[3], and every physician should be able to at least establish the diagnosis, as the
complaints suggestive of this disease are not restricted to a single specialty[4].
Since the thorough description of RLS/WED by Karl Ekbom[5],[6], the diagnostic criteria have been revised and updated[1],[2],[7],[8],[9] with the objective of facilitating recognition of the disease and reducing false-positive
diagnoses[10]. Several clinical conditions[10] can meet all four diagnostic criteria for RLS/WED[2] and still not constitute the disease per se. Thus, symptoms that mimic these criteria are confounding factors that affect the
proper diagnosis of RLS/WED. Within this context, an attempt was made to facilitate
diagnosis of this disease by introducing a fifth criterion (Appendix), which addresses
clinical conditions and symptoms important for differential diagnosis; therefore,
this criterion reinforces what RLS/WED is not[1].
In a previous study, we found that replacing the name “restless legs syndrome” with
“Willis-Ekbom disease” greatly altered the prevalence of self-diagnosis. This indicates
that the name RLS tends to be misinterpreted, particularly because the term “restless”
is suggestive of physical and psychological conditions unrelated to the disease, such
as the habit of fidgeting or otherwise moving one's legs in different situations of
daily life[11]. Our hypothesis is that the use of the name WED is enough to improve diagnostic
accuracy, reducing or abolishing the need for the fifth criterion altogether and,
consequently, facilitating proper diagnosis.
METHODS
We studied the impact of the fifth IRLSSG diagnostic criterion on the prevalence of
self-diagnosed Willis-Ekbom disease among recent medical graduates applying for residency
at a highly-competitive Brazilian university. The respondents were divided into two
groups and analyzed their own clinical condition with respect to the diagnosis of
WED. One group (All Five group) completed a self-assessment questionnaire that included
all five IRLSSG criteria (Appendix), while the other completed a questionnaire with
no fifth criterion (No Fifth group).
Population and setting
For this study, we used the same population of recent medical graduates applying for
residency positions at Escola Paulista de Medicina - Universidade Federal de São Paulo,
as described in detail in a prior study[11]. Candidates of both sexes, from all regions of Brazil, completed a less than 10-minute
long questionnaire in the waiting rooms, while awaiting the start of their residency
entrance exams. This was the most appropriate time to approach them, as the candidates
had an interval of around 40 minutes between evaluation phases. There was no restriction
on age or ethnicity. This study was approved by the Universidade Federal de São Paulo
Research Ethics Committee (process number 115.917/2012), and all participants provided
written informed consent.
Self-diagnosis of WED
The participants were instructed to perform a selfassessment of the possibility of
having a diagnosis of WED (yes or no), according to criteria proposed by the IRLSSG.
One group of participants received a form without the fifth criterion (No Fifth group),
while the other received a form containing all five diagnostic criteria for the disease
(All Five group).
Randomization
The investigators randomly distributed two types of forms to the residency exam invigilators.
The invigilators were unaware of which forms they were delivering to the study participants,
and the distribution was made without any specific rules, depending solely on proximity
to the participants or convenience, which further helped ensure randomization.
Statistical analysis
Data found to be normally distributed on the Kolmogorov-Smirnov test were analyzed
through descriptive statistics. We used the error of the sample proportion, considering
a 95% confidence interval (95%CI), to analyze the variability of the sample. The Student's
t-test was used to compare mean age between groups, while the chi-square test was used
to compare the proportion of self-diagnosis.
RESULTS
Demographic data
We invited 783 recent medical graduates to take part in the study. Of these, 705 agreed
to participate and 78 (10%) did not. Among the 705 participants, 360 (51.06%) were
women (95%CI: 47.32%-54.68%) and 345 (48.94%) were men (95%CI: 45.25%-52.69%), with
no statistically significant difference between genders (p > 0.05) ([Table]). The All Five and No Fifth subgroups also did not differ regarding age and sex.
Table
Distribution of participants by sex.
|
Variables
|
N
|
%
|
95%CI
|
p
|
|
All participants
|
|
Women
|
360
|
51.06
|
47.32%-54.68%
|
> 0.05
|
|
Men
|
345
|
48.94
|
45.25%-52.69%
|
|
Stratification of participants into groups All-Five and No-Fifth
|
|
All-Five Group
|
|
|
Women
|
190
|
51
|
45.93%-56.07%
|
> 0.05
|
|
|
Men
|
183
|
49
|
43.93%-54.07%
|
|
No-Fifth Group
|
|
Women
|
170
|
51.20
|
45.90%-56.50%
|
> 0.05
|
|
Men
|
162
|
48.94
|
43.57%-54.31%
|
All Five group
Three hundred and seventy-three recent medical graduates, with a mean age of 26 ±
3 years, received the form containing all five IRLSSG criteria and answered the question:
“Do you have Willis-Ekbom disease?”. Among these, 190 (51%) were female (95%CI: 45.93%-56.07%)
and 183 (49%) were male (95%CI: 43.93%-54.07%) ([Table]).
In this group, 9 (2.41%) (95%CI: 0.8%-4.0%) physicians self-diagnosed WED: 4 women
(44.44%; 95% CI 11.98%-76.90%) and 5 men (55.56%; 95%CI: 23.10%-88.02%) ([Figure]).
Figure Self-diagnosis of WED without vs. with the fifth IRLSSG diagnostic criterion.
No Fifth group
Three hundred and thirty-two recent medical graduates received the form without the
fifth IRLSSG criterion and answered the question: “Do you have Willis-Ekbom disease?”.
Of these, 170 (51.20%) were female (95%CI: 45.90%-56.50%) and 162 (48.94%) were male
(95%CI: 43.57%-54.31%) ([Table]). The mean age in this group was 26 ± 2 years.
In this group, 8 respondents self-diagnosed with WED (2.41%) (95%CI: 0.8%-4.1%): 5
women (62.50%; 95%CI: 28.96%-96.04%) and 3 men (37.50%; 95%CI: 3.96%-71.04%). The
prevalence of self-diagnosis of WED did not differ between men and women ([Figure]).
The proportion of respondents who self-diagnosed with WED per four diagnostic criteria
(No Fifth group) did not differ from that of respondents who used all five diagnostic
criteria (All Five group) (p > 0.05). The proportions of diagnosis of WED did not
differ by sex.
DISCUSSION
In this study, we demonstrated that the presence of the fifth criterion proposed by
the IRLSSG in 2014[1] was not relevant for self-diagnosis of WED among recent medical graduates. We used
the self-diagnosis strategy as a motivation tool, aiming to obtain maximal attention
from the participants. This study was not designed to explore the prevalence of RLS/WED.
In a previous study, we showed that the different names given to this disease altered
the prevalence of self-diagnosis, which was much higher when using the expression
“RLS”[11]. As discussed previously, the term “restless”, in particular, promotes a major change
in the way the individual perceives information; their own experiences and concepts
alter the meaning associated with the word. The fifth diagnostic criterion is thus
only necessary because of the use of the term “restless legs syndrome”.
Diagnosis of RLS is based on the patient's report. However, symptoms that mimic the
disease may be present[10], including cramping, positional discomfort, pain, and local leg pathology[12], as these may satisfy all four criteria for diagnosis of RLS. To improve diagnostic
specificity, these clinical conditions must be ruled out[12]. Hence, the addition of the fifth diagnostic criterion in 2014. In other words,
the fifth criterion aims to facilitate recognition of the disease by characterizing
what it is not, to define it in its full clinical and technical sense.
There are several controversies regarding the use of eponyms (from the Greek epi, “upon”, and onyma, “name”). However, in the case of WED, the eponymous term has several advantages,
including obviating the need for an additional diagnostic criterion, thus streamlining
recognition and treatment of this very common disease.
Furthermore, there is still much ignorance about this condition among physicians and
the general population. When healthcare providers are faced with the expression “restless
legs syndrome”, the term “restless” calls to mind meanings that do not represent WED
accurately. This prevents clinicians from seeking more information about the disease
and asking patients for a detailed description of their symptoms, as the clinicians'
own experiences with the term “restless” lead them to assume they are knowledgeable
about the condition and thus neglect its true technical definition[11]; the term “Willis-Ekbom disease” is not associated with such preconceived notions.
Another speculated reason why the 5th criterion was not relevant in our present study is related to a possible consecutive
understanding of each criterion as the participant reads the sequence of items, allowing
them to complete their understanding even before they reach the 5th criterion.
Our study has some strength since we randomly distributed the forms, the population
is highly educated making it easier to understand our purpose, we have a large number
of participants and, most important, they were physicians (recently graduated) and
the main reason and to whom these criteria are finally destined. A possible weakness
would be a low probability of some mimics, although positional discomfort, and habitual
foot tapping are the most relevant confounders and frequently reported in this population,
according to our data from our previous study.
In summary, our study suggests that the fifth IRLSSG criterion is unnecessary when
using the expression WED in a population of recently graduated physicians, reinforcing
the need for and importance of introducing it into daily clinical practice. In addition
to giving this highly impactful condition the seriousness it deserves, this shift
would motivate clinicians to understand its true symptoms, facilitate and streamline
diagnosis, and prevent derogatory and degrading remarks related to the expression
“restless legs”.
