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Acta Scientific Ophthalmology (ISSN: 2582-3191)
Volume 3 Issue 8 August 2020
The Role of Jankovic Spasm Grading, Orbicularis Oculi Muscle Function and Functional
Improvement Scale Pre-and Post-Treatment in Dosing Botulinum Toxin A in Treatment of
Essential Blepharospasm, Meige’s Syndrome and Hemifacial Spasm
Bastola P1* and Koirala S2
1Associate Professor, Consultant Ophthalmologist and Oculoplastic Surgeon,
Botulinum Toxin A Expert, Kathmandu, Nepal
2Head of Department, Department of Neurology, DM Neurologist, National
Academy of Medical Sciences, Kathmandu, Nepal
*Corresponding Author: Bastola P, Associate Professor, Consultant
Ophthalmologist and Oculoplastic Surgeon, Botulinum Toxin A Expert,
Kathmandu, Nepal.
Research Article
Received: June 12, 2020
Published: July 10, 2020
© All rights are reserved by Bastola P and
Koirala S.
Abstract
Background: Botulinum Toxin A (BTX A) is a proven medication used in neurological disorders like Meige’s syndrome (MS), essen-
tial blepharospasm (ES) and hemifacial spasm (HS). Jankovic spasm grading is a time trusted grading system to detect the severity
of these movement disorders pre-treatment. Orbicularis oculi muscle weakness and functional improvement ratings post treatment
Aim/Objective:
grading pre-treatment and orbicularis oculi muscle weakness and functional impairment improvement post treatment.
Methods: This was a hospital based, interventional, prospective study. All diagnosed consecutive patients of HS, ES and MS attend-
ing the neuro-Ophthalmologic/oculoplastic clinic, general outpatient department of Ophthalmology and or referred diagnosed cases
were enrolled for the study, an informed consent was taken from all the patients before the treatment for medico-legal issues. The
pre-treatment dosing was done using Jankovic spasm grading, post treatment assessment was done using orbicularis oculi muscle
assessment and functional improvement. The study subjects were followed up to 9 months or till spasms reappeared.
Results: A total of 50 study subjects were enrolled in the study, 80% of them were women. The highest number of study subjects
were in ES (50%) group followed by HS (40%) and MS (10%). Mean Jankovic spasm grading in all study subjects was 3.59 (± 0.38 Std.
deviation). The study groups ES and MS required a higher dose of BTX A (P value: < 0.0001) when compared with HS study group.
Mean orbicularis oculi muscle weakening and functional impairment improvement scale in all study subjects was 2.7 (± 0.3 Std. de-
viation) and 3.8 (± 0.6) respectively. Mean duration of action till the spasms reappeared in the study subjects was 5.1 months (± 1.7
Std. deviation). HS and ES study groups had better treatment when compared to MS.
Conclusions: Jankovic spasm grading pre-treatment and orbicularis oculi muscle weakness grading and functional improvement
score post treatment plays a very effective role to titrate the correct dose of BTX A in patients of HS, ES and MS.
Keywords: Botulinum Toxin A; Essential Blepharospasm; Hemifacial Spasm; Jankovic Spasm Grading; Meige’s Syndrome
Citation: Bastola P and Koirala S. “The Role of Jankovic Spasm Grading, Orbicularis Oculi Muscle Function and Functional Improvement Scale Pre-and
Post-Treatment in Dosing Botulinum Toxin A in Treatment of Essential Blepharospasm, Meige’s Syndrome and Hemifacial Spasm”. Acta Scientific
Ophthalmology 3.8 (2020): 06-12.
Introduction
The movement disorders of face and neck regions are mainly
essential blepharospasm (ES), hemifacial spasm (HS) and Meige’s
spasm was found in the 16th century in a painting titled De Gaper.
At that time, and for several ensuing centuries, patients with such
spasms were regarded as being mentally unstable and often were
institutionalized in insane asylums. Little progress was made in the
diagnosis or treatment of blepharospasm until the early 20th cen-
tury, when Henry Meige (pronounced “mehzh”), a French neurolo-
gist, described a patient with eyelid and midface spasms, spasm
facial median, a disorder now known as Meige’s syndrome [1].
Hemifacial spasm (HS) represents a segmental myoclonus of
Gowers in the year 1884 [2].
The cause remains obscure, treatment less than ideal, and frus-
tration is a major factor as it is with blepharospasm. Many phy-
sicians, even regarding the more common ES, may have had no
experience with MS and the patient may be told the process is psy-
chological. Movement disorders such as ES, HS and MS are treated
by different modalities, including surgery [3,4].
Botulinum toxin (BTX) is a neurotoxic protein produced by the
bacterium Clostridium botulinum and related species. It prevents
the release of the neurotransmitter acetylcholine from axon end-
-
ralysis. Infection with the bacterium causes the disease botulism.
Types A and B can cause disease in humans and are also used com-
mercially and medically [5].
Botulinum toxin is the most poisonous substance known. Intox-
ication can occur naturally because of either wound or intestinal
infection or by ingesting preformed toxin in food. The estimated
human lethal dose of type A toxin is 1.3 - 2.1 ng/kg intravenously or
intramuscularly, 10 - 13 ng/kg when inhaled, or 1000 ng/kg when
taken by mouth. Commercial forms are marketed under various
brands. Botulinum toxin is used to treat many disorders character-
ized by overactive muscle movement including spasms of the head
and neck, eyelid, vagina, limbs, jaw, and vocal cords and cosmetic
purpose, migraine, strabismus and excessive sweating etc. to men-
tion few [5-7].
Botulinum toxin A; injection is now widely accepted treatment
modality for ES, HS, and MS and is in practice for temporary relief
of symptoms in head and neck movement disorders like ES, MS and
HS [5]. BTX A known for its potency, when used in ES, HS and MS
with correct dosing can be very effective, this study with the back
ground of movement disorders of head and neck region and the
by correlating the effect of BTX A with patients with various grades
of Jankovic spasm and relief post treatment. This study also is in-
tended to provide some literature regarding correct dosing of BTX
A and various gradings to be used specially in countries; where the
existing literature is not much.
Methodology
This was a hospital based interventional; descriptive study car-
ried out over a decade in various eye hospitals and private clin-
ics of Nepal. All the consecutive patients diagnosed with essential
blepharospasm, Meige’s syndrome or hemifacial spasm at neuro-
Ophthalmology clinic, oculoplasty clinic, outpatient department
of Ophthalmology or referred cases were included in the study. All
patients underwent full clinical evaluation, including neurological
as well as ocular examinations. An informed consent was taken
from all subjects. The study followed the declaration of Helsinki.
The subjects also gave consent to take videos and photographs
during and post treatment during the study. All the subjects were
provided an information sheet about treatment with BTX A and its
potential complications.
Jankovic spasm grading [8] was used as a standard pre-treat-
ment assessment tool to calculate BTX A dose as given below:
· Grade: 0 - Normal,
· Grade 1- Slight disability, no functional impairment,
· Grade 2 - Moderate disability, no functional impairment,
· Grade 3 - Moderate disability, functional impairment and
· Grade 4 - Incapacitated.
Two observer’s observed the spasm and graded it as given
above. Cohen’s Kappa (k) value of 1 was taken as complete agree-
ment between the observers.
07
Citation: Bastola P and Koirala S. “The Role of Jankovic Spasm Grading, Orbicularis Oculi Muscle Function and Functional Improvement Scale Pre-and
Post-Treatment in Dosing Botulinum Toxin A in Treatment of Essential Blepharospasm, Meige’s Syndrome and Hemifacial Spasm”. Acta Scientific
Ophthalmology 3.8 (2020): 06-12.
The Role of Jankovic Spasm Grading, Orbicularis Oculi Muscle Function and Functional Improvement Scale Pre-and Post-Treatment in Dosing
Botulinum Toxin A in Treatment of Essential Blepharospasm, Meige’s Syndrome and Hemifacial Spasm
Botox, Allergan, Inc. was used for treatment in all cases. The
manufacturer’s instructions were carefully followed. The toxin was
used within 4 hours of its reconstitution and was kept in fridge for
cooling purpose. A vial of 100 units freeze dried BTX A was recon-
stituted with 4ml of preservative free 0.9% saline solution to yield
toxin in a concentration of 2.5 units per 0.1 ml. The toxin was in-
jected subcutaneously into the selected sites using a 1ml tuberculin
syringe with a 27 - 291/2-gauge needle. Atropine, adrenaline injec-
tions were kept standby during the treatment process with antial-
lergics to combat any complications. The initial dose was calculated
based on clinical judgement of the spasm and after observing the
subjects video for few minutes. Five to seven periocular injections
in ES/MS were selected with each site getting 2.5 - 7.5 units of BTX
A depending upon the severity of spasm of the affected muscle. In
only in one side of the face. The cheek was injected in cases hav-
ing a severe degree of retraction of angle of mouth. Patients were
evaluated at two weeks, six weeks and six months and 9 months’
time or when the spasm reappeared. The subjects were advised to
maintain a diary to note the onset of clinical response, peak effect,
duration of clinical improvement and complications on follow up
assessment. Telephone contact with the patients was encouraged
for follow up assessment and through designated social media.
Post treatment the patients were evaluated for the following after
detailed ocular examination.
Assessment of functional improvement [7,8] was done follow-
ing a grading system given below:
· 0 - no effect,
· 1 - Mild effect but no functional improvement
· 2 - Moderate improvement but no change in functional
disability
· 3 - Moderate change in both severity and function
· 4 - Marked improvement in severity and function in a
similar way.
Orbicularis oculi muscle weakening [9] grading tool was done
by assessing the closure of the eyes:
· Grade 0: Incomplete eyelid closure,
· Grade 1: Lids just closing, minimal resistance to overcome
· Grade 2: Closing well, some resistance, easily overcome
·
· Grade 4: Very strong closure, cannot be overcome or
over
Assessment of functional impairment rating and grading of or-
bicularis oculi muscle weakening post injections was done by two
different observers. Cohen’s Kappa (k) value of 1 was taken as com-
plete agreement between the observers.
Grade 0, 1, 2 were considered as good outcome post treatment
with BTX A.
Complications were looked for and noted specially blepharop-
tosis, deviation of angle of mouth, bruises and ecchymosis. Patients
were followed up at six weeks and six months and nine months
and similar examinations were carried out as mentioned in the
two weeks visit (disappearance of complications if present were
also noted). Patients were told to note the date and follow up im-
proforma was maintained to record all the regarding patient, treat-
ment and follow up visits. Useful data was entered in the database
for statistical analysis SPSS 20, Stata were used for statistical analy-
sis. A statistician was consulted when and where felt necessary.
Results
Fifty subjects were enrolled for the study, 50% (25 subjects)
were diagnosed with ES, followed by 40% (20 subjects) with HS
and 10% with MS. 80% (40) of them were females and the 20% (10
subjects) males. Mean age of the female and male subjects was 52
(± 10, range 29 - 73) and 54 (± 9.8, range: 45 - 70) years. Dry eyes,
stress, watching television, talking, lack of sleep were prominent
exacerbating factors. Pre-treatment Jankovic spasm grading for
calculation of dose of BTX A in different sub groups and the impact
of BTX A in the study group with regard to orbicularis oculi muscle
function, functional impairment improvement scale and reappear-
ance of the spasms have been described in table 1 and 2.
08
Citation: Bastola P and Koirala S. “The Role of Jankovic Spasm Grading, Orbicularis Oculi Muscle Function and Functional Improvement Scale Pre-and
Post-Treatment in Dosing Botulinum Toxin A in Treatment of Essential Blepharospasm, Meige’s Syndrome and Hemifacial Spasm”. Acta Scientific
Ophthalmology 3.8 (2020): 06-12.
The Role of Jankovic Spasm Grading, Orbicularis Oculi Muscle Function and Functional Improvement Scale Pre-and Post-Treatment in Dosing
Botulinum Toxin A in Treatment of Essential Blepharospasm, Meige’s Syndrome and Hemifacial Spasm
Jankovic spasm (JS) grading in study subjects pre-treatment
Diagnosis Number of subjects JS grading (Minimum) JS grading (Maximum) Mean (SD**)
ES‡ 25 3 4 3.6 (± 0.5)
MS† 5 - 4 4
HS* 20 2 4 3.18 (± 0.66)
Total 50 - - 3.59 (± 0.38)
Botulinum toxin A (BTX A) treatment and dose in units for each study sub groups
BTX A Dose Diagnosed sub groups
ES MS HS Total (%) P value
20
< 0.0001
23 3 (15%) 3 (6%)
25 12 (60%) 12 (24%)
30 5 (25%) 5 (10%)
35 1 (4%) 1 (2%)
40
45 5 (20%) 5 (10%)
50 10 (40%) 10 (20%)
60 9 (36%) 5 (100%) 9 (18%)
Total 25 (100%) 5 (100%) 20 (100%) 50 (100%)
Table 1: Pre-treatment Jankovic spasm grading and calculation of dose of botulinum toxin A based on the grading.
*Hemifacial spasm, † Meige’s syndrome, ‡ Essential blepharospasm, **Standard deviation.
Table 1 showing that the mean JS grading was higher for ES and MS and BTX A dose was also given higher for the same study groups.
of BTX A when compared to HS.
Orbicularis oculi muscle weakening grading in study sub groups in two weeks follow up
Diagnosis Number of subjects Grading (Minimum) Grading (Maximum) Mean (SD*)
ES 25 1 3 2 (± 0.2)
MS 5 1 2 1.5 (± 0.3)
HS 20 1 3 1.5 (± 0.3)
Total 50 1 3 2.7 (± 0.3)
Diagnosis Number of subjects Grading (Minimum) Grading (Maximum) Mean (SD*)
ES 25 2 4 3.5 (± 0.7)
MS 5 2 4 3.2 (± 1.0)
HS 20 3 4 3.9 (± 0.1)
Total 50 2 3.8 3.8 (± 0.6)
The noted time of spasm reappearance by the study subjects in different treatment sub-groups
Diagnosis Study subjects Minimum (Months) Maximum (Months) Mean (SD*)
ES 25 2.5 6.5 5 (± 1.4)
MS 5 2.5 6.5 4.5 (± 2.4)
HFS 20 3.5 8.5 6.0 (± 1.3)
Total 50 2.5 8.5 5.1 (± 1.7)
Table 2: Orbicularis oculi muscle weakening post treatment with BTX A, functional impairment improvement scale and reappearance of
spasms in the study sub groups.
*Standard deviation.
Table 2 showing the study sub groups ES and HS having comparatively better outcome with the treatment than MS.
09
Citation: Bastola P and Koirala S. “The Role of Jankovic Spasm Grading, Orbicularis Oculi Muscle Function and Functional Improvement Scale Pre-and
Post-Treatment in Dosing Botulinum Toxin A in Treatment of Essential Blepharospasm, Meige’s Syndrome and Hemifacial Spasm”. Acta Scientific
Ophthalmology 3.8 (2020): 06-12.
The Role of Jankovic Spasm Grading, Orbicularis Oculi Muscle Function and Functional Improvement Scale Pre-and Post-Treatment in Dosing
Botulinum Toxin A in Treatment of Essential Blepharospasm, Meige’s Syndrome and Hemifacial Spasm
Discussion
Amongst 50 subjects enrolled in the study the study had 80%
of women gender wise, the mean age of study subjects in the study
was 52 and 54 years respectively for women and men. Common
exacerbating factors in study subjects were dry eyes, stress, lack of
-
ing knowledge about essential blepharospasm, hemifacial spasm
and Meige’s syndrome and other studies done elsewhere [8-10].
Current study had 50% cases of essential blepharospasm fol-
lowed very closely by 40% cases of HS, which did not correlate with
some other reported prevalence of ES, HS and MS. The reported
prevalence for HS is highest followed by ES and MS [8,10,11] this
may be contributed to the small number of study population and
various hospitals for treatment and also probably contributed by
the fact that subjects who had severe symptoms or were unable to
carry out their day to day work attended the clinic more.
Mean Jankovic spasm grading in the study subjects was 3.6 (±
0.5), 4 (± 0) and 3.18 (± 0.66) in ES, MS and HS respectively, while
the mean combined of all subjects was 3.59 (± 0.66), which high-
lighted the fact that study subjects came for treatment when their
movement disorder and spasms were severe enough to disturb
their daily routine work and in MS group using Jankovic spasm
grading all cases were incapacitated. This was very important in
dosing BTX A for treatment of these subjects. Study subjects hav-
ing high Jankovic spasm grading (Table 1) were given more BTX A,
whereas subjects with lower Jankovic spasm grading were given
comparatively lower dose of BTX A injection and in lesser sites
(Table 1).
In the current study both pre-tarsal as well as pre-septal injec-
tions of BTX A was given, and the treatment outcome was better
-
where; where the results of injecting BTX A also in pretarsal por-
tion of orbicularis oculi muscle was more effective [12,13].
The study subjects in ES and MS required more dosage of BTX
A than HS, this can be explained by the following; the study sub-
jects in the former groups had higher mean Jankovic spasm grading
(Table 1) and also due to the fact that hemifacial spasms subjects
with studies done in the past [2,8].
In our study, the mean (Standard deviation) orbicularis oculi
± 0.2), 1.5 (±
0.3) and 1.5 (0.3) respectively, with an overall mean of 2.7 (± 0.3)
for all the study subjects. This was an excellent result and showed
that the dosing for BTX A was correct and correlated very well with
Jankovic spasm grading pre-treatment (Table 1 and 2).
Similarly, in the current study the mean functional impairment
improvement in ES, MS and HS study sub groups was 3.5 (± 0.7),
3.2 (± 1.0) and 3.9 (± 0.6) with an overall mean of 3.8 (± 0.6). This
Jankovic spasms and calculate the dose (Table 1 and 2). An inter-
lesser than ES and HS it is primarily due to the fact that the study
-
pacitated when graded and hence were given maximum possible
best outcome with both orbicularis oculi weakening and functional
improvement rating, which correlated with studies done in the
past [4,7,8,14].
The mean reappearance of spasm in months in all study subjects
in the current study was 5.1 months (± 1.7). However, HS study sub
group had an excellent temporary relief with the treatment lasting
up to 8.5 months, closely followed by essential blepharospasm, this
study sub group had a mean of 5 months (± 1.4) and many study
participants reported the spasms after 8 months. MS study group
reappearance correlated well with orbicularis oculi weakening
grading and functional improvement rating, a smaller sample size
and initial cautious approach could be the reasons for temporary
relief lasting up to 2.5 - 6.5 months (Table 2).
that initial Jankovic spasm grading is very important to get the
-
where [4,7,8].
Minor complications like deviation of angle of mouth, transient
ptosis and bruise or ecchymosis were seen in three study subjects
studies done in the past [15-17].
10
Citation: Bastola P and Koirala S. “The Role of Jankovic Spasm Grading, Orbicularis Oculi Muscle Function and Functional Improvement Scale Pre-and
Post-Treatment in Dosing Botulinum Toxin A in Treatment of Essential Blepharospasm, Meige’s Syndrome and Hemifacial Spasm”. Acta Scientific
Ophthalmology 3.8 (2020): 06-12.
The Role of Jankovic Spasm Grading, Orbicularis Oculi Muscle Function and Functional Improvement Scale Pre-and Post-Treatment in Dosing
Botulinum Toxin A in Treatment of Essential Blepharospasm, Meige’s Syndrome and Hemifacial Spasm
Recommendations
Jankovic spasm grading plays a very important role in dosing
botulinum toxin A to treat movement disorders of head and neck
hemifacial spasm. However, we recommend that post treatment as-
sessment of orbicularis oculi muscle and functional improvement
must assessed to know whether the given dose of BTX A was near
correct low or high, so as to verify and give correct dose when the
subject follows up with spasms again.
Limitations
The prevalence of movement disorders is very low so the study
larger study done in titrating the dose of botulinum toxin A.
Conclusion
Botulinum toxin A is a very safe, effective temporary medical
treatment modality to treat movement disorders like essential
blepharospasm, Meige’s syndrome and hemifacial spasm. Botu-
linum toxin A when administered with correct dosing following
the grading systems used in the study can provide a temporary re-
lief up to eight months or more. The results of treatment are very
promising with near correct dose, we recommend it for a wider use
in other parts of Nepal and world.
Acknowledgements
We would like to thank all the study subjects who uncondition-
ally accepted to be part of the research and did not leave the study
in between. We would also like to thank Prof. JP Agrawal, Prof. DN
Shah who initiated idea of using botulinum toxin A in Nepal and
supported the researcher to register it in Nepal for therapeutic use.
We would like to thank Mrs. Sheela Koirala Bastola for keeping all
the study subjects records intact and also for entering the data in
SPSS and Stata.
None.
Source of Funding
None.
Bibliography
11
The Role of Jankovic Spasm Grading, Orbicularis Oculi Muscle Function and Functional Improvement Scale Pre-and Post-Treatment in Dosing
Botulinum Toxin A in Treatment of Essential Blepharospasm, Meige’s Syndrome and Hemifacial Spasm
Citation: Bastola P and Koirala S. “The Role of Jankovic Spasm Grading, Orbicularis Oculi Muscle Function and Functional Improvement Scale Pre-and
Post-Treatment in Dosing Botulinum Toxin A in Treatment of Essential Blepharospasm, Meige’s Syndrome and Hemifacial Spasm”. Acta Scientific
Ophthalmology 3.8 (2020): 06-12.
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The Role of Jankovic Spasm Grading, Orbicularis Oculi Muscle Function and Functional Improvement Scale Pre-and Post-Treatment in Dosing
Botulinum Toxin A in Treatment of Essential Blepharospasm, Meige’s Syndrome and Hemifacial Spasm
Citation: Bastola P and Koirala S. “The Role of Jankovic Spasm Grading, Orbicularis Oculi Muscle Function and Functional Improvement Scale Pre-and
Post-Treatment in Dosing Botulinum Toxin A in Treatment of Essential Blepharospasm, Meige’s Syndrome and Hemifacial Spasm”. Acta Scientific
Ophthalmology 3.8 (2020): 06-12.
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