Adduction laryngeal dystonia: Proposal and evaluation of nasofibroscopy

Speech and Hearing School, Pontíficia Universidade Católica de São Paulo, Brazil.
Brazilian journal of otorhinolaryngology (Impact Factor: 0.65). 07/2006; 72(4):443-6. DOI: 10.1016/S1808-8694(15)30987-3
Source: PubMed
ABSTRACT
Dystonias are organic central motor processing disorders characterized by involuntary muscular contractions or incontrollable spasms induced by task-specific movements. Adduction laryngeal dystonias present with important speech impairments, with inappropriate spasms and abrupt voice breaks. The diagnosis is based on clinical features, evaluation by a speech therapist and transnasal fiber optic laryngoscopy.
Our objective is to propose and evaluate a task-oriented transnasal fiber optic laryngoscopy protocol, which shows the spasms, and propose maneuvers that reduce or make them disappear, in order to facilitate the diagnosis.
transversal study. Analysis of the transnasal fiber optic laryngoscopy records of 15 patients with adductor laryngeal dystonia using the proposed protocol.
most of the speech and non-vocal tasks allowed us to identify the spasms and reduce or make them disappear. We propose the exclusion of two of the maneuvers that dont bring new data to the evaluation.
the protocol was useful for the evaluation of the patients, showing changes in muscle behavior in the structure under investigation.

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Adduction laryngeal dystonia:
proposal and evaluation of
nasofibroscopy
Summary
Noemi Grigoletto De Biase
1
, Paula Lorenzon
2
,
Mariana Dantas Aumond Lebl
3
, Marina Padovani
4
,
Ingrid Gielow
5
, Glaucya Madazio
6
, Miriam
Moraes
7
1
PhD. Associate Professor – Speech and Hearing School - Pontifícia Universidade Católica de São Paulo. MD, collaborator at the Department of Otorhinolaryngology/
Head and Neck Surgery – Voice and Larynx Lab – Federal University of São Paulo – Paulista School of Medicine.
2
MD. Otorhinolaryngologist and postgraduate student (MS) of Otorhinolaryngology – Federal University of São Paulo – Paulista School of Medicine (UNIFESP-EPM).
3
MD. MS in Otorhinolaryngology– Federal University of São Paulo – Paulista School of Medicine (UNIFESP-EPM).
4
Speech and Hearing Therapist. MS in Human Communications Sciences – Paulista School of Medicine (UNIFESP-EPM).
5
Speech and Hearing Therapist. PhD in Human Communications Sciences – Paulista School of Medicine (UNIFESP-EPM). Head of the Rehabilitation Department
– Head and Neck Surgery/Voice Lab - UNIFESP-EPM.
6
Speech and Hearing Therapist. MS in Communications Sciences – Paulista School of Medicine (UNIFESP-EPM). PhD Student in Human Communications Sciences
(UNIFESP-EPM).
7
Speech and Hearing Therapist, Expert in Voice – CEV (Voice Study Center). Collaborator at the Voice and Larynx Lab.
Federal University of São Paulo – Paulista School of Medicine. Department of Otorhinolaryngology – Voice and Larynx Lab.
Mailing Address: Rua Madre Rita Amada de Jesus, 106 Granja Julieta 04721-050 São Paulo SP.
Paper submitted to the ABORL-CCF SGP (Management Publications System) on June 28th, 2005 and accepted for publication on March 29th, 2006.
Dystonias are organic central motor processing disorders
characterized by involuntary muscular contractions or
incontrollable spasms induced by task-specific movements.
Adduction laryngeal dystonias present with important
speech impairments, with inappropriate spasms and abrupt
voice breaks. The diagnosis is based on clinical features,
evaluation by a speech therapist and transnasal fiber optic
laryngoscopy. Aim: Our objective is to propose and evaluate
a task-oriented transnasal fiber optic laryngoscopy protocol,
which shows the spasms, and propose maneuvers that reduce
or make them disappear, in order to facilitate the diagnosis.
Methods: transversal study. Analysis of the transnasal fiber
optic laryngoscopy records of 15 patients with adductor
laryngeal dystonia using the proposed protocol. Results:
most of the speech and non-vocal tasks allowed us to identify
the spasms and reduce or make them disappear. We propose
the exclusion of two of the maneuvers that don’t bring new
data to the evaluation. Conclusion: the protocol was useful
for the evaluation of the patients, showing changes in muscle
behavior in the structure under investigation.
Keywords: neurological disease, dystonia, glottal
incompetence, vocal fold.
ORIGINAL ARTICLE
Rev Bras Otorrinolaringol
2006;72(4):443-6.
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INTRODUCTION
Dystonias are central motor processing organic
disorders characterized by involuntary muscle contrac-
tions or uncontrollable spasms during an activity
1,2
. These
abnormal movements may be sustained for a variable
amount of time, from one second to minutes and may
occur in any part of the body; they are clearly not psycho-
genic in origin; however, they can get worse with fatigue,
stress and emotions
3
. Dystonias have an unknown cause,
although most authors think that the base ganglions are
involved
4
. The classification of dystonias is usually related
to the muscle group involved, being focal when it affects
a specific muscle group, and generalized when it affects
a great number of muscle groups. Between both types we
find the segmentary type, which affects muscle groups
which are close to each other in proximity. In children,
the symptoms are of focal onset, followed by its spread
to other body parts, while in adults, the symptoms usually
remain focal and frequent in the head and neck
1
- more rare
focal forms involve the larynx intrinsic muscles. As far as
the latter is concerned, the following types are described:
adduction, abduction and respiratory
5-7
. The respiratory is
the less frequent one and also the more concerning one,
since it causes respiratory restriction of varied degrees,
without dysphonia during speech
3,8
. Abduction larynx focal
dystonia is not very common and spasms occurs during
speech formation on the posterior crycoarytenoid muscles.
With this there is air escaping during sound production,
translated by intermittent blowing voice in chained speech,
more rare forms involve the larynx intrinsic muscles.
Focal adduction dystonia is more frequent. In this
type there is a strong contraction of adducting muscles
during speech formation, in other words, hyper adduc-
tion is innadequate
3
. In this type, voice is tense/strangled,
with frequent sound breaks and clear vocal strain. The
involvement varies and may seriously impair communica-
tion. Usually, there is little alteration in laughter, singing,
whisper and falsetto
6
. The breaks occur when the vocal
cords coming together is so intense that they do not allow
air to pass through. The strongest contraction occurs when
the vocal folds are adducted, that is during sound produc-
tion, and they are more evident in words that start by a
vowel. It may be followed by constant larynx tremor alone
or involving pharyngeal muscles, or from the so called
dystonic tremor seen only during speech production
4,5
.
Since there is no specific examination, diagnosis is based
on clinical signs: hearing-perceptive voice assessment
and laryngoscopy, specially through flexible optical fiber
nasofibroscope
3,10
. Adduction larynx dystonia must be
specially differentiated from the skeleton-muscles tension
syndromes and some cases of psychogenic dysphonia
which bear resemblance in chained speech. Voice acoustic
analysis objectively translates audible voice patterns and
allows for the identification of voice tension and breaks.
Electromyography may be useful in diagnostic confirma-
tion, and the most frequently found signs are a sudden
and periodic increase in electrophysiological potentials
of the thyroarythenoid muscle and extension on pre and
post-phonatory electrical activity
11
. Exam by nasofibro-
laryngoscopy, more physiologically compatible than the
telescopic exam, allows for the execution and evaluation
of tasks that show clearly the clinical characteristics of
adduction laryngeal dystonia.
Thus, our goal is to propose and assess a protocol
of nasofibroscopy that uses tasks that show spasms and
tasks that reduce them or make them disappear, in order
to facilitate analysis and diagnosis.
MATERIALS AND METHODS
A protocol of nasofibroscopy exam for the as-
sessment of the palate, pharynx and larynx (Attachment
1), performed in 15 patients with adduction laryngeal
dystonia, with diagnosis by audible-perceptive analysis,
anterior nasofibrolaryngoscopy, electromyography and
improvement after the injection of Botulin toxin in the
vocal fold. Nasal-laryngeal fibroscopy was carried out after
the patients signed an informed consent, and a Machida
ENT-30 PIII device was used; the exam was recorded on
a videocassette tape for later analysis which was carried
out by three otorhinolaryngologists. The study was carried
out in the Larynx and Voice outpatient ward of the Federal
University of São Paulo - Paulista School of Medicine. The
nasal-laryngeal fibroscopy exam was carried out at least six
months after the injection of the Botulin Toxin and after
the patient and the authors have observed a return of the
tense/strangled characteristic in the patient’s voice quality
through audible-perceptive analysis. The nasofibroscope
was introduced through either the right or the left nostril,
and its tip placed close to the choana, in order to assess
the palate. The analysis counted on more or less low tone
emissions, with and without glottal adduction participa-
tion and during rest and swallowing. We also requested
the patients to produce phrases. Afterwards, the device
was placed on the rhinopharynx in order to observe the
movement of the pharynx during rest and during the ut-
terance of low and high sounds. Most of the assessment
was carried out with the tip close to the larynx, in such a
position that allowed the glottis to be seen. The patients
were asked to utter low and high sounds, sound produc-
tion with and without glottal adduction, phrases with a
predominance of sonorous sounds, whispered voice, high
intensity sound production and unusual tasks, and also,
other non-phonatory sounds such as inhaling sound pro-
duction, whistling and sniffing (Attachment 1). The exams
were recorded for later analysis under normal and slow
speeds, by three otorhinolaryngologists with experience
in laryngology and dystonias, with agreement among the
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observers.
RESULTS AND COMMENTS
In assessing the palate, all the patients presented
complete closure of the pharyngeal sphincter. The assess-
ment during phrases uttering was made difficult because
of the spasm and did not add data in relation to the other
tasks. Most of the patients did not show palate tremor dur-
ing rest, only two of them did. The uttering of vowels “é”,
“i” and “u” allows us to see differences in spasm intensity
according to pitch variations. The uttering of sounds “s”
and “z” allowed the assessment of a reduction or even
disappearance of spasms during blunt uttering in adduc-
tion dystonias.
As to the pharynx, no patient had rest spasms and
during the production of vowel “é”, in six patients we
observed contractions of the pharynx walls.
Evaluation during inhaling did not show epiglottis
or larynx movements, and such investigation will be useful
in cases of respiratory dystonia. Laryngeal tremor during
rest was seen in one case.
In assessing the larynx we noticed spasms during
the “é” vowel sound in the habitual tone of voice, with
disappearance or reduction with high glissing for most
of the patients studied. Only three patients did not show
any change, although in three other patients there was no
change, three other could not gliss. We observed that our
patients can not always do the ascending or the descending
glissing sound. The increase in fundamental frequency with
the utterance of the hyperacute “i” was possible with all
of them, and only in two of them we did not see spasms
reduction or disappearance. Thus, there was a difference
in relation to the presence or reduction of spasms in all
the patients during hyperacute sound production, except
in two, and such task is useful due to its easy performance
and spasms modification. The highest sounds, specially in
falsetto, are uttered with a slight separation of the vocal
folds, and this reduces spasm stimulus.
The utterance of phrases with a predominance
of sonorous sounds followed by the phrase with blunt
sounds allowed us to see a reduction of spasms in the
blunt utterances. The comparison shows the difference,
not always seen in chained speech. During whispering,
the spasm disappeared in six patients, reduced in two,
and the others were not able to produce the whisper and
only repeated the phrase at a lower intensity, keeping the
spasms. During whispering, since the vocal folds do not
touch each other, we were able to see a spasm reduction
or even disappearance. The utterance of the word “Gol”
at a high intensity did not contribute to the assessment
because it allows for “pitch” and intensity variations in a
very short word. We could notice spasms in 7 patients, just
like it happened with the utterance of the “é”, reduction
in 3 patients and disappearance in the others, in whom
we noticed high utterances.
In the tasks - inhaling phonation, whistling and sniff-
ing - no patients had spasms. Such movements are not the
ones commonly used for phonation and, therefore, should
not trigger spasms, which depend on habitual phonation.
It is likely that the brain circuit used for these tasks be dif-
ferent from the one used for phonation and be intact.
Thus, the protocol proved to be useful in the as-
sessment of patients with laryngeal dystonia, bearing tasks
that allow us to show the presence of spasms and tasks
that show its reduction or even disappearance. Some tasks
do not prove useful, and we propose their exclusion, thus
making the assessment faster and more objective - they
are: velopharyngeal closure investigation, including the
use of phrases, during swallowing and the utterance of
the word “Gol”.
CONCLUSION
The protocol was useful in the assessment of pa-
tients, showing a change in the behavior of the muscle
groups studied according to the tasks performed.
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Universidade Federal de São Paulo/UNIFESP
Department of Otorhinolaryngology
Laryngology and Voice
Joint Scale for Laryngeal Dystonia Assessment
Nasofibroscopy Assessment Protocol
Name:
Date:
General impression:
Functional assessment of the soft palate
Tasks Comments
Rest
Swallowing
É- I -U
S
SS-ZZZ
Papai pediu pipoca para Pedro
Mamãe comeu mamão
Um homem e uma mulher viram um anjo voando
O sapo saltou o sapato
Assessment of non-phonatory tasks, speech and voice
Tasks Comments
rest
Long “eh”
“eh” ascending gliss
“ih” Hyperacute
Inhaling phonation
Snif
-snif
whistle
whisper “Shh! The baby is asleep“
Shout “Gol!”
Repeat: “ Um homem e uma mulher viram um anjo voando.”
Repeat: “O sapo saltou o sapato”
Adapted from the Joint Scale of Spasmodic Dysphonia Assessment (USDRS)
Stewart CF et al. Adductor spasmodic dysphonia: standard evaluation of symptoms and severity. J V
oice 1997;11(1):95-103.
Attachment 1.
Page 4
  • [Show abstract] [Hide abstract] ABSTRACT: Although the latency between the initiation of thyroarytenoid electrical activity and the onset of phonation generally is increased in patients with adductor laryngeal dystonia, there is disagreement about whether there is overlap of latency values in these patients and normal subjects. The goal of this article was to compare the severity of dysphonia with the latency between electrophysiological activation of the thyroarytenoid muscle (TA) and the onset of phonation in patients with adductor laryngeal dystonia and compare the values with normal controls. Twenty-one patients with adductor dystonia and 15 control patients underwent laryngeal electromyographic (EMG) examination of the left TA. We measured the latency from initiation spike of the electric activity of the TA muscle to the onset of phonation. Three speech-pathologists/voice specialists arrived at a consensus to rate the perceptual evaluation of voice quality for the study group. The average latency measured for patients with mild dysphonia was 332 milliseconds, for moderate dysphonia was 426 milliseconds, and for the severe dysphonia was 792 milliseconds. We used the Spearman's correlation test to compare the latency time values and the dysphonia's degree of severity (P<0.05). Latency was significantly and directly related to the degree of severity of dysphonia.
    No preview · Article · Sep 2009 · Journal of voice: official journal of the Voice Foundation
  • Source
    [Show abstract] [Hide abstract] ABSTRACT: Dystonia is a central motor processing neurological disorder characterized by abnormal, often action-induced, involuntary movements or uncontrolled spasms. To compare patients with the diagnoses of focal and segmental adductor laryngeal dystonia at the Neurolarynx Outpatient Clinic of the Federal University of São Paulo. A clinical retrospective study of data collected from patient registries from 2003 to 2009. Of 34 patients, 25 presented focal dystonia and 9 presented segmental dystonia. There were 30 females (88. 2%) and 4 males (11. 8%). A relation with a traumatic event was reported in 11 cases (32. 4%). Vocal tremor was observed in 21 patients (61. 8%). The mean age at onset, the age at diagnosis, and time between the onset and the diagnosis were respectively 55, 61. 3 and 6. 3 years. There was no statistical difference between patients with focal laryngeal adductor dystonia and segmental dystonia in the study data. There were no statistical differences among patients with focal adductor laryngeal dystonia and segmental dystonia relating to age of onset, age of diagnosis, gender, time between onset and diagnosis, presence of associated tremor, and relation to trauma.
    Full-text · Article · Aug 2011 · Brazilian journal of otorhinolaryngology
  • Source
    [Show abstract] [Hide abstract] ABSTRACT: Introduction: Although syndromes that cause voice tremor have singular characteristics, the differential diagnosis of these diseases is a challenge because of the overlap of the existing signs and symptoms. Objective: To develop a task-specific protocol to assess voice tremor by means of nasofibrolaryngoscopy and to identify those tasks that can distinguish between essential and dystonic tremor syndromes. Methods: Cross-sectional study. The transnasal fiberoptic laryngoscopy protocol, which consisted of the assessment of palate, pharynx and larynx tremor during the performance of several vocal and non-vocal tasks with distinct phenomenological characteristics, was applied to 19 patients with voice tremor. Patients were diagnosed with essential or dystonic tremor according to the phenomenological characterization of each group. Once they were classified, the tasks associated with the presence of tremor in each syndrome were identified. Results: The tasks that significantly contributed to the differential diagnosis between essential and dystonic tremor were /s/ production, continuous whistling and reduction of tremor in falsetto. These tasks were phenomenologically different with respect to the presence of tremor in the two syndromes. Conclusion: The protocol of specific tasks by means of transnasal fiberoptic laryngoscopy is a viable method to differentiate between essential and dystonic voice tremor syndromes through the following tasks: /s/ production, continuous whistling and reduction of tremor in falsetto.
    Full-text · Article · Dec 2015 · Brazilian journal of otorhinolaryngology