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Impact on Quality of Life of Botulinum Toxin Treatments for Spasmodic Dysphonia and Oromandibular Dystonia
Neil Bhattacharyya, MD;
Daniel Tarsy, MD
Arch Otolaryngol Head Neck Surg. 2001;127:389-392.
ABSTRACT
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Objective To determine the impact on quality of life of botulinum toxin treatments
for common dystonias of the head and neck.
Design Cross-sectional survey study of a patient cohort treated with botulinum
toxin injections for spasmodic dysphonia (SD) or oromandibular dystonia (OMD).
Interventions and Outcome Measures The Glasgow Benefit Inventory was used to quantify the health benefit
of treatment. Data were collected for demographics, time intervals relative
to diagnosis, treatment duration, and frequency of injections. The groups
were compared to determine whether differences existed in benefit from treatment.
Correlation analysis was conducted for inventory scores and time intervals.
Results A total of 23 patients (5 with OMD and 18 with SD) completed the questionnaire.
The mean total benefit score was +38.04 (possible range, -100 to +100)
for the whole group (P<.001). The OMD group derived
a nonsignificantly smaller benefit (+21.67 vs +42.59) (P = .07). The mean subscores for the combined group were +39.67, +26.81,
and +42.75 for the general, social support, and physical health subscores,
respectively (P .001). The difference in mean
subscores between the 2 groups was not statistically significant, although
patients with OMD had a lower social support subscore (+6.67 vs +32.41). No
correlation was found between duration of therapy or frequency of injections
and the Glasgow Benefit Inventory score.
Conclusions Patients with OMD or SD derive considerable benefit when treated with
botulinum toxin. The magnitude of benefit is largely independent of the time
course of therapy. Treatment with botulinum toxin for these conditions is
effective on the basis of quality-of-life criteria.
INTRODUCTION
SPASMODIC dysphonia (SD) and oromandibular dystonia (OMD) constitute
a group of dystonias relatively frequently encountered by otolaryngologists.
The diagnosis is often made by an otolaryngologist or neurologist after an
exhaustive search by the patient. Treatment for these conditions has centered
on the use of intramuscular injections of botulinum toxin.1, 2
Unfortunately, these dystonias rarely remit spontaneously and, thus, usually
require long-term therapy. As the effect of botulinum toxin is temporary,
the patient must submit to periodic injections, the frequency of which ranges
from several weeks to several months.
Because many patients endure a marked social stigma due to communication
difficulties or disfiguring involuntary movements, a diagnosis of SD or OMD
carries with it a significant impact on quality of life.3
Several studies have examined quality-of-life impact of botulinum toxin treatments
for cervical dystonia and other dystonic conditions, but relatively little
information is available on the impact of such treatment in SD or OMD.4, 5 Furthermore, the recurring nature of
these treatments, as well as the cost of botulinum toxin, makes treatment
for these conditions expensive on a case-by-case basis. Therefore, measures
of quality-of-life impact may aid in counseling patients who undergo botulinum
toxin treatments, and may also provide evidence justifying this treatment
modality in the face of an ever-shrinking health care dollar.
PATIENTS AND METHODS
This study was approved by our medical center's Committee on Clinical
Investigations. The Glasgow Benefit Inventory (GBI) is a well-studied and
validated measure of patient benefit developed especially for otolaryngologic
interventions.6 This survey was adapted to
examine the quality-of-life impact of repeated botulinum toxin injections
for patients with an established diagnosis of SD or OMD. The questionnaire
was then administered to a group of 31 patients currently being treated for
either SD or OMD with botulinum toxin in a multidisciplinary clinic for movement
disorders. Informed consent was obtained from each individual participating
in the study.
The data were tabulated and entered into a statistical spreadsheet for
analysis. Additional demographic information was gathered from the medical
record and confirmed by patient responses on the survey. These data included
current age, sex, and dystonia diagnosis. The time interval between onset
of dystonic symptoms and diagnosis, average time interval between injections,
and duration of therapy with botulinum toxin were also recorded. The data
gathered from the GBI were scaled in standard fashion to range from -100
(maximal negative benefit) to +100 (maximal positive benefit).6
Statistical analysis was conducted with the SPSS statistical package
(SPSS Inc, Chicago, Ill). Descriptive statistics were computed for the total
score, the general subscore, the social support subscore, and the physical
health subscore for the GBI elements. The t test
for a population was used to test the hypothesis that the GBI scores would
differ from 0 (as a score of 0 implies no positive or negative benefit). The
data were further examined to determine whether differences in GBI score existed
between patients with a diagnosis of SD and OMD. Finally, the data were examined
to determine whether any correlation existed between patient benefit as measured
by the GBI and time interval between diagnosis and therapy, injection frequency,
or duration of therapy.
RESULTS
Of 31 patients (23 with SD and 8 with OMD), 23 returned a completed
survey (response rate, 74%). There were 5 patients with OMD and 18 with SD.
The cohort was composed of 17 women and 6 men, with an average age of 55.7
years (range, 26-81 years). The average time interval between the onset of
dystonic symptoms and diagnosis was 109.8 months (range, 1 week to 49.7 years).
The mean treatment duration was 59.5 months (range, 6-168 months), and the
mean time interval between botulinum toxin injections (injection frequency)
was 17.8 weeks (standard deviation, 7.3 weeks).
The calculated data for the total score and subscores on the GBI for
the entire group and each diagnosis subgroup are displayed in Table 1. The combined group derived considerable benefit from recurrent
therapy with botulinum toxin for their dystonia, as measured by total score
and subscores. Each of these scores demonstrated a statistically significant
patient benefit from the injections (Table
1, t test). Combined-group patients reported
the least benefit from botulinum toxin injections with respect to their social
support subscore (+26.81).
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Table 1. Scores on the Glasgow Benefit Inventory*
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The patients with OMD reported lower derived benefit from the botulinum
toxin injections than the SD group. However, these differences were not significant
(P>.05, Mann-Whitney statistic for differences in
means between groups). Notably, patients with OMD reported a lower social
support subscore benefit than patients with SD (+6.67 vs +32.41, respectively; P = .23). All scores were in the positive range, indicating
that patients derived no negative benefit from the injections.
Data from the correlation analysis are presented in Table 2. No statistically significant correlation was identified
between total GBI score and time interval between symptom onset and diagnosis,
duration of therapy, or frequency of injections. Similarly, no correlation
was identified between these time intervals and any of the subscores.
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Table 2. Correlation Between Time Intervals and Scores on the Glasgow
Benefit Inventory
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COMMENT
The GBI is an outcomes research tool specifically developed to measure
patient benefit from otolaryngologic interventions. As a well-studied and
validated research tool, it has been found to be sensitive to changes in quality
of life and benefit derived from otolaryngologic interventions.6
It has previously been used to study degree of patient benefit derived from
rhinoplasty, acoustic neuroma surgery, and other otolaryngologic interventions,
with good success.7, 8 The standard
GBI was modified according to existing recommendations for use in quantifying
patient benefit from the intervention of botulinum toxin injections for OMD
or SD.9 The orientation of the questions was
kept identical to the basic GBI, so as to prevent response bias. In addition
to a total score, the GBI also includes subscales related to general health
and well-being (general subscale), social support, and physical health. Each
of the subscales measures a change in health status possibly produced by the
intervention under consideration. As a postintervention questionnaire, it
is maximally sensitive to the change in health status produced by the intervention.6 This questionnaire method was chosen as our outcomes
tool because it is simple, it is not overly burdensome for the patient to
complete, and it can be applied to patients who have already initiated their
therapy. This is especially important because the standard before-and-after
design for an outcomes measure would be very difficult to apply to patients
who undergo repeated or recurrent forms of therapy, such as botulinum toxin
injections. We believe our high response rate at least partially reflects
the ease of completion of the GBI outcomes tool.
Two specific questions contained in the GBI merit some comment. Question
16 (Since your "intervention," have you been to a doctor more or less often?)
and question 18 (Since your "intervention," have you taken more or less medicine?)
may contribute to lower benefit scores for this group of patients. Generally,
with a single intervention, such as a surgical procedure, success after that
procedure would be characterized by fewer physician visits and the requirement
for less medicine. However, patients receiving botulinum toxin therapy require
repeated visits for injections, as well as medicine in the form of the botulinum
toxin. Therefore, benefit scores on these 2 questions will tend to be low,
as evidenced by our data, with mean patient benefit scores of only +13.0 and
+17.0, respectively.
The diagnosis and treatment of OMD and SD were brought to the attention
of the otolaryngology and neurology communities largely because of the efforts
of Blitzer et al1, 10 and Ludlow
et al.11, 12 Until the late 1980s,
the adductor and abductor forms of SD (laryngeal dystonia) were often attributed
to psychogenic causes. Once the efficacy of botulinum toxin was reported and
accepted, these diagnoses became better recognized as manifestations of organic
dystonia. Before the establishment of botulinum toxin as an effective therapeutic
modality, these patients had limited options for long-term control of their
dysphonia or dystonia. It was not uncommon for patients to go many years with
an inaccurate diagnosis and without opportunity for adequate treatment. Our
data highlight this, with an average interval from onset of symptoms to final
diagnosis of more than 9 years. With growing awareness of these dystonias
among otolaryngologists and neurologists, and the emergence of botulinum toxin
as a safe and effective treatment modality, it is likely that more patients
will be properly diagnosed and treated.
Proper diagnosis and treatment of these conditions is essential because
both OMD and SD may have a substantial impact on quality of life. Patients
with OMD often have distracting or disfiguring involuntary jaw and perioral
facial movements, which may affect mastication and speech. Patients with adductor
SD usually have a "strained-strangled" pattern of voicing that decreases vocal
projection and may limit intelligibility of speech. Patients with abductor
SD may be perceived as extremely nervous and difficult to understand because
of their involuntary voice breaks. Several of our patients have been unable
to secure employment because employers perceived their speech or movement
patterns as abnormal and even potentially offensive to prospective customers.
Several studies have documented an association between SD and psychological
dysfunction. Patients with SD exhibit significantly elevated levels of depression
and anxiety, which decrease with botulinum toxin therapy.3, 13
In a controlled study, psychological and emotional symptoms and an overall
poor quality of life in patients with SD were found to be secondary to, rather
than the cause of, the voice disorder.14 Although
these studies examined patients with SD, one would expect similar findings
for patients with OMD.
Fortunately, botulinum toxin treatment of these dystonias has been successful.
Several studies have documented high treatment efficacy as well as a low incidence
of side effects in the treatment of OMD and SD. In a 12-year review, Blitzer
and associates2 reported an average benefit
of 90% for patients with adductor SD, lasting an average of 15.1 weeks. Benefit
was measured by means of the universal SD rating scale. A lower average benefit
of 66.7%, lasting an average of 10.5 weeks, was noted for patients with abductor
SD. Other studies have documented similar success rates with respect to voice
outcomes both qualitatively and quantitatively.15
Our data confirm that patients with OMD and SD derive meaningful and
substantial subjective benefit from therapy with botulinum toxin. This holds
true for the social support and physical health domains as well. Our finding
that the SD group showed a tendency toward greater benefit in total score
and in each of the subscores may result from the fact that patients with SD
have a more dramatic communication difficulty and perceive a greater benefit
when it improves. Patients with OMD usually experience pain and discomfort,
with less impact on social interaction, and therefore may derive less benefit
on the social support subscore. Notably, however, none of the treatment groups
demonstrated a negative benefit score on any element or subscale (Table 1).
We found no correlation between total GBI score or subscores and time
interval from symptom onset to diagnosis, duration of therapy, or interval
between injections. This suggests that therapeutic benefit is independent
of duration of symptoms before treatment, treatment duration, or frequency
of injections. Similarly, patient benefit is maintained even years into treatment.
Treatment with botulinum toxin for OMD and SD is clearly justified on the
basis of patient benefit criteria.
AUTHOR INFORMATION
Accepted for publication September 5, 2001.
This study was funded in part by a grant from the Brigham Surgical Group
Foundation, Boston, Mass.
From the Division of Otolaryngology, Brigham and Women's Hospital (Dr
Bhattacharyya), and Department of Neurology, Beth Israel Deaconess Medical
Center (Dr Tarsy), Harvard Medical School, Boston, Mass.
Corresponding author and reprints: Neil Bhattacharyya, MD, Division
of Otolaryngology, 333 Longwood Ave, Boston, MA 02115.
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