Partial Epiglottoplasty for Pharyngeal Dysphagia due to Cervical

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Original Research—Laryngology and Neurolaryngology
Partial Epiglottoplasty for Pharyngeal
Dysphagia due to Cervical Spine
Pathology
Otolaryngology–
Head and Neck Surgery
2015, Vol. 153(4) 586–592
Ó American Academy of
Otolaryngology—Head and Neck
Surgery Foundation 2015
Reprints and permission:
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DOI: 10.1177/0194599815601025
http://otojournal.org
Nausheen Jamal, MD1, Andrew Erman, MS2, and
Dinesh K. Chhetri, MD3
Sponsorships or competing interests that may be relevant to content are disclosed at the end of this article.
Abstract
Objective. To examine the role of epiglottoplasty in patients
with pharyngeal dysphagia due to pharyngeal crowding from
cervical spine pathology and to assess swallowing outcomes
following epiglottoplasty.
Study Design. Retrospective case series.
Setting. Academic tertiary care medical center.
Subjects and Methods. Dysphagia can occur in patients with
cervical spine pathology because of hypopharyngeal crowding. Swallowing studies, such as modified barium swallow
study and fiberoptic endoscopic evaluation of swallowing,
may demonstrate a nonretroflexing epiglottis owing to cervical spine osteophytes or hardware, thus impeding pharyngeal bolus transit. We performed partial epiglottoplasties in
a series of these patients. A retrospective review of swallowing outcomes was performed to assess the efficacy of
this surgery in this patient population.
Results. Epiglottic dysfunction causing dysphagia due to cervical spine pathology was diagnosed by modified barium swallow study and/or fiberoptic endoscopic evaluation of
swallowing in 12 patients. Findings included hypopharyngeal
crowding because of cervical osteophytes (n = 8) or cervical
hardware (n = 4) associated with absent epiglottic retroflexion and retained vallecular residue. Partial epiglottoplasty
resulted in significant reduction of vallecular residue and a
significant increase in functional swallow outcomes without
an increase in swallow morbidity.
Received April 1, 2015; revised June 22, 2015; accepted July 27, 2015.
S
wallowing is a highly complex neuromuscular event
that is composed of 3 phases: oral, pharyngeal, and
esophageal. Dysphagia may occur with disruption or
inefficiency at any 1 of these phases and is a common
reason for consultation with an otolaryngologist. Frequently
encountered causes of dysphagia include stroke, neurodegenerative disorders, chemoirradiation, cricopharyngeal dysfunction, malignancies, and esophageal stenosis.1 A less
commonly diagnosed cause of dysphagia is pharyngeal
crowding from cervical spine pathology. Anterior cervical
osteophytes (ACOs) are present in 10.6% of patients presenting with dysphagia and are thought to cause problems
in the pharyngeal and esophageal phases of swallow.2
Pharyngeal-phase dysphagia caused by ACOs is typically
due to pharyngeal narrowing from a protuberant posterior
pharyngeal wall with concurrent alteration of swallow physiology, including reduction or absence of epiglottic retroflexion, leading to attenuation of intrabolus pressures above
the obstructing epiglottis and consequently resulting in vallecular residue.2-4 The same finding is often noted in patients
who have undergone cervical spine surgery with hardware
placement. Irritation of the cricopharyngeus muscle by the
ACOs may lead to cricopharyngeus muscle spasm and dysphagia.4,5 Unfortunately, resection of cervical osteophytes
may lead to worsening dysphagia and can be fraught with
other complications, including pharyngeal, esophageal, and
laryngeal nerve injury.2,6 Furthermore, ACOs typically occur
in the elderly population, in whom age-related swallow
Conclusion. There is a role for partial epiglottoplasty in patients
with dysphagia attributed to hypopharyngeal crowding from
cervical spine pathology. Surgery enables reduced vallecular
residue and improved functional swallowing outcomes.
1
Department of Otolaryngology–Head and Neck Surgery, Temple University School of Medicine, Philadelphia, Pennsylvania, USA
2
Departments of Audiology and Speech, David Geffen School of Medicine
at the University of California, Los Angeles, Los Angeles, California, USA
3
Department of Head and Neck Surgery, David Geffen School of Medicine
at the University of California, Los Angeles, Los Angeles, California, USA
Keywords
dysphagia, cervical osteophyte, epiglottoplasty, epiglottidectomy, epiglottic dysfunction, diffuse idiopathic skeletal hyperostosis, Forestier’s disease
Corresponding Author:
Nausheen Jamal, MD, Department of Otolaryngology–Head and Neck
Surgery, Temple University School of Medicine, 3440 North Broad St,
Kresge West #300, Philadelphia, PA 19140, USA.
Email: nausheen.jamal@tuhs.temple.edu
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Jamal et al
587
Table 1. Grading of Vallecular and Pyriform Sinus Residue.11,a
0
1
2
3
4
5
None to trace coating
Thick coating
Up to 50% full of residual
Greater than 50% full of residual
Overflowing with residual
Oropharyngeal stasis (entire bolus stays within vallecula)
a
Reproduced with permission from: Jamal N, Erman A, Chhetri DK.
Transoral partial epiglottidectomy to treat dysphagia in post-treatment head
and neck cancer patients: a preliminary report. Laryngoscope. 2014;124:665671. Ó 2013 The American Laryngological, Rhinological and Otological
Society, Inc.
Figure 1. Example of the endoscopic appearance of a patient with
anterior cervical osteophytes resulting in protuberance of the posterior pharyngeal wall. Fiberoptic endoscopic evaluation of swallowing was performed in this patient, resulting in vallecular residue.
changes, such as depletion of pharyngeal strength and intrabolus pressure generation, contribute to baseline worsening of
the swallow. Furthermore, the presence of other comorbidities may not allow for a major operation, and conservative
therapy (eg, diet modification and swallow therapy) may not
be adequate to improve quality of life.4
Although present in up to a third of the elderly population, ACOs are typically asymptomatic until they reach a
size that disrupts pharyngeal or esophageal swallow.2-4,7 For
those patients who present to a physician with symptomatic
ACOs, the most common presenting symptom is dysphagia.2-4,7-10 In evaluating our dysphagia population, we have
observed on endoscopic evaluation a number of patients
who present with a narrow oropharynx and hypopharynx
and fullness of the midline posterior pharyngeal wall.
Swallow testing with fiberoptic endoscopic evaluation of
swallowing (FEES) or modified barium swallow study
(MBSS) demonstrated that in these patients, swallow efficiency was at least partly impeded by lack of epiglottic retroflexion owing to collision with the protuberant posterior
pharyngeal wall from osteophytes or cervical spinal hardware (Figure 1). We performed partial epiglottoplasty in
several patients with cervical spine pathology (ACOs or
hardware) with good results and seek to report our preliminary findings. We hypothesized that this would address the
pharyngeal narrowing and enable physiologic epiglottic retroflexion, thereby increasing swallow efficiency and
decreasing pharyngeal residue, as noted when a similar
operation was performed in chemoirradiated head and neck
cancer patients who developed epiglottic dysfunction and
marked dysphagia.11
Methods
This retrospective study was approved by the Medical
Internal Review Board of the University of California, Los
Angeles. Medical records were reviewed to identify all
consecutive patients with dysphagia who underwent partial
epiglottoplasty during a 15-month period. Criteria for inclusion in the study were as follows: patient complaint of dysphagia (with or without need for diet modification), limited
or absent epiglottic inversion noted on MBSS or FEES, and
presence of cervical spine pathology (ACOs or cervical
spine hardware), as noted on radiographic or endoscopic
studies. The subjective nature of the dysphagia was typically
described by patients as a ‘‘sticking’’ sensation. Criteria for
exclusion were as follows: history of treatment for head and
neck cancer, stroke, vocal fold immobility, and lack of preand postoperative MBSS or FEES.
When .1 swallow evaluation was available for review,
we used the last study before surgery and the first study
after surgery (typically performed 1 month postoperatively
to account for recovery from surgical trauma). Whenever
available, the same study type was used for pre- and postoperative comparison (ie, FEES compared with FEES and
MBSS with MBSS). Pre- and postoperative MBSS and/or
FEES studies were compared to assess for the following: (1)
amount of vallecular residue after the first swallow, as an
indicator of the amount of obstruction provided by the dysfunctional epiglottis (Table 1); (2) pharyngeal phase time,
defined as the time taken from initiation of the pharyngeal
phase (bolus entering the pharynx) until cessation of volitional bolus clearance; and (3) degree of penetrationaspiration as defined by Rosenbek et al.12 Patients served as
their own controls (pre- vs postoperative) in this comparison. A single consistent texture and amount of food was
used for each subject to compare pre- and postoperative
swallows (typically, 0.5 or 1 tsp of puree).
Office notes, FEES reports, and MBSS reports were
reviewed by a speech and language pathologist (A.E.) who
was not involved in the surgery, to assess pre- and postoperative functional outcome swallowing scale (FOSS)
scores, pneumonia history, diet recommendations, and subjective change in the swallow following surgery.13
Suprahyoid epiglottoplasty was performed via a transoral
approach under general anesthesia as previously described.11
In brief, the epiglottis was trimmed halfway between the
hyoepiglottic ligament and the tip of the epiglottis, with the
use of the carbon dioxide laser. All patients tolerated the
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588
Otolaryngology–Head and Neck Surgery 153(4)
Table 2. Demographic Data.
Swallow Study
Patient
1
2
3
4
5
6a
7
8
9
Age, y
85
82
70
83
70
62
78
85
84
Sex
F
M
M
M
F
M
M
M
M
Cervical Spine Pathology
Preoperative
Postoperative
Postoperative Follow-up, mo
Osteophytes
Osteophytes
Anterior cervical spine surgery
Anterior cervical spine surgery
Anterior cervical spine surgery
Osteophytes
Osteophytes
Osteophytes
Osteophytes
MBSS
FEES
MBSS
MBSS
MBSS
FEES
MBSS
MBSS
MBSS
MBSS
MBSS
MBSS
MBSS
MBSS
FEES
MBSS
FEES
FEES
3
11
6
8
4
1
1
7
1
Abbreviations: FEES, fiberoptic endoscopic evaluation of swallowing; MBSS, modified barium swallow study.
a
Also underwent laser-assisted endoscopic cricopharyngeal myotomy at the time of epiglottidectomy to address cricopharyngeal dysfunction diagnosed on
preoperative swallow evaluation. This patient had also undergone posterior spinal surgery (laminectomy). Subsequent procedures for dysphagia were not performed in any patient as of latest follow-up.
procedure well. There was 1 complication of a postoperative
pneumonia in this patient population. Patients were discharged home the same day, on the diet that they were previously tolerating.
Statistical analysis was performed with the Wilcoxon
signed-rank test to assess for significance in the differences
between pre- and postoperative data.
Results
Epiglottic dysfunction (ie, lack of normal epiglottic retroflexion) causing symptoms of dysphagia due to cervical spine
pathology was diagnosed by MBSS and/or FEES in 12
patients. A total of 9 patients fitting the inclusion and exclusion criteria underwent epiglottoplasty in the specified time
frame (Table 2). Three patients were excluded because of a
history of stroke, vocal fold immobility, or lack of appropriate pre- or postoperative swallow studies. Seven patients
were men and 2 were women, ranging in age from 62 to 85
years (median, 82). Length of follow-up ranged from 1 to 11
months. All patients presented with a complaint of dysphagia
and were diagnosed with cervical spine abnormalities. Two
men and 1 woman had undergone anterior cervical spine surgery causing retropharyngeal bulging and oropharyngeal narrowing prior to onset of dysphagia. The remaining patients
were diagnosed with ACOs contributing to dysphagia.
Preoperative vallecular residue scores ranged from 0 to 3
(Table 3). Except for 1 patient whose preoperative score was
0 and did not change after surgery, all patients demonstrated
a reduction in vallecular residue postoperatively, with an
average score reduction of 1.8 (P \ .05). Penetration-aspiration scores (PASs) were unchanged postoperatively in 3
patients, with score improvement in 6 patients. Overall, the
average change in PAS following surgery was a score
improvement of 1.6 (P \ .05). This was presumably due to
improved or resolved epiglottic dysfunction. Notably, no
patients demonstrated worsening of their penetration-aspiration
status following epiglottoplasty. Similarly, the average change
in FOSS was a significant score improvement of 1.2 points (P
\ .05), with score improvement noted in 8 patients and no
change noted in 1 patient.
Pharyngeal phase time was calculated for the 6 patients
who underwent MBSS pre- and postoperatively (Table 4).
Four patients (patients 3, 5, 7, 9) demonstrated reduction in
their pharyngeal phase times by approximately one half, and
1 patient’s time (patient 1) was essentially unchanged (4%
reduction). Patient 4’s pharyngeal phase time, however,
demonstrated a marked increase. This patient had improvement in vallecular residue as well as FOSS scores but performed extra swallows to clear the residue, thus increasing
pharyngeal transit time. The average time change was a
reduction of 2.4 seconds, but this did not reach statistical
significance despite improvement in the majority (P = .5).
Five patients had a preoperative history of aspiration pneumonia (Table 3). At the time of latest follow-up, 1 of these 5
reported an episode of postoperative pneumonia 1 week after
surgery. This was the only noted complication. However,
MBSS performed about 1 month after surgery revealed
improvement in swallowing parameters without evidence of
penetration or aspiration. This patient’s swallowing on FEES
continued to improve over the next 3 to 6 months.
Seven patients continued their preoperative diet type,
while 2 were able to advance their diets. Of these 2, 1 was
nil per os prior to surgery and was advanced to a puree
diet. The other patient was advanced from a soft diet to a
regular diet. When asked the question ‘‘Do you feel that
swallowing has become easier?’’ all patients responded in
the affirmative, describing an improvement in the ease of
swallow and reduction of the previously described ‘‘sticking’’ sensation.
Discussion
ACOs are believed to occur in 20% to 30% of the general
population, although the rate of cervical vertebral modification
may be higher than 75% in the 651 age population.2,4,7 The
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Jamal et al
589
Table 3. Pre- and Postoperative Residue, PAS, FOSS Results, and Patient Subjective Reports.
Patient
Subjective improvement?
Diet
Postoperative
Preoperative
Aspiration pneumonia
Postoperative
Preoperative
FOSS
Postoperative
Preoperative
PAS
Postoperative
Preoperative
Vallecular residue score
Postoperative
Preoperative
1
2
3
4
5
6
7
8
9
Yes
Yes
Yes
Yes
Yes
Yes
Yes
Yes
Yes
Soft
Soft
Regular
Regular
Regular
Regular
Regular
Soft
Soft
Soft
Regular
Regular
Puree
NPO
Regular
Regular
Regular
Regular
No
No
No
No
No
No
No
Yes
No
Yes
No
Yes
No
Yes
No
No
Yes
Yes
2
3
1
1
1
2
1
3
2
4
0
1
2
4
0
1
2
3
2
4
1
1
1
1
1
1
2
3
1
6
3
6
1
2
3
5
0
3
0
2
0
2
1
3
0
3
0
0
1
3
0
1
1
2
Abbreviations: FOSS, functional outcome swallowing scale; NPO, nil per os; PAS, penetration-aspiration score.
Table 4. Pharyngeal Phase Time Measurements.
Pharyngeal Phase Time, s
Patient
1
3
4
5
7
9
Preoperative
Postoperative
Change, %
13
9.2
2.5
11.7
11
11.8
12.5
4.3
10.2
5.8
5.9
6
–4
–53
308
–50
–46
–49
most common causes of ACOs are degenerative spine conditions and diffuse idiopathic skeletal hyperostosis (DISH),
which are also the etiologies thought to lead to osteophyterelated dysphagia.1 In the majority, these ACOs are asymptomatic. However, when they become symptomatic—typically
once the osteophyte reaches a size of at least 10 mm2—solid
food dysphagia is the most common presenting symptom,
affecting 17% to 28% of patients with DISH.4,5,8 The cervical
vertebral levels most often involved in this population are C5C6 (40%), followed by C4-C5 (23%) and then C3-C4 and C2C3 (14% each).1,7,9
The logical way to address bony overgrowths of the cervical spine would be to surgically remove the osteophytes.
Indeed, after failure of conservative therapy, an anterolateral
transcervical approach to a partial anterior vertebral corpectomy is the gold standard in addressing cervical osteophytes.2,8
In appropriately selected patients, the rate of swallow improvement following osteophytectomy with return to regular diet is
reportedly 77% to 100%.3,7,10 Unfortunately, ACO caused by
DISH has the potential for recurrence because of an average
bony regrowth rate of 1 mm per year.4 Additionally, a study
by Carlson et al noted postoperative postswallow pharyngeal
residue in 62% following osteophytectomy, with silent aspiration in 8%.3
For some patients, diet modification and swallow therapy
are either not sufficient for symptomatic improvement or unsatisfying from a quality-of-life perspective.4 Unfortunately,
many of these patients are not ideal candidates for a major surgical intervention, typically because of age and other comorbidities. Furthermore, the risks of osteophytectomy, even in a
healthy patient, cannot be discounted. Documented risks
include hematoma with possible airway compromise, injury to
the superior and recurrent laryngeal nerves, injury to the marginal mandibular branch of the facial nerve, hypoglossal nerve
injury, cervical sympathetic chain injury, esophageal perforation, salivary fistula formation, vertebral artery injury, and
even death.2,7 In a retrospective review of 71 patients who
underwent single- or dual-level cervical corpectomies for spondylosis or osteophytosis with titanium mesh or cage placement,
there was a 25% complication rate. The most significant complications were failure of osteosynthesis, hardware subsidence,
incorrect screw placement, and dysphagia related to hardware
placement requiring reoperation (2 patients). Five patients
experienced permanent dysphagia and dysphonia postoperatively. Of note, this study excluded patients who had undergone 3-level corpectomy due to their ‘‘high rate [of]
morbidity.’’6 This high complication rate and lack of dysphagia improvement after corpectomy are in line with the senior
author’s clinical experience (D.K.C.). Thus, we have sought
alternative ways to treat dysphagia in this population.
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590
Otolaryngology–Head and Neck Surgery 153(4)
Figure 2. Preoperative modified barium swallow study for patient
3, demonstrating osteophytes. Note that the epiglottis (arrow)
abuts the osteophytes without retroflexing, causing bolus obstruction at the vallecular level.
Figure 3. Postoperative modified barium swallow study for patient
3, at the same point of the pharyngeal swallow as Figure 2. Note
lack of pharyngeal residue. The epiglottic remnant (arrow) is no
longer attempting to retroflex against osteophytes.
Review of baseline swallow and radiographic studies in a
series of our patients presenting with dysphagia demonstrated the presence of either ACO or previously placed cervical hardware, with associated pharyngeal narrowing and
reduction of epiglottic mobility, often leading to vallecular
residue (Figure 2). In those patients who either were not
candidates for a major spinal operation or were unwilling to
undergo (further) spinal surgery, we proposed partial epiglottoplasty. We hypothesized that this would address the
pharyngeal narrowing and enable physiologic epiglottic retroflexion, thereby increasing swallow efficiency and
decreasing pharyngeal residue, as noted when a similar
operation was performed in chemoirradiated head and neck
cancer patients who developed epiglottic dysfunction, leading to marked dysphagia.11
Indeed, we did note improvement in a number of objective
and subjective measures of swallow. Compared with preoperative findings, scoring of vallecular residue, penetrationaspiration, and functional swallow outcome showed significant improvement after surgery. Based on observations from
MBSS, the reduced vallecular and pyriform sinus residue
appears to be a direct result of improved epiglottic motion
without collision with (and obstruction by) the cervical spine
protuberance (Figure 3). Rates of aspiration were not
increased by performing partial epiglottoplasty, as demonstrated by the significant improvement in PAS following surgery and no increase in PAS in any patient. Pharyngeal
efficiency calculations based on pharyngeal phase times
demonstrated improvement in at least 4 of the 6 patients in
whom MBSS data were available. One patient developed an
episode of aspiration pneumonia postoperatively. Interestingly,
his postoperative MBSS (performed 1 month after surgery)
revealed no evidence of penetration or aspiration, suggesting
that despite this complication, he quickly recovered from a
deglutition standpoint. Four other patients with documented
preoperative aspiration pneumonia history remained
pneumonia-free at latest follow-up. Two of these patients were
actually able to advance their diets, 1 of whom was able to
transition from nil per os status to a puree diet by mouth. In
addition, all patients reported subjective improvement in swallowing after epiglottoplasty.
There are a number of weaknesses in this study. First, it
is a retrospective review of a small case series. Cervical
spine pathology is not uncommon, but it is usually asymptomatic. Although dysphagia is believed to be the most
common presenting symptom, most reports of dysphagia
associated with cervical spine pathology in the literature are
case reports of 1 to 3 patients, and all refer to osteophytectomy as the only available surgical treatment option.2,4,5,8,9
Therefore, despite our small number of patients, we believe
this to be a relatively large series of patients who have
undergone a treatment paradigm that has not previously
been described in the literature.
Another weakness is that not all patients underwent both
pre- and postoperative MBSS. For example, when FEES
performed in the office at follow-up visits demonstrated
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Jamal et al
591
improved swallow function, either patients were not routinely referred for a repeat postoperative MBSS to confirm
these findings, or the patients themselves chose not to
undergo follow-up MBSS. However, other dysphagia
experts have argued that these 2 studies demonstrate good
agreement with regard to penetration, aspiration, and pharyngeal residue, which were the primary treatment end
points considered in this study.14
Although about half of the patients in this series achieved
a postoperative FOSS score of 0 or 1 (regular diet with or
without episodic dysphagia symptoms), the other half did
not improve beyond a FOSS score of 2 (significant diet
modification). However, the latter patients improved their
FOSS score by at least 1 point. Not surprising, this suggests
that although partial epiglottoplasty helped improve their
swallow to some degree, other causes of dysphagia were
present. These were naturally not adequately addressed by
epiglottoplasty alone. Indeed, dysphagia is typically multifactorial in nature.
Given the advanced age range of this group (seventh to
ninth decades), another likely contributor to disordered swallowing is lingual and pharyngeal weakness, which leads to
diminished intrabolus pressure generation. This aspect was
not adequately examined in this review, although some
patients underwent some form of swallow therapy (with limited benefit) prior to consideration of surgery. Unfortunately,
manometry was not used in this patient cohort; this would
have been helpful in evaluation of pharyngeal strength and
assessing for possible improvement in postoperative intrabolus pressures.
Another possibility in this population is that the cervical
spine pathology may have also affected the esophageal phase
of swallow, as suggested by other authors.2 This aspect was
not adequately investigated in this group of patients.
Follow-up in this preliminary study was limited. However,
the postoperative swallow study in the majority of patients
was performed 1 to 2 months postoperatively, which allowed
for surgical outcome comparison at a similar time point with
relation to recovery. With further follow-up, swallow outcomes may change. For patient 4, by way of example, an
MBSS performed 2 months postoperatively was used to
assess postsurgical swallow outcome. However, a follow-up
FEES was performed 8 months after surgery, which demonstrated a safe and efficient swallow without any pharyngeal
residue whatsoever. It has been our experience that following
removal of pharyngeal obstruction, pharyngeal muscle
strength may improve over the subsequent 3 to 6 months,
leading to continuous, gradual improvement in one’s swallow
efficiency, which has been shown.15
Despite these drawbacks, objective and subjective assessment of swallow in these patients suggests a reduction of
dysphagia that is not attributable to other causes. Although
this is a preliminary study, we believe that in a select group
of individuals—those with cervical spine pathology causing
dysphagia related to epiglottic dysfunction—partial epiglottoplasty may be a treatment option.
Conclusion
Cervical spine pathology is an often-overlooked etiology of
dysphagia, despite its presence in .10% of patients with
this complaint. Although the primary way to address spinal
pathology is by performing spinal surgery, this is not an
option for all patients. For a select group of patients with
dysphagia due to cervical spine pathology as well as epiglottic dysfunction, partial epiglottoplasty may be an option.
Surgery in these patients may improve postswallow vallecular residue and functional swallow outcomes.
Author Contributions
Nausheen Jamal, study conception; acquisition, analysis, and
interpretation of data; drafting of work; final manuscript approval;
accountable for all aspects of work; Andrew Erman, analysis and
interpretation of data; drafting of work; final manuscript approval;
accountable for all aspects of work; Dinesh K. Chhetri, study conception; acquisition, analysis, and interpretation of data; drafting of
work; final manuscript approval; accountable for all aspects of
work.
Disclosures
Competing interests: None.
Sponsorships: None.
Funding source: National Institutes of Health (RO1 DC011300).
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