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Factors affecting, Neurologia i Neurochirurgia Polska od 2012
[ Pobierz całość w formacie PDF ]ORIGINAL PAPER/ARTYKU£ ORYGINALNY
Factors affecting the quality of life in hemifacial spasm patients
Czynniki determinuj¹ce jakoœæ ¿ycia chorych na po³owiczy kurcz twarzy
Monika Rudziñska
1
, Magdalena Wójcik
1
, Michalina Malec
1
, Natalia Grabska
1
, Micha³ Szubiga
1
, Marcin Hartel
2
, Andrzej Szczudlik
1
1
Katedra i Klinika Neurologii, CollegiumMedicumUniwersytetu Jagielloñskiego, Kraków
2
Medyczne Centra Diagnostyczne Voxel, Zabrze
Neurologia i Neurochirurgia Polska 2012; 46, 2: 121-129
DOI: 10.5114/ninp.2012.28254
Abstract
Streszczenie
Background and purpose: Hemifacial spasm (HFS), a move-
ment disorder manifested by unilateral spasms of the mus-
cles innervated by the facial nerve, interferes with social life
in about 90% of patients, causing social isolation and depres-
sion and having a significant impact on the quality of life.
The aim of the study was to assess factors affecting the qual-
ity of life in patients with HFS in respect of influence of the
severity of depression symptoms and botulinum toxin type A
(BTX-A) therapy.
Material and methods: Eighty-five out of 129 patients includ-
ed in the HFS database of the Movement Disorders Out-
patient Clinic, Department of Neurology, University Hos-
pital, Cracow who fulfilled the inclusion criteria and had no
exclusion criteria (suffering from concomitant movement dis-
orders, other severe chronic diseases or cognitive impairment)
were studied. Demographic and clinical data (age at onset,
disease duration and accompanying symptoms) were collect-
ed. Severity of HFS was assessed by the five-point clinical
scale and seven-point Clinical Global Impression scale. Qual-
ity of life was assessed with the HFS-36 questionnaire and
severity of depressive symptoms was evaluated with the Beck
Depression Inventory. HFS-36 was performed twice, before
BTX-A injection and two weeks later.
Results: The mean global score of HFS-36 was 47 ± 31
(maximum: 140 pts). Decreased HFS-36 score resulted from
divergent deterioration in all subscales included in the ques-
tionnaire. Independent risk factors of deterioration in HFS-36
Wstêp i cel pracy: Po³owiczy kurcz twarzy (
hemifacial spasm
– HFS), charakteryzuj¹cy siê przewlek³ym wystêpowaniem
jednostronnych skurczów miêœni unerwianych przez nerw
twarzowy, zaburza funkcjonowanie spo³eczne prawie 90%
chorych, powoduj¹c ich spo³eczn¹ izolacjê, a nawet depresjê,
i w konsekwencji pogarsza jakoœæ ¿ycia. Celem badania by³a
ocena czynników determinuj¹cych jakoœæ ¿ycia chorych
z HFS z uwzglêdnieniem wp³ywu nasilenia objawów depre-
syjnych i leczenia toksyn¹ botulinow¹ (BTX-A).
Materia³ i metody: Badaniem objêto 85 ze 129 chorych leczo-
nych w Poradni Kliniki Neurologii Szpitala Uniwersytec-
kiego w Krakowie, którzy spe³niali kryteria w³¹czenia do
badania i u których nie stwierdzono cech wy³¹czaj¹cych
z badania (wspó³istniej¹ce choroby ruchu i inne przewlek³e
choroby o du¿ym nasileniu objawów oraz zaburzenia funk-
cji poznawczych). U wszystkich chorych rejestrowano dane
demograficzne i kliniczne (wiek zachorowania, czas trwania,
objawy towarzysz¹ce). Nasilenie objawów HFS oceniano za
pomoc¹ 7-stopniowej skali
Clinical Global Impression
i 5-stop-
niowej skali klinicznej; badanie jakoœci ¿ycia przeprowadzo-
no z u¿yciem kwestionariusza HFS-36, a nasilenie objawów
depresyjnych oceniano za pomoc¹ inwentarza depresji Bec-
ka. Kwestionariusz HFS-36 wype³niano dwukrotnie, bezpo-
œrednio przed podaniem BTX-A oraz 2 tygodnie póŸniej.
Wyniki: Œredni globalny wynik oceny jakoœci ¿ycia w kwe-
stionariuszu HFS-36 wyniós³ 47 ± 31 pkt (na 140 mo¿li-
wych). Obni¿enie jakoœci ¿ycia dotyczy³o w ró¿nym stopniu
Correspondence address: dr n. med. Monika Rudziñska, Katedra i Klinika Neurologii, Uniwersytet Jagielloñski CollegiumMedicum, ul. Botaniczna 3,
31-503 Kraków, tel. +48 12 424 86 00; faks +48 12 424 86 26, e-mail: rudzinsk@neuro.cm-uj.krakow.pl
Received: 22.07.2011; accepted: 18.01.2012
121
Neurologia i Neurochirurgia Polska 2012; 46, 2
Monika Rudziñska, Magdalena Wójcik, Michalina Malec, Natalia Grabska, Micha³ Szubiga, Marcin Hartel, Andrzej Szczudlik
were increased severity of HFS and depressive symptoms as
well as accompanying trismus. The HFS-36 score depend-
ed on the number and type of accompanying symptoms as
well. Botulinum toxin type A therapy led to a significant
improvement of HFS-36, particularly high in patients with
multiple (> 4) HFS-related symptoms.
Conclusions: The HFS-36 score depends mostly on severi-
ty of HFS, depressive symptoms and occurrence of accom-
panying trismus. It improves after BTX-A treatment.
Key words: hemifacial spasm, quality of life, HFS-36.
wszystkich podskal kwestionariusza. Niezale¿nym czynni-
kiem ryzyka gorszej jakoœci ¿ycia by³o wiêksze nasilenie obja-
wów HFS, wiêksze nasilenie objawów depresyjnych oraz
wspó³wystêpuj¹cy szczêkoœcisk. Wynik w HFS-36 zale¿a³
tak¿e od liczby oraz rodzaju wystêpuj¹cych objawów towa-
rzysz¹cych. Leczenie BTX-A poprawia³o jakoœæ ¿ycia, szcze-
gólnie u chorych z du¿¹ liczb¹ objawów towarzysz¹cych.
Wnioski: Jakoœæ ¿ycia w HFS zale¿y od nasilenia objawów
HFS, nasilenia objawów depresyjnych oraz wystêpowania
szczêkoœcisku i poprawia siê po leczeniu BTX-A.
S³owa kluczowe: po³owiczy kurcz twarzy, jakoœæ ¿ycia,
HFS-36.
nia [10], dementia [15], abnormal executive functions
[21], and limited social support [15,17].
The aim of the study was to identify factors having
a major impact on the quality of life in HFS patients in
respect of depressive symptoms severity and effective
treatment with BTX-A.
Introduction
Hemifacial spasm (HFS) is a nervous system dis-
order manifesting with involuntary clonic or tonic con-
tractions of the muscles innervated by the facial nerve
and affecting one half of the face. Chronic HFS symp-
toms interfere with social functioning in almost 90%
of patients, leading to the loss of self-confidence, social
isolation, and even depression that markedly impair the
quality of life in those patients [1]. Only a few papers
have been published so far on the quality of life in HFS
patients, using various questionnaires and showing a sig-
nificant decrease of quality of life in HFS patients when
compared with control groups [2,3].
The results of the questionnaire-based quality of life
assessment depend on multiple different factors. Tan
and colleagues found that better educated patients who
were better informed about the causes, course and man-
agement of HFS had better quality of life improvement
after botulinum toxin type A (BTX-A) injections than
patients having more limited knowledge on that disease
[4]. Studies on factors that determine the quality of life in
HFS are scarce, however. Despite the known impact of
depression and anxiety on HFS symptoms [5-7], no stud-
ies have been performed so far to evaluate the influence of
those emotions on the quality of life assessed with ques-
tionnaires dedicated to HFS patients, such as HFS-36.
The results of studies published to date and related to oth-
er movement disorders show that depression is the most
important factor determining the quality of life in Parkin-
son disease [8-15], dystonia [16-19], essential tremor
[21], progressive supranuclear palsy [22], multisystem
atrophy [23], Gilles de la Tourette syndrome [24], and
orthostatic tremor [25]. Other factors that affect the qual-
ity of life in those disorders include anxiety [11,20],
severity of symptoms [10,11,15,18,21,23,24], insom-
Material and methods
This study included patients of the Movement Dis-
orders Outpatient Clinic of the Department of Neurol-
ogy, University Hospital of Cracow, who were treated
between 2004 and 2010. Participation in this study was
offered to each patient who attended the clinic and had
HFS diagnosed by a specialist with expertise in move-
ment disorder (M.R.) according to the typical clinical
picture. Patients were included if they provided informed
consent to participate and had no exclusion criteria,
which consisted of concomitant movement disorders,
heart failure, pulmonary, renal or hepatic insufficiency,
or malignancy. Patients were also excluded if they had
cognitive impairment and could not therefore reliably
answer the questions included in the quality of life ques-
tionnaire and in the Beck Depression Inventory (BDI).
Demographic and clinical data of those patients were
collected prospectively in a dedicated electronic data-
base. The collected data included age at onset of the dis-
ease, duration and course of the disease, the affected
side, accompanying symptoms (visual disturbances, pain,
discomfort, dysarthria, sialorrhoea, paraesthesias, brux-
ism, trismus, photophobia, lacrimation, conjunctival irri-
tation, hearing disturbances), factors aggravating or alle-
viating the facial muscle contractions, concomitant
disorders, family history and treatment used. The sever-
ity of HFS symptoms was assessed in each patient before
the BTX-A injection with the 7-point Clinical Global
122
Neurologia i Neurochirurgia Polska 2012; 46, 2
Quality of life in hemifacial spasm
Impression (CGI) scale and with the 5-point scale pro-
posed by Tan and Jankovic [26]. Each patient had mag-
netic resonance imaging (MRI) of the head performed
with the detailed assessment of cerebellopontine angles
to discern between idiopathic and symptomatic HFS.
The quality of life was assessed in all patients with
the HFS-36 questionnaire [27]; severity of depressive
symptoms was evaluated with BDI [28]. The HFS-36
questionnaire used in the present study contains sub-
scales related to eight functional domains, i.e. mobility
(5 questions), activity of daily living (5 questions), emo-
tional well-being (7 questions), stigma related to the dis-
ease (5 questions), social support (3 questions), cogni-
tion (3 questions), communication (3 questions), and
complaints of bodily discomfort (5 questions) associat-
ed with symptoms accompanying HFS, such as hear-
ing problems, sleep disorders, facial paraesthesias, eye
irritation, lacrimation, photophobia and sialorrhoea.
Each question in the HFS-36 questionnaire is scored
from 0 to 4 points; maximum score is 140 points.
The severity of depressive symptoms was evaluated
with BDI, which contains 21 questions, each scored
from 0 to 3 points. The BDI score was interpreted
according to the following scores: 0-9 points – normal;
10-19 points – mild depressive symptoms; 20-30 points
– moderate depressive symptoms; > 30 points – severe
depressive symptoms [29]. Depression was diagnosed
according to the opinion of the consulting psychiatrist
or due to ongoing treatment with antidepressant(s)
prescribed previously by the psychiatrist after earlier
diagnosis of depression.
The quality of life and severity of depressive symp-
toms were assessed always after the patient’s qualifica-
tion for the next BTX-A injection, at least 12 weeks after
the previous injection, i.e. after the resolution of the ther-
apeutic effect of the previous BTX-A dose. Patients were
evaluated just before the BTX-A (Botox
®
) injection in
a total dose of 25 U to the five standard locations with-
in the face and were scheduled for the follow-up visit
after two weeks for the repeated assessment of quality
of life.
coefficient was used to assess the reciprocal associations
between numerical variables. Univariate and multivari-
ate logistic regression models were used to assess risk
factors. A
p
-value < 0.05 was considered significant for
all analyses. All statistical analyses were conducted using
commercial statistical software (STATISTICA for Win-
dows, v.6.0, StatSoft Inc., version 9.2, Poland) licensed
to Jagiellonian University.
Results
This study included 85 out of the 129 patients diag-
nosed with HFS and registered in the database (Table 1).
Twenty-six patients did not consent to participate,
and 18 other patients had various exclusion criteria.
The studied group did not differ from all registered
patients regarding age (mean age of studied patients and
all registered patients: 60.8 ± 10.3 vs. 61.6 ± 11.4,
Table 1. Characteristics of the studied patients
Number of patients
85
Sex (women)
58 (69%)
Age [years], mean ± SD
60.8 ± 10.3
Age at disease onset [years], mean ± SD
53.2 ± 12.6
Disease duration [years], mean ± SD
7.1 ± 5.1
Severity of hemifacial spasm:
Clinical Global Impression Scale score,
5.3 ± 1.1
mean ± SD
Five-point scale score, mean ± SD 2.8 ± 0.8
Number (%) of patients with particular severity grade:
grade 0
0
grade 1
6 (7.0%)
grade 2
19 (22.3%)
grade 3
47 (55.3%)
grade 4
13 (15.3%)
Number (%) of patients with depression
17 (20%)
diagnosed with DSM-IV
Severity of depressive symptoms according
to Beck Depression Inventory (score):
0-9 (normal)
Statistical analysis
43 (50.6%)
Numerical variables were characterised with mean
± standard deviation (SD). All analysed numerical vari-
ables had normal distribution; hence Student’s
t
-test was
used to assess the differences in variances. The statisti-
cal significance between categorical variables was
assessed with the
10-19 (mild depressive symptoms)
20 (23.5%)
20-30 (moderate depressive symptoms)
11 (12.9%)
> 30 (severe depressive symptoms)
11 (12.9%)
χ
2
test. Spearman’s rank correlation
SD – standard deviation
123
Neurologia i Neurochirurgia Polska 2012; 46, 2
Monika Rudziñska, Magdalena Wójcik, Michalina Malec, Natalia Grabska, Micha³ Szubiga, Marcin Hartel, Andrzej Szczudlik
respectively), sex (women: 69% vs. 68%, respectively),
mean age at onset of symptoms (53.2 ± 12.6 vs.
52.5 ± 12.2 years, respectively) and mean duration of
disease (7.1 ± 5.1 vs. 9.1 ± 10.3 years, respectively).
Mean score of the global quality of life assessment,
i.e. the sum of all subscores obtained in HFS-36, was
47.0 ± 30.9 (maximum: 140 pts). Mean scores in par-
ticular quality of life domains assessed in subscales of that
questionnaire were as follows: mobility 5.45 ± 4.9 (max-
imum 20 pts); activity of daily living 5.7 ± 4.4 (maximum
16 pts); emotional well-being 9.1 ± 7.3 (maximum 28 pts);
stigma 9.2 ± 5.9 (maximum 20 pts); social support
2.6 ± 3.0 (maximum 12 pts); cognition 4.7 ± 3.6 (max-
imum 12 pts); bodily discomfort 7.9 ± 4.9 (maximum
16 pts); communication 2.2 ± 2.8 (maximum 16 pts).
The severity of symptoms in HFS-36 correlated
with the severity of symptoms in the CGI scale (
r
= 0.28,
p
= 0.009) and in the 5-point scale assessing the sever-
ity of HFS (
r
= 0.37,
p
= 0.002), as well as with the
number of symptoms accompanying HFS (
r
= 0.37,
p
= 0.001). The strongest correlation was noted between
symptom severity in HFS-36 and severity of depressive
symptoms in BDI (
r
= 0.56,
p
= 0.0000) (Fig. 1);
there was no correlation, however, between HFS-36
score and age or duration of the disease or age at onset
of symptoms (Table 2). Women had worse quality of life
than men in the subscale assessing the stigma (χ
panying HFS with the greatest impact on the quality of
life assessed in many HFS-36 subscales. Some accom-
panying symptoms, such as dysarthria (reported by
22.3% of patients), sialorrhoea (17.6%), bruxism
(5.9%), lacrimation (34.1%), or eye irritation (27%),
had no significant impact on quality of life. Some other
symptoms, such as visual disturbances, paraesthesias,
photophobia, hypoacusis and ear clicks, significantly
worsen quality of life in single subscales, e.g. bodily dis-
comfort or communication (Table 3).
Multivariate logistic regression revealed that the
independent risk factors of worse quality of life in
patients with HFS included greater severity of disease
symptoms in the five-point scale (OR: 2.37; 95% CI:
1.14-4.9,
p
= 0.02), greater severity of depressive symp-
toms (OR: 1.10; 95% CI: 1.04-1.17,
p
= 0.001) and
the presence of trismus (OR: 6.44, 95% CI: 1.21-34.31;
p
= 0.03). In the parallel model, including the CGI
scale instead of the five-point scale, greater severity of
HFS symptoms was shown to be an independent risk
factor of worse quality of life in HFS patients as well
(OR: 1.73; 95% CI: 1.24-2.41,
p
= 0.001). The five-
point scale and CGI scale were alternately included in
the multivariate logistic regression model because they
measure the same feature, i.e. severity of HFS symp-
toms, and their close correlation could disturb the appro-
priate modelling of independent risk factors.
Treatment with BTX-A markedly improved the qua-
lity of life scores in studied patients with HFS; mean
HFS-36 score decreased from 47.0 ± 30.9 before treat-
ment to 28.6 ± 23.7 after treatment (
t
= 4.3,
p
=
0.00003). Significant improvement was noted in all
subscales except for the social support subscale (Table 4,
Fig. 2).
The impact of treatment with BTX-A on quality of
life was also assessed in relationship to the number of
symptoms accompanying HFS. Patients were divided
into two groups: (a) those with less than 4 accompany-
ing symptoms, and (b) those with
2
= 2.3,
p
= 0.04).
The BDI score did not correlate with age, sex, age
at onset of symptoms or with duration of the disease;
it correlated, however, with scales used to assess the se-
verity of HFS: CGI (
r
= 0.32,
p
= 0.003) and five-
point scale (
r
= 0.35,
p
= 0.001).
Facial pain or discomfort (reported by 20% of pa-
tients) and trismus (15.3%) were the symptoms accom-
140
120
100
80
60
40
20
0
4 accompanying
symptoms. Sixty-two (72.9%) patients reported less than
4 accompanying symptoms and 23 (27.1%) had at least
4 accompanying symptoms. Patients with a smaller num-
ber of accompanying symptoms did not differ from those
with a greater number of accompanying symptoms
regarding age (59.7 ± 12.4 vs. 59.4 ± 8.1 years, respec-
tively), age at onset of symptoms (52.7 ± 13.1 vs. 53.1
± 9.2 years, respectively) or duration of the disease
(6.8 ± 4.9 vs. 7.3 ± 6.0 years, respectively). Patients
with a greater number of accompanying signs had
greater severity of HFS motor symptoms in the CGI
≥
0
10
20
30
40
50
60
BDI
Fig. 1. Correlation between the severity of depressive symptoms according to
the Beck Depression Inventory (BDI) and the quality of life assessed by HFS-36
124
Neurologia i Neurochirurgia Polska 2012; 46, 2
Quality of life in hemifacial spasm
Table 2. The relation of quality of life assessments (HFS-36 total score and subscale scores) with the clinical features of the disease
Clinical
HFS-36 subscale scores
HFS-36
features
total
Mobility
Activity Emotional Stigma
Social
Cognition
Bodily
Communi-
of HFS
score
of daily
well-being
related
support
discomfort
cation
living
to the
disease
Age*
NS
NS
NS
r
= –0.23
NS
NS
NS
NS
NS
p
= 0.03
Sex**
NS
NS
NS
women:
NS
NS
NS
NS
NS
t
= 2.3;
p
= 0.02
men: NS
CGI score*
NS
r
= 0.26
r
= 0.25
NS
r
= 0.25
r
= 0.27
NS
NS
r
= 0.28
p
= 0.016
p
= 0.02
p
= 0.02
p
= 0.01
p
= 0.009
Five-point scale
r
= 0.25
r
= 0.34
r
= 0.32
r
= 0.29
r
= 0.30
r
= 0.37
r
= 0.33
r
= 0.31
r
= 0.37
score (severity of
p
= 0.02
p
= 0.001
p
= 0.003
p
= 0.007
p
= 0.005
p
= 0.001
p
= 0.002
p
= 0.003
p
= 0.0000
HFS symptoms)*
Side**
NS
NS
NS
NS
NS
NS
NS
NS
NS
Number of
NS
r
= 0.30
r
= 0.32
r
= 0.21
r
= 0.29
r
= 0.36
r
= –0.45
r
= 0.35
r
= -0.37
accompanying signs*
p
= 0.006
p
= 0.003
p
= 0.05
p
= 0.008
p
= 0.001
p
= 0.00
p
= 0.001
p
= 0.001
Duration
NS
NS
NS
NS
NS
NS
NS
NS
NS
of the disease*
Beck Depression
r
= 0.33
r
= 0.38
r
= 0.56
r
= 0.47
r
= 0.47
r
= 0.52
r
= 0.53
r
= 0.41
r
= 0.56
Inventory score*
p
= 0.002
p
= 0.0000
p
= 0.0000
p
= 0.0000
p
= 0.0000
p
= 0.0000
p
= 0.0000
p
= 0.0000
p
= 0.0000
Age at onset
NS
NS
NS
NS
NS
NS
NS
NS
NS
of the disease*
HFS – hemifacial spasm; CGI – Clinical Global Impression; NS – non-significant
* Statistical analysis was performed using Spearman’s rank correlation coefficient
** Statistical analysis was performed using Student’s t-test
contain any studies using HFS-36 in a control group
except for studies performed for validation purposes;
this paucity results from the specificity of the question-
naire, which is directed towards the symptoms typical
for the disease; the expected result of the study in a con-
trol group would therefore be close to zero.
Both our study and the study of Huang
et al.
[27]
showed that the following subscales had the greatest
impact on the global score of quality of life scale: stig-
ma (mean score in our own study and in the mentioned
study: 9.9 and 31.7 pts, respectively), bodily discomfort
(8.0 and 16.9 pts) and emotional well-being (9.5 and
15.7 pts). Communication (2.6 and 2.8 pts) and social
support (2.9 and 1.1 pts) had the smallest impact on
quality of life. Despite the similar relationships regard-
ing the impact on the global score, there is a marked dif-
ference in terms of mean scores between our own study
and the paper of Huang and colleagues [27]. It may
result from cultural differences. Huang
et al.
[27] per-
scale (5.6 ± 0.9 vs. 4.7 ± 1.3 pts;
p
= 0.005) and in
the five-point scale (3.0 ± 0.5 vs. 2.5 ± 1.0 pts,
p
= 0.01); they also had greater severity of depressive
symptoms in the BDI (20.3 ± 11.4 vs. 10.9 ± 9.3 pts,
p
= 0.003). Patients with a greater number of accom-
panying symptoms achieved significantly greater
improvement in all HFS-36 subscales after the treat-
ment with BTX-A than the patients with a small num-
ber of HFS accompanying symptoms (Table 4).
Discussion
This study revealed that the mean score of the glob-
al assessment of the quality of life with the HFS-36
questionnaire was 47.0 ± 30.9 pts out of the maximum
score of 140 points. This finding suggests the disease-
related worsening of the quality of life, but its direct
interpretation, e.g. regarding the degree of that wors-
ening, is impossible. The available literature does not
125
Neurologia i Neurochirurgia Polska 2012; 46, 2
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