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PubMed1995

The blepharospasm disability scale: an instrument for the assessment of functional health in blepharospasm.

Assessment of the functional status in patients with blepharospasm is of major importance for clinical practice and outcome studies. The Blepharospasm Disability Scale (BDS) is specifically directed to measure the disability in these patients. The metric properties of this instrument were evaluated. Reliability, validity, and responsiveness to within-patient health changes over time of the BDS were assessed in 40 patients with essential blepharospasm treated with botulinum toxin injections. The reliability of the scale was sufficient for use on group level (Cronbach's alpha coefficient, 0.69). Evidence of discriminant validity was provided by the difference in median score on the BDS between 21 newly admitted patients and 19 patients already under treatment (p < 0.001). Convergent validity was supported by correlations between BDS and neurological impairment scores (range, Spearman correlation coefficients, 0.65-0.79). Responsiveness to health changes was demonstrated by a significant difference between median BDS scores before treatment and 2 weeks after treatment with botulinum toxin (p < 0.01). The BDS is a useful disease-specific instrument to assess disability. Completion of the questionnaire is easy and takes only a few minutes. The instrument is suitable for use in patient care, descriptive outcome studies, and should be considered in controlled clinical trials.

Activities of Daily Living↗
PubMed1999

Risk factors for spread of primary adult onset blepharospasm: a multicentre investigation of the Italian movement disorders study group.

OBJECTIVES: Little is known about factors influencing the spread of blepharospasm to other body parts. An investigation was carried out to deterrmine whether demographic features (sex, age at blepharospasm onset), putative risk, or protective factors for blepharospasm (family history of dystonia or tremor, previous head or face trauma with loss of consciousness, ocular diseases, and cigarette smoking), age related diseases (diabetes, hypertension), edentulousness, and neck or trunk trauma preceding the onset of blepharospasm could distinguish patients with blepharospasm who had spread of dystonia from those who did not. METHODS: 159 outpatients presenting initially with blepharospasm were selected in 16 Italian Institutions. There were 104 patients with focal blepharospasm (mean duration of disease 5.3 (SD 1.9) years) and 55 patients in whom segmental or multifocal dystonia developed (mainly in the cranial cervical area) 1.5 (1.2) years after the onset of blepharospasm. Information was obtained from a standardised questionnaire administered by medical interviewers. A Cox regression model was used to examine the relation between the investigated variables and spread. RESULTS: Previous head or face trauma with loss of consciousness, age at the onset of blepharospasm, and female sex were independently associated with an increased risk of spread. A significant association was not found between spread of dystonia and previous ocular diseases, hypertension, diabetes, neck or trunk trauma, edentulousness, cigarette smoking, and family history of dystonia or tremor. An unsatisfactory study power negatively influenced the validity and accuracy of the negative findings relative to diabetes, neck or trunk trauma, and cigarette smoking. CONCLUSIONS: The results of this exploratory study confirm that patients presenting initially with blepharospasm are most likely to experience some spread of dystonia within a few years of the onset of blepharospasm and suggest that head or face trauma with loss of consciousness preceding the onset, age at onset, and female sex may be relevant to spread. The suggested association between edentulousness and cranial cervical dystonia may be apparent because of the confounding effect of both age at onset and head or face trauma with loss of consciousness. The lack of influence of family history of dystonia on spread is consistent with previous findings indicating that the inheritance pattern is the same for focal and segmental blepharospasm.

Age of Onset↗
PubMed2005

Modulation of sensory photophobia in essential blepharospasm with chromatic lenses.

PURPOSE: To determine the effect of photochromatic modulation with tinted lenses on the sensory symptoms of photophobia in blepharospasm patients. DESIGN: Nonrandomized case-control study. PARTICIPANTS: Thirty-four subjects (24 benign essential blepharospasm patients and 10 normal controls). METHODS: Subjects were seated in front of a calibrated light source. Beginning at 0%, the intensity of the light source was increased gradually until the patient reported symptomatic photophobia. The intensity of the light source was then measured with a light meter. This procedure was performed first with no chromatic lens and then with 7 different chromatic lenses, each blocking specific wavelengths of the visible spectrum. The subject was then asked which lens provided the greatest symptomatic improvement of photophobia. Statistical significance was calculated with analysis of variance and t test analysis. MAIN OUTCOME MEASURES: Objective measurement of light intensity tolerated and subjective assessment of photophobia were obtained for each chromatic lens tested. RESULTS: The light intensity tolerated by the normal subjects compared with the blepharospasm group was not statistically significant with no lens, but grew to 3.5 times that tolerated by the blepharospasm group as more of the higher wavelengths of the visible spectrum were blocked (P = 0.048). Lenses 4, 5, 6, and 7 allowed blepharospasm patients to tolerate a significantly higher intensity of light when compared with no lens (P = 0.04, P = 0.007, P = 0.03, and P = 0.01, respectively). Although the highest intensity of light tolerated was measured with lens 6, 71% of blepharospasm patients reported the greatest relief of photophobia with lens 7. CONCLUSIONS: Blepharospasm patients tolerate a lower intensity of light when compared with normal subjects; this differential in light tolerance becomes significantly more pronounced as the higher wavelengths of the visible spectrum are blocked. The symptoms of photophobia in blepharospasm patients can be reduced significantly with photochromatic modulation. Despite lens 6 allowing the patients to tolerate a higher intensity of light, the majority of patients preferred lens 7 for symptomatic relief of photophobia. These findings suggest that sensory photophobia may be related more to the wavelength than to the intensity of the light exposure.

Blepharospasm↗
PubMed2006

Bilateral grey-matter increase in the putamen in primary blepharospasm.

BACKGROUND: Primary blepharospasm is a focal dystonia characterised by excessive involuntary closure of the eyelids. The pathophysiology of primary blepharospasm is unresolved. AIM: To pinpoint grey-matter changes that are associated with primary blepharospasm. METHODS: 16 right-handed patients with primary blepharospasm (mean age 67.4 (SD 4.3) years; 12 women) were compared with 16 healthy volunteers matched for sex and age. High-resolution T1-weighted magnetic resonance imaging of each participant was obtained and analysed by voxel-based morphometry, a method to detect regionally specific differences in grey matter between patients and control group. To evaluate whether the identified grey-matter changes were correlated with the duration of primary blepharospasm or botulinum neurotoxin treatment (BoNT), separate regression analyses were carried out. RESULTS: In patients with primary blepharospasm, grey-matter increase in the putamina was observed, whereas regression analyses did not indicate a correlation between grey-matter increases and the duration of primary blepharospasm or BoNT. Grey-matter decrease was detected in the left inferior parietal lobule; here regression analyses of grey-matter decrease showed a significant (p = 0.013) correlation of grey-matter decrease with the duration of BoNT. CONCLUSIONS: The data suggest structural changes in primary blepharospasm and point to a crucial role of the putamen for the pathophysiology of this focal dystonia.

Aged↗
PubMedDate not supplied

[Costs and efficacy of type A botulinum toxin for the treatment of essential blepharospasm and hemifacial spasm].

PURPOSE: To evaluate the costs and efficacy of type A botulinum toxin in the treatment of essential blepharospasm and hemifacial spasm. METHODS: Pacients with essential blepharospasm and hemifacial spasm had their files analyzed. All patients were treated with type A botulinum toxin (Dysport) between April 2002 and May 2004 at the Oculoplastic Clinics of "Santa Casa de São Paulo". Twenty-seven patients presented essential blepharospasm and 23 presented hemifacial spasm. Information about the patient's degree of satisfaction after treatment, complaints and personal costs were recorded by a questionnaire, and information about the costs of Dysport treatment were obtained at the administration department of "Santa Casa de São Paulo". Wilcoxon and Mann-Whitney tests were used for statistical analysis. RESULTS: 1- The annual treatment costs were R Dollars 1,239.32 for essential blepharospasm and R Dollars 661.72 for hemifacial spasm. 2- The patient's annual costs were R Dollars 145.48 for essential blepharospasm and R Dollars 6.07 for hemifacial spasm. 3- The hospital's annual costs for the treatment were R Dollars 1,095.84 for essential blepharospasm and R Dollars 535.65 for hemifacial spasm. 4- Dysport treatment is successful in both essential blepharospasm and hemifacial spasm. CONCLUSIONS: The costs of essential blepharospasm and hemifacial spasm treatment with Dysport are high, mainly because of the toxin price. On economic analysis of health, we can conclude that this procedure has an excellent cost-benefit ratio.

Aged↗
PubMed2005

Botulinum toxin type A therapy for blepharospasm.

BACKGROUND: Blepharospasm is a focal dystonia characterized by chronic intermittent or persistent involuntary eyelid closure due to spasmodic contractions of the orbicularis oculi muscles. Other facial and neck muscles are also frequently involved. Most cases are idiopathic and blepharospasm is generally a life-long disorder. Its severity can range from repeated frequent blinking to persistent forceful closure of the eyelids with functional blindness. Botulinum toxin type A (BtA) is the current first line therapy. OBJECTIVES: To determine whether botulinum toxin (BtA) is an effective and safe treatment for blepharospasm. SEARCH STRATEGY: We identified studies for inclusion in the review using the Cochrane Movement Disorders Group trials register, the Cochrane Central Register of Controlled Trials (CENTRAL), MEDLINE, EMBASE, handsearches of the Movement Disorders Journal and abstracts of international congresses on movement disorders and botulinum toxin, communication with other researchers in the field, reference lists of papers found using above search strategies, and contact with authors and drug manufacturers. SELECTION CRITERIA: Studies were eligible for inclusion in the review if they evaluated the efficacy of BtA for the treatment of blepharospasm. They must have been randomised and placebo-controlled. DATA COLLECTION AND ANALYSIS: We used a paper pro-forma to collect data from the included studies using double extraction by two independent reviewers. The two reviewers separately assessed each trial for internal validity and they settled differences between them by discussion. The outcome measures used included adverse events, improvement in symptomatic rating scales, subjective evaluation by patients and clinicians, and changes in quality of life assessments. MAIN RESULTS: We found few controlled trials. They were of short duration and enrolled small numbers of patients. Because of their poor internal validity, the characteristics of the populations studied, and the types of interventions and outcomes, none of the trials fitted our criteria for inclusion. However, all these trials found BtA to be superior to placebo as did large case-control and cohort studies, which reported that around 90% of patients benefited. AUTHORS' CONCLUSIONS: There are no high quality, randomised, controlled efficacy data to support the use of Bt for blepharospasm. Despite this, other studies suggest that BtA is highly effective and safe for treating blepharospasm and support its use. The effect size (90% of patients benefit) seen in open studies makes it very difficult and probably unethical to perform new placebo-controlled trials of efficacy of BtA for blepharospasm. Future trials should explore technical factors such as the optimum treatment intervals, different injection techniques, doses, Bt types and formulations. Other issues include service delivery, quality of life, long-term efficacy, safety, and immunogenicity.

Blepharospasm↗
PubMed1999

Idiopathic blepharospasm does not lead to a parkinsonian syndrome: results of a questionnaire-based follow-up study.

It has been suggested that a lesion in the dopaminergic neurons of the substantia nigra pars compacta combined with eye irritation is involved in the pathophysiology of idiopathic blepharospasm. If so, these patients might be prone to develop Parkinson's disease or a parkinsonian syndrome. We therefore conducted a validated questionnaire-based follow-up study to estimate (a) the frequency of local eye disorders at onset and (b) frequency of development of parkinsonian symptoms in blepharospasm patients. Ninety-nine patients previously diagnosed with idiopathic blepharospasm were sent a two-part questionnaire to assess parkinsonian and other symptoms associated with their condition. The average period of follow-up was 12.7 years, ranging from 3 to 26 years, with an average age at onset of 53.5 years. Sixty-two patients reported other ocular symptoms prior to or at the onset of blepharospasm, and therefore ocular disease may act as a trigger to produce blepharospasm in those already predisposed. Only two patients had developed a score on the parkinsonism rating scale indicating possible Parkinson's disease, but clinical examination confirmed this not to be the case. If a lesion in the dopaminergic neurons is involved in blepharospasm, it would appear to be relatively minor (and non-progressive), since patients with idiopathic blepharospasm do not seem prone to develop parkinsonian symptoms.

Aged↗
PubMed1997

Maladaptive neural compensatory mechanisms in Bell's palsy-induced blepharospasm.

We described four patients with Bell's palsy and blepharospasm and evaluated potential mechanisms that may be responsible for an apparent association between the two disorders. Eyelid movements in spontaneous blinks were studied by the search coil technique in patients with this novel disorder. Kinematic analyses documented bilateral eyelid spasm subsequent to unilateral Bell's palsy. The temporal interval between the onset of palsy and onset of blepharospasm was highly variable (weeks to > 20 years). Changes in the relationship between spontaneous blink peak velocity and amplitude, the main sequence, shared features previously found in uncomplicated Bell's palsy and blepharospasm patients. Furthermore, as in patients with typical Bell's palsy and idiopathic blepharospasm, both normal blinks and spasms were conjugate in spite of interocular differences in blink amplitude/peak velocity. We suggest that there is a correlation between the eyelid palsy and subsequent blepharospasm, and have designated this potentially new disease entity as Bell's palsy-induced blepharospasm. We propose a two-stage model for Bell's palsy-induced blepharospasm in which blink adaptive systems may produce the maladaptive consequence of eyelid spasms.

Adult↗
PubMed1984

Blepharospasm: demographic and clinical survey of 250 patients.

The pathophysiologic mechanisms of blepharospasm are poorly understood and the condition is often misdiagnosed. In an attempt to improve our understanding of blepharospasm, 250 patients with blepharospasm were surveyed. This heretofore largest reported series of blepharospasm patients reveals findings of clinical and demographic interest. For example, there is a 3 to 1 female preponderance, the age of onset is in the fifth or sixth decade of life in two-thirds of patients, the onset is usually gradual with progressive deterioration and involuntary spasms of other portions of face and body. Although specific ocular abnormalities may predispose a patient to blepharospasm, in most cases the blepharospasm is a form of focal dystonia often associated with other involuntary movements and neurologic abnormalities. Increased awareness of blepharospasm among primary care physicians as well as ophthalmologists, neurologists, psychiatrists, and other specialists should lead to early recognition and management of this disabling condition.

Adult↗
PubMed1993

[Blepharospasm; results of treatment with botulin].

OBJECTIVE: Discussion of clinical symptoms and differential diagnosis of blepharospasm and treatment with botulinum A toxin. Blepharospasm is an involuntary spasmodic contraction of the eyelids. Within a few years 35%-70% of the patients becomes severely disabled. DESIGN: Prospective, open study. SETTING: Academical Medical Centre, Amsterdam. METHOD: In the period 1985-1992 we have seen 85 patients with blepharospasm. Of these 69 were treated with botulinum toxin, a total of 436 treatments, with a mean dose of 25 IU for each eye. RESULTS: The cause of blepharospasm was unknown in 71 patients. Secondary blepharospasm occurred in: peripheral facial palsy (one patient), herpes zoster infection of the trigeminal nerve (2), brain infarct (1), use of neuroleptics (2), progressive supranuclear palsy (2), Shy-Drager syndrome (1), kernicterus (1), and morbus Sjögren (4). There were 18 patients with autoimmune diseases. 77 (91%) patients had a (very) severe form of blepharospasm. Electromyographic registration revealed a dysfunction of M. levator palpebrae in 7 patients. More than 70% of the patients were free of symptoms for a mean period of two months after each treatment. Local side effects were seen in 61 (14%) of the 436 treatments: ptosis, haematoma, dry eyes, and diplopia. CONCLUSION: Blepharospasm is a disabling disease and occurs sometimes in association with other neurological and ophthalmological diseases. Botulinum A toxin is a safe and effective therapy. Electrophysiological investigation is important in the differential diagnosis; it is unnecessary to do CT or MRI routinely.

Aged↗
PubMed2002

Bell's palsy-induced blepharospasm.

We report two patients with blepharospasm that appeared during the recovery phase of Bell's palsy. It is well known that hemifacial spasm occasionally appears after Bell's palsy; however, blepharospasm associated with Bell's palsy has been rarely reported so far. Blepharospasm appeared within a month after the onset of Bell's palsy, suggesting that a certain causal relationship may be present between Bell's palsy and blepharospasm. We speculate that corneal irritation caused by lagophthalmos contributes to the induction of blepharospasm. Another speculation is that abnormal afferent input from the paralyzed side contributes to the abnormal sensitization of the blink reflex, thereby facilitating the induction of abnormal facial motor outputs such as blepharospasm.

Adult↗
PubMed2004

Blepharospasm associated with multiple system atrophy: a case report and review of the literature.

Although blepharospasm has been occasionally associated with parkinsonism, it has rarely been reported in patients with multiple system atrophy (MSA). We report a 65-year-old woman with MSA who developed blepharospasm seven years after onset, rendering her functionally blind. Clinical course and the findings of magnetic resonance imaging indicated cerebellar type MSA. The blink reflex studies showed prolonged R2 response and enhanced recovery cycle, indicating an increased excitability of the brainstem interneurons. These results suggest that pathophysiology of blepharospasm in MSA is similar to that of essential blepharospasm. Recognition of blepharospasm in MSA patients is important, as blepharospasm is a treatable feature in this otherwise intractable disorder.

Aged↗
PubMed2003

Botulinum toxin A treatment in patients suffering from blepharospasm and dry eye.

BACKGROUND: Many patients with essential blepharospasm also show dry eye signs and symptoms. Botulinum toxin A is an effective treatment for reducing spasms in these patients. In this investigation, the effect of botulinum toxin A injections on tear function and on the morphology of the ocular surface in patients suffering from blepharospasm in combination with a dry eye syndrome was investigated. METHODS: Botulinum toxin A injections were applied to 16 patients with blepharospasm. All patients complained of dry eye symptoms and had reduced tear break up time values. A subjective questionnaire and ocular examinations including tear break up time, Schirmer test without local anaesthesia, and rose bengal staining were evaluated before, 1 week, 1 month, and 3 months after injection. Impression cytology was performed before, 1 month, and 3 months after botulinum toxin A treatment. RESULTS: Although all patients were relieved of blepharospasm after botulinum toxin injections, only three noticed an improvement in dry eye symptoms. Eight patients noticed no difference and five complained of worsening. Tear break up time was found to be increased 1 week and 1 month after injections. Schirmer test measurements were reduced up to 3 months. Rose bengal staining slightly increased 1 week after injections. Impression cytology showed no definite change in conjunctival cell morphology 1 month and 3 months after botulinum toxin A injections. CONCLUSION: In the patients presented here suffering from blepharospasm and dry eye, botulinum toxin A injections were effective in relieving blepharospasm but were not successful in treating dry eye syndrome.

Adult↗
PubMed2004

Primary blepharospasm: diagnosis and management.

Primary blepharospasm is an adult-onset focal dystonia characterised by involuntary contractions of the orbicularis oculi muscles. Patients may have various types of movements arising from the different parts of the orbicularis oculi muscle. These include typical blepharospasm associated with Charcot's sign, pretarsal blepharospasm and flickering of the eyelids. Primary blepharospasm may be associated with so-called apraxia of eyelid opening as well as dystonia in the lower face, jaw or cervical muscles. Unless there are clinical clues to a symptomatic cause, adults presenting with blepharospasm do not require extensive aetiological investigation because the condition is rarely due to an identifiable condition. As the aetiology of primary blepharospasm is largely unknown, therapeutic approaches are symptomatic, with type A botulinum toxin being the treatment of choice.

Blepharospasm↗
PubMed2004

[Results of long term treatment of essential blepharospasm and facial hemispasm with botulinum toxin A].

Botulotoxin A (BT) was administered to 51 patients (40 patients with essential blepharospasm, 11 patients with facial hemispasm) 299 times since December 1993 till January 2002. Total doses of 20-48 U of the preparation BOTOX (Allergan Inc.) were administered bilaterally in essential blepharospasm, doses of 12-24 U were administered unilaterally in facial hemispasm. The mean duration of therapeutic effect evaluated by the patient was 12.2 weeks (3-28 weeks) in the blepharospasm group and 13.8 weeks (7-35 weeks) in the hemispasm group. No general side effects were noted. Local complications in the blepharospasm group were observed in 8.83% (ptosis in 5.62%) and those in the facial hemispasm in 8.16% (ptosis in 2.04%) from the total number of BT administration in the respective groups. The therapeutic effect during the long-term administration was decreasing and required increasing doses of BT by more than 10%, as observed in 9 patients with essential blepharospasm (22.5%). The mean increase of BT dose required to maintain sufficient therapeutic effect was by 32% (14-66%). Two patients with the longest 8-year period of observation have not required increasing dosages above 16 U unilaterally over the last 5 years. The treatment with BT-A exerted the same effects in most cases even during long-term therapy lasting several years. The need to increase the doses of the administered BT was more frequent during the first two years of the treatment. Locally administered botulotoxin A is a safe and efficient remedy for the therapy of essential blepharospasm and facial hemispasm, the efficiency being unchanged in more than 80% of patients.

Adult↗
PubMed1991

Memory-contingent saccades and the substantia nigra postulate for essential blepharospasm.

Essential blepharospasm and cranial dystonia are related focal dystonias of unknown aetiology. Blepharospasm induced by acute dopamine depletion in parkinsonism restricts saccade initiation possibly via the substantia nigra pars reticulata (SNpr). If essential blepharospasm and cranial dystonia similarly restrict saccades, then a selective, somatotopically arranged pathway such as the SNpr may be involved. To test this possibility memory-contingent and visually-guided saccades were measured in patients with essential blepharospasm and cranial dystonia. The latency of both forms of saccades was either significantly prolonged or excessively variable, while the accuracy and peak velocity of these fast eye movements were similar to age-matched control subjects. Essential blepharospasm and cranial dystonia alter the initiation of saccadic eye movements. Subcortical brain regions or pathways where eyelid, saccade and cranial/cervical motor control are somatotopically approximated, such as the SNpr, may be involved in blepharospasm.

Blepharospasm↗
PubMed1990

Ocular movements in essential blepharospasm.

In essential blepharospasm histopathologic and electrophysiologic evidence supports the existence of lesions in proximity to brainstem nuclei controlling ocular movements. We studied horizontal ocular movements in eight patients who had been treated previously with surgery or botulinum toxin injection to control essential blepharospasm (mean age, 58 years) and compared these with seven control subjects who did not have blepharospasm (mean age, 68 years). We examined fixation stability, saccades, the vestibulo-ocular reflex, visual enhancement and suppression of the vestibulo-ocular reflex, optokinetic nystagmus, and pursuit by using digitally sampled, direct current electro-oculography. Patients with blepharospasm exhibited no ocular movement abnormalities. Since quantitative aspects of ocular movements are sensitive to nonspecific brainstem lesions, the absence of abnormal ocular movements suggests that the lesion in blepharospasm is specifically limited to neurons regulating the facial muscles.

Adult↗
PubMed1987

Effectiveness of botulinum toxin therapy for essential blepharospasm.

The effectiveness of Botulinum toxin (Oculinum) therapy in 76 patients with the diagnosis of essential blepharospasm was analyzed. Botulinum offers relief to almost all patients suffering from essential blepharospasm, however, this relief is usually temporary. The response time for repeated treatments tended to be longer than the first treatment. Patients with mild blepharospasm responded significantly longer to Botulinum injection, than those with severe spasms. The response to Botulinum was not significantly different in patients with Meige syndrome than in patients with only essential blepharospasm. Patients previously treated surgically for essential blepharospasm did not respond differently than those patients with no previous surgical therapy. The authors believe that Botulinum toxin injection is an effective, although temporary, mode of therapy for the signs and symptoms of this focal dystonia. The authors recognize that there may be psychologic factors affecting the response.

Blepharospasm↗