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 Impact on Quality of Life of Botulinum Toxin Treatments for Spasmodic Dysphonia and Oromandibular Dystonia
Neil Bhattacharyy, Daniel Tarsy
Arch Otolaryngol Head Neck Surg
2001;127; 389-392
 Outcomes of Botulinum Toxin Treatment for Patients With Spasmodic Dysphonia
Michael S. Benninger, Glendon Gardner, Cynthia Grywalski
Arch Otolaryngol Head Neck Surg
2001; 127; 1083-1085







mise à jour du
16 octobre 2003
Clin Exp Neurol
A Review of 20 Cases of Spastic Dysphonia
S Whyte, P Darveniza
St Vincent hospital, Sydney, Australie
Blepharospasm-oromandibular dystonia syndrom CD Marsden
Le bâilleur, Yawning Man or De Gaper Pieter Brueghel the Elder
Les convulsions de la face, une forme clinique de convulsion faciale, bilatérale et médiane Meige H


Spastic dysphonia (SD) is an uncommon, sometimes debilitating, disorder of unknown aetiology, probably a form of focal dystonia, originally described as nervous hoarseness by Traube in 1871.

In 1973 a subdivision of the entity was suggested by Aronson who distinguished between abductor SD, where there is a widening of the glottis causing intermittent aphonia with breathy phonation and adductor SD, associated with adduction of the true and/or false vocal cords, with speech characterised by a strained, low-pitch, harsh, monotonous, staccato, effortful voice with intermittent voice stoppages.

Over a 6-year period 20 patients (7 male and 13 female) with the characteristics of adductor SD were seen at St Vincent's Hosptial, Sydney. All underwent a routine medical and neurological examination, voice analysis and review by an ear nose and throat surgeon (the latter two reported elsewhere). The clinical notes of these patients were reviewed and combined with the results of a mail survey (response 17 of 20 subjects).

Clinical Features : Nine of the patients experienced a voice tremor associated with an essential tremor (SD+ET), variably involving the head, trunk and limbs (one patient had a family history of tremor). Seven patients' employment required the active use of their voices (e.g. teachers, a telephonist).

The patients were divided into those with essential tremor (SD+ET) and those without essential tremor (SD alone).

Onset : Ten patients experienced a gradual onset of dysphonia, with the remaining 10 noting an abrupt onset, in 3 related to an upper respiratory tract infection and in 7 to psychosocial stress. The mean age of onset in patients with SD alone was 45 years (range 30 to 54 years) and for SD+ET 52 years (range 15 to 67 years).

Course and Duration : The mean duration of the speech disorder prior to review was 8.8 years for SD alone, and 14 years for SD+ET. Of the 17 patients responding to the later postal survey, 8 noted no change, 4 a worsening and 5 an improvement in their speech since the onset of SD (equally divided between patients with or without ET).

Factors Affecting the Quality of Speech : Patients with or without ET were equally divided with respect to factors that worsened speech, but no patients with SD+ ET noted an improvement with strong emotional statements, waking in the morning, use of a loud voice or on being woken from sleep. Only 1 or 2 patients with SD+ET reported improving with yawning, swallowing, speaking in a low or high pitch, laughing, making spontaneous statements or singing. Two patients with SD alone noted that inspiratory speech was more effective than expiratory speech.

Abnormalities on Physical Examination : The following abnormalities were noted on physical examination: a brisk jaw or facial jerk (2/20 subjects); mild cogwheel rigidity of the upper limbs (1/20); hypertensive retinopathy grade 2 (3/20) and dysphagia (pharyngeal web) (1/20). No structural abnormalities were noted on ENT examination in any subject.

Therapy : Only a small number of patients received pharmacological therapies including alcohol (SD+ET 2/2 excellent responses, SD alone, 2/4 slight to good response), propranolol (SD+ET 3/5 slight to good response), benzhexol (SD+ET 1/2 slight response; SD alone, 3/3 slight response) and doxepin (SD+ET 1/2 slight response; SD alone, 2/2 slight response). Two patients were intolerant of the side effects of benzhexol. Other therapies given in 1 or 2 patients only with no or only a slight response included phenobarbitone, clonazepam, clobazam, diazeparn, benzhexol, carbamazepine and intravenous lignocaine/mexiletine.

Speech therapy yielded a slight to good response in 7 of 13 patients. Relaxation therapy resulted in a slight to good response in 6 of 8 patients. Patients with SD+ET and SD alone responded equally to speech and relaxation therapy.

Two patients with SD alone underwent recurrent laryngeal nerve sectioning, one with a good response and the other with a temporary improvement (although the speech remained better than prior to operation).

Discussion : The patient group described is similar to other published series although showing a female predominance. The onset was mainly in middle age (slightly later for patients with SD+ET), and was in half related to an upper respiratory tract infection or psychosocial stress, probably fortuitous.

The over all course showed little tendency for spontaneous improvement, with a number of factors such as stress, public speaking, tiredness, strong emotions, upper respiratory tract infections and prolonged use of the voice temporarily worsening speech in patients with both SD+ET and SD alone. Patients with SD alone experienced a temporary improvement in speech related to laughing, yawning, slow speech, use of a quiet voice, making spontaneous statements,employing high and low pitch, chewing, swallowing and on first waking in the morning. Little improvement in speech was noted by patients with SD+ET.

To date, SD has proven to be a distinct nosological entity. The presence of any other neurological abnormality should alert the clinician to the existence of another disease process. Dysphagia, present in one patient in this series, was due to a pharyngeal web.

The number of patients given pharmacological therapy was small, with SD+ET patients responding to alcohol or propranolol, whereas patients with SD alone responded less frequently and less well to alcohol or benzhexol or doxepin. Speech therapy offered slight to good improvement and relaxation therapy slight improvement in both patient groups.

Of 2 patients undergoing recurrent laryngeal nerve sectioning (RLNS) one experienced a good outcome, whereas the other noted only temporary good improvement, although speech remained better than prior to surgery. Dedo et al in a review of 121 patients undergoing RLNS for SD found a 92% satisfactory reduction in spasticity in the first 1 to 3 years. In a slightly different patient group (including more patients with voice tremor) Aronson et al. recorded only a 36% improvement at 3 years.

Botulinum toxin injection has recently been shown to bc of benefit, but awaits further detailed investigation.

An association between essential tremor and the characteristic features of SD bas been noted frequently. Aronson et al. noted the regular occurrence of voice stoppages at the point of highest pitch on each tremor cycle (tremor median frequency of 5.5 Hz) and a constant strained-strangled dysphonia in patients with SD+ET. In contrast, patients with SD alone tended to experience irregular voice stoppages and an intermittent strained-strangled dysphonia. Aronson therefore postulated that patients with SD+ET represent an extreme variant of essential tremor. Alternatively this variant may represent an association between essential tremor and a focal dystonia, as has been noted with other disorders (e.g. spasmodic torticollis and essential tremor)