American Journal of Otolaryngology–Head and Neck Medicine and Surgery 42 (2021) 102940

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American Journal of Otolaryngology–Head and Neck

Medicine and Surgery
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Sulcus vocalis in spasmodic dysphonia—A retrospective study

Nupur Kapoor Nerurkar *, Deeksha Agrawal, Dipali Joshi
Bombay Hospital Voice and Swallowing Centre, Bombay Hospital & Medical Research Centre, Mumbai, Maharashtra, India

A R T I C L E I N F O A B S T R A C T

Keywords: Background: Spasmodic dysphonia (SD) is a neurological condition of the larynx characterised by task specific,
Sulcus vocalis involuntary spasms of the intrinsic laryngeal muscles causing frequent voice breaks during speech. The current
Spasmodic dysphonia treatment modality involves Botulinum Toxin injections into the affected group of muscles. This has yielded
Abductor spasmodic dysphonia
satisfactory results in Adductor SD (ADSD) and mixed SD but not in Abductor SD (ABSD). Sulcus vocalis is a
Mixed spasmodic dysphonia
Stroboscopy
morphological condition of the vocal folds with invagination of the superficial epithelium into the lamina propria
or deeper layers. It is characterised by breathiness in voice and hypophonia.
In our voice clinic, patients diagnosed with SD were occasionally found to have a sulcus on flexible stroboscopy.
Studies have revealed an asymmetric stimulation of both the adductor and abductor group of muscles in ABSD
and a predominant possibly symmetric stimulation of the adductor group of muscles in ADSD. Our objective was
to study any significant association between vocal fold sulcus and two groups within SD; group one being ADSD
and group two being both ABSD and Mixed SD. A literature review did not reveal any studies suggesting an
association between SD and vocal fold sulcus to date.
Methods: A retrospective review of the stroboscopic video recordings as well as file records of all patients
diagnosed with SD between January 2016 and September 2019 was conducted at our voice clinic. The first
author was the laryngologist who had diagnosed SD and its type on the basis of hearing the voice and making the
patient perform various vocal tasks with and without flexible videostroboscopy. The SD patients were divided
into two groups with the first group consisting of ADSD patients and the second group consisting of ABSD as well
as Mixed SD patients. The presence or absence of vocal fold sulcus was noted in all the SD patients. Odds ratio
was used to establish statistical significance of the presence of vocal fold sulcus in the two SD groups.
Results: Among the 106 patients of SD, 62 patients were males and 44 were females. A total of 84 patients were
diagnosed as ADSD, 10 as ABSD and 12 as Mixed SD patients.
Vocal fold sulcus was noted in 5 out of 84 patients of ADSD, 4 out of 10 patients of ABSD, and in 3 out of 12
patients of mixed SD. Odds Ratio of 7.37 (C.I. = 2.063–26.35) was obtained for the second group of patients i.e.
ABSD and Mixed SD.
Conclusion: Our study revealed a significant association between patients of SD having an abductor component
(ABSD and mixed SD) and vocal fold sulcus. The two hypothesis proposed for this are the possibility of asym­
metrical adductor and abductor muscle stimulation in SD being responsible for the development of a vocal fold
sulcus or the primary presence of a vocal fold sulcus contributing to altered sensory feedback resulting in SD.
Further study to evaluate this, as well as a study of the vocal response to medialisation procedures for patients of
ABSD with sulcus is recommended.

1. Introduction initiation and termination of speech with vowels [3]. ABSD comprises a
majority of the remaining cases and presents with hypophonia and
Neurological disorders can have a profound influence on speech and breathy breaks in speech which are prolongations of the voiceless con­
phonation. SD is a task-specific focal laryngeal dystonia characterised by sonants [4]. Rarer forms of SD include Mixed SD, where patients have
irregular and uncontrolled voice breaks that interrupt normal speech features of both ADSD and ABSD; singer’s SD, where patients only have
flow [1]. ADSD, which comprises 82% to 96.6% of cases of SD [2] symptoms during singing; and adductor breathing SD, where adductor
presents with a strained or strangled voice and voice breaks with abrupt spasms occur during breathing resulting in stridor [3].

* Corresponding author at: Bombay Hospital Voice and Swallowing Centre, 2nd Floor MRC, Bombay Hospital and Medical Research Centre, Mumbai 400020, India.
E-mail address: nupurkapoor@[Link] (N.K. Nerurkar).

[Link]
Received 15 September 2020;
Available online 28 January 2021
0196-0709/© 2021 Elsevier Inc. All rights reserved.
N.K. Nerurkar et al. American Journal of Otolaryngology–Head and Neck Medicine and Surgery 42 (2021) 102940

SD is rare with the estimated incidence as low as 1 per 100,000 cases had been taken for all patients to rule out any other underlying neuro­
[5] with the exact pathophysiology not known. There seem to be three logical condition as per the protocol. Flexible videolaryngostroboscopy
different neurological mechanisms involved in the pathophysiology of had been performed using the Olympus flexible stroboscope ENFV3.
SD: loss of cortical inhibition, sensory input disturbances, and neuro­ Vocal fold motion during adduction, abduction and the mucosal wave
anatomical changes [1]. Simonyan et al. suggested a disorganization of patterns with a pitch glide saying “eee”, with fluent speech (rainbow
both basal ganglia-thalamo-cortical and cerebello-thalamo-cortical cir­ passage), with a whispered voice and during singing are noted as a
cuits in SD patients [6]. Currently it is not clear whether the disorder has routine in suspected cases of SD. Ventricular fold hyper-adduction,
a genetic basis; most cases are sporadic [5], but case series report as high anteroposterior squeeze and vocal tremors are specifically noted. On
as 20% may have other forms of focal dystonia such as writer’s cramp reviewing the stroboscopy recordings of all the SD patients, the presence
[7]. Blitzer et al. reported that 12% had a family history of dystonia [8]. or absence of a vocal fold sulcus, or any other lesion, was confirmed with
SD can occur in conjunction with an irregular vocal tremor on phonation the chart records. The first author was the laryngologist who had diag­
in 25% patients [9]. Muscle tension dysphonia (MTD) may mimic the nosed SD and its type on the basis of hearing the voice and making the
voice characteristics of ADSD thus leading to diagnostic confusion even patient perform various vocal tasks with and without flexible
among experienced clinicians [10]. ABSD is a less clearly understood videostroboscopy.
entity and more challenging to manage than ADSD. In ABSD, the vocal Odds ratio was used to establish an association between vocal fold
fold hypo-adduction was thought to be due to spasmodic bursts in one or sulcus and the two groups of SD, Group 1 being ADSD and Group 2 being
both of the posterior cricoarytenoid (PCA) muscles, the primary both ABSD and mixed SD.
abductor muscle of the larynx [11]. It is not known, however, if the
disorder is caused by a lack of muscle activation, or spasmodic bursts or 3. Observations
hyperactivity in the abductor muscles, interfering with vocal fold
closure for speech [12]. A study by Ludlow et al. reported abnormal A total of 106 patients with SD, both new and follow-up, visited our
muscle activation primarily affecting the cricothyroid muscles in 6/18 Voice Clinic from January 2016 to September 2019. The age at onset of
patients of ABSD and the rest of the patients revealed abnormal muscle symptoms ranged from 16 years to 73 years with an average of 47.8
activation primarily affecting the PCA muscles [13]. However, a study years. Mean duration of symptoms was 6 months at first consultation. A
by Cyrus et al. [4] revealed that the Electromyography differences from male predominance was noticed in our SD patients with 62 (58.5%)
normal in ABSD patients involved the thyroarytenoid (TA) and crico­ males and 44 (41.5%) females. Among the 106 patients, 84 (79.2%) had
thyroid muscles, the primary adductor and tensing muscles of the lar­ predominant adductor spasms (43 males and 41 females), 10 (9.4%) had
ynx, and not the PCA muscle, as has been previously suggested. A predominant abductor spasms (8 males and 2 females) and 12 (11.3%)
significant asymmetry was found in the TA levels between the 2 sides in had features of both or Mixed SD (11 males and 1 female) as depicted in
these ABSD patients [4]. Usually the diagnosis of SD can be reached by Table 1. Sulcus vocalis was documented as an associated finding in 4
perceptual analysis of voice and stroboscopic findings. Other tools, such patients of ABSD (Fig. 1) (40%), 5 patients of ADSD (5.95%) and 3 pa­
as laryngeal electromyography, acoustic and aerodynamic analysis and tients of mixed SD (25%) as represented in Fig. 1. Vocal tremor was an
high-speed laryngoscopy have also been used, particularly to measure associated finding in a total of 29 patients (24 patients of ADSD, 1 pa­
response to treatment, but none offers any diagnostic certainty [3]. tient of ABSD and 4 patients of Mixed SD). Secondary MTD was noted in
To the best of our knowledge no co-relation between SD and vocal 3 patients of ADSD. Flaring of the ala nasi was documented in all pa­
fold sulcus has been discussed in existing literature. Sulcus vocalis is an tients of ABSD and in none of the ADSD and mixed SD group. There were
invagination of the mucosal layer of the vocal fold into the deeper layers, 3 patients of ADSD besides the 106 cases discussed who had associated
creating a groove within the vocal fold [14]. It may be congenital [15] or leukoplakia that were excluded from the study.
acquired which could be due to trauma, infection, rupture of a vocal cyst A statistical analysis of the data compiled was performed using Odds
or a combination of both [16]. With a vocal fold sulcus the two primary ratio where the presence of sulcus was found to be significant (odds
problems are inadequate closure of glottis due to thinning of lamina ratio = 7.37 with C.I. = 2.063–26.35) in the group of SD patients having
propria and a decrease in the vibratory mucosal wave resulting in a an abductor component i.e. group 2 consisting of ABSD and mixed SD,
breathy high-pitched harsh voice with vocal fatigue and hypophonia. when compared to the ADSD group.
However, this is a constant breathiness of the voice unlike the spasms of
breathiness heard in a patient of ABSD. The management of vocal fold 4. Discussion
sulcus is strengthening voice therapy and medialisation procedures,
both of which are typically not associated with success in the case of ABSD is rare, comprising 15% of patients with SD [17]. It is distin­
ABSD. guished from ADSD on the basis of flexible laryngoscostroboscopic
Patients diagnosed as ABSD and mixed SD in our voice clinic were assessment and perceptual analysis of voice. Vocal fold sulcus is also an
occasionally found to have a co-existing sulcus on flexible stroboscopy. uncommon finding and amounts to 8% of patients visiting the voice
Our study is a retrospective observational study performed over a 45- clinic with hoarseness [14].
month period to evaluate the co-existence of vocal fold sulcus in SD. Demographics of SD have been studied in detail by Blitzer et al. [8]
The presence of a sulcus in a patient of SD may be either the result of the and Patel et al. [2]. They mention the average age of onset of symptoms
SD or the etiology of the SD. Our research question was to ascertain if to be 39 years and 51 years respectively, which coincided with the mean
there was a significant association between vocal fold sulcus and age of 47.8 years as seen in our study. Creighton et al. [18] reported that
different types of SD in our patients.
Table 1
2. Materials and methods The distribution of vocal fold sulcus within various types of SD (and any asso­
ciated vocal tremor) in our study.
This was a retrospective study undertaken in the voice clinic of a Type of Number of Sulcus vocalis Vocal Males Females
tertiary hospital where the stroboscopic video recordings and clinical SD SD patients tremor
Bilateral Unilateral
records of all patients diagnosed with SD between January 2016 and (N = 106)
September 2019 were reviewed. Those patients with coexisting neuro­ ADSD 84 3 2 24 43 41
logical disorders and malignancy were excluded. The gender and age of ABSD 10 1 3 1 8 2
the patient, duration of symptoms at presentation and findings of the Mixed 12 3 0 4 11 1
SD
flexible videolaryngostroboscopy were noted. A neurologist’s opinion

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N.K. Nerurkar et al. American Journal of Otolaryngology–Head and Neck Medicine and Surgery 42 (2021) 102940

Cyrus et al. [4] have demonstrated a significant asymmetry in the

thyroarytenoid and cricothyroid activity levels between the two sides in
ABSD patients which may be responsible for phonation onset difficulty
and intermittent loss of voice. They concluded that an asymmetry in
adductor muscle tone between the two sides in ABSD may account for
difficulties with maintaining phonation and voice onset after voiceless
consonants and that these abnormalities may indicate why PCA Botu­
linum toxin injections have not been as effective in ABSD as thyroar­
ytenoid injections have been in ADSD. Ludlow et al. [13] conducted a
study to determine laryngeal muscle activation abnormalities associated
with speech symptoms in ABSD where bilateral laryngeal muscle re­
cordings from the posterior cricoarytenoid, thyroarytenoid, and crico­
thyroid muscles were conducted in 12 ABSD patients and 10 controls.
They concluded that injection of the cricothyroid muscles with botuli­
num toxin was beneficial in those patients of ABSD having abnormal
muscle activation primarily affecting the cricothyroid muscles. Our
second hypothesis is that the asymmetric stimulation of the thyroar­
ytenoid, cricothyroid and PCA muscles in SD may result in atrophy of the
lamina propria and vocalis muscle with resultant sulcus formation.
Fig. 1. Laryngoscopy revealing bilateral vocal fold sulcus in a patient of ABSD.
Furthermore, keeping both our hypothesis in consideration, the group of
patients with sulcus and ABSD may benefit from medialisation thyro­
plasty. Postma et al. [25] have proposed that bilateral medialisation
it took patients an average of 4.43 years (53.21 months) to be diagnosed
laryngoplasty is a safe and effective treatment for patients with glottal
with SD after first going to a physician with vocal symptoms. However,
incompetence, attributable to a wide array of causes. They found that in
the mean duration of symptoms was 6 months at first consultation in our
some patients of ABSD not responding to Botox injections, this treatment
voice clinic which may be an indication of growing awareness regarding
showed significant benefit by preventing the vocal folds from flying
SD. Internationally female preponderance has been documented, as re­
apart as a result of abductor voice breaks. It would be a surgery worth
ported by Adler et al. [19] and Patel et al. [2]. However, in our study
considering especially for the group of patients who have a concurrent
based in a tertiary care hospital in India we encountered 62% males and
Sulcus vocalis with the ABSD or mixed SD.
we attribute this to social bias [20]. ADSD is reported as a more prev­
Whether it is the asymmetric stimulation of the abductor and
alent subtype [6]. This was also seen in our study where 79% cases
adductor intrinsic muscles that results in atrophy of the lamina propria
documented had ADSD. ABSD is rare and was reported in 17% patients
and vocalis muscle or the presence of the sulcus that triggers SD due to
in a study by Blitzer et al. [8] among a total of 901 patients of laryngeal
an altered sensory feedback from the laryngeal mucosa is debatable and
dystonia. In our study we had 9.4% cases with ABSD and 11.3% with
needs to be studied further. The possible sequence of events in SD with
mixed SD. Tanner K et al. [21] have reported the coexistence of vocal
vocal fold sulcus is represented in Fig. 2.
tremors in 29% cases of SD. In our study 27% patients showed a coex­
Though stroboscopy is considered as a gold standard for diagnosis of
isting vocal tremor as detailed in our results. In a study by White et al.
glottic lesions, superficial sulci may be missed during stroboscopy and
[22] which included 146 voice disorder controls and 128 patients with
picked up only during microlaryngoscopic examination under general
SD, 26% patients with SD had vocal tremor and 21% had non-vocal
anaesthesia with palpation [26]. Thus, it is possible that we have missed
tremor. They reported that patients with SD were 2.8 times more
diagnosing shallow sulci in both the groups. Furthermore the numbers of
likely to have co-prevalent tremor than the control group.
ABSD are low due to its rarity resulting in a wide confidence interval.
Our study revealed the presence of vocal fold sulcus in 40% cases of
Though we have found a significant association between the presence of
ABSD, 25% cases of Mixed SD and in 5.95% cases of ADSD. A statistical
vocal fold sulcus and ABSD/mixed SD, the two hypothesis proposed
significance was found between patients of SD having an abductor
need further research to validate them. These are the limitations of our
component (ABSD and mixed SD) and vocal fold sulcus. In a study by
study.
Patel et al. [23] on concurrent laryngeal abnormalities in patients with
paradoxical vocal fold dysfunction, vocal fold sulcus was noted in 1 out
5. Conclusion
of a total of 30 patients. Tanner et al. [21] in a case control study
comparing voice disorders and risk factors for SD did not encounter any
Our study revealed a significant association between patients of SD
patient with a finding of vocal fold sulcus. Sulcus vocalis has been
having an abductor component (ABSD and mixed SD) and vocal fold
postulated to alter the glottic airflow and contribute in the formation of
sulcus. The mutual influences of sulcus and ABSD need to be further
vocal fold polyps in a study by Carmel-Neiderman et al. [14]. It is
studied for clarity in etiology and optimization of treatment in SD with
possible that this altered glottic airflow provides an altered sensory
an abductor component.
feedback from the laryngeal mucosa to the central control centre trig­
gering the onset of SD. In SD patients, the lack of symptoms during
Financial disclosure
whispering when the vocal folds are not vibrating suggests that changes
in laryngeal sensory feedback either from the vocal fold mucosa or
This research did not receive any specific grant from funding
subglottal pressures in the trachea may play a role in the pathophysi­
agencies in the public, commercial, or not-for-profit sectors.
ology of the disorder [5]. Bhabu et al. [24] have demonstrated initiation
of adductor response by air stimulus to laryngeal mucosa thereby
CRediT authorship contribution statement
proving the role of mucosal mechanoreceptors in dynamic sensory
feedback to the Central Nervous System. The presence of the vocal fold
All authors have contributed in the research of this manuscript titled
sulcus prior to the onset of SD in such a hypothesis would be essential,
“Sulcus Vocalis in Spasmodic Dysphonia- A Retrospective Study”. The
however at presentation our patients had SD for a variable period (as
first (senior) author has been responsible for the conception, design,
determined by the history) and the diagnosis of the sulcus was incidental
analysis and writing of the paper. The second and third authors have
on flexible videostroboscopy.
contributed in collecting data, analysis and writing the paper.

3
N.K. Nerurkar et al. American Journal of Otolaryngology–Head and Neck Medicine and Surgery 42 (2021) 102940

Fig. 2. Possible sequence of events in SD with vocal fold sulcus.

Declaration of competing interest [13] Ludlow CL, Naunton RF, Terada S, Anderson BJ. Successful treatment of selected
cases of abductor spasmodic dysphonia using botulinum toxin injection.
Otolaryngology–Head and Neck Surgery 1991 Jun;104(6):849–55.
None. [14] Carmel-Neiderman NN, Wasserzug O, Ziv-Baran T, Oestreicher-Kedem Y.
Coexisting vocal fold polyps and sulcus vocalis: coincidence or coexistence?
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