International Journal of Pediatric Otorhinolaryngology 82 (2016) 38–42

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International Journal of Pediatric Otorhinolaryngology

journal homepage: www.elsevier.com/locate/ijporl

Swallowing dysfunction among patients with laryngeal cleft: More

than just aspiration?
Julie E. Strychowsky a,c,*, Pamela Dodrill a, Ethan Moritz a, Jennifer Perez a, Reza Rahbar a,b
a
Department of Otolaryngology and Communication Enhancement, Boston Children’s Hospital, 333 Longwood Ave, 3rd Floor, Boston, MA 02115, USA
b
Department of Otology and Laryngology, Harvard Medical School, Boston, MA, USA
c
Department of Otolaryngology-Head and Neck Surgery, University of Western Ontario, 800 Commissioners Rd E, VH B3-444, London, ON, N6A 5W9, Canada

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

Article history: Background: The Modified Barium Swallow (MBS) is the most widely utilized instrumental assessment of
Received 12 June 2015 swallowing disorders in children; however, the exact role in the evaluation of laryngeal clefts remains
Received in revised form 30 November 2015 controversial.
Accepted 28 December 2015
Methods: This study was an IRB-approved retrospective review on patients diagnosed with laryngeal
Available online 7 January 2016
cleft from 2002 to 2014. The objective was to describe the range of swallowing dysfunction that may be
present in patients with laryngeal clefts both pre- and post-intervention (conservative management
Keywords:
versus surgery). A speech-language pathologist reviewed MBS studies and medical records to determine
Laryngeal cleft
Penetration–Aspiration Scale (PAS) and Functional Oral Intake Scale (FOIS) scores.
MBS
Modified barium swallow Results: One hundred seventy-five patients who underwent laryngeal cleft repair during the study
Swallow study period (type 1, n = 111; type 2, n = 54; type 3, n = 9; type 4, n = 1) were included. Fifty patients who were
Dysphagia managed conservatively (type 1) were also included. Swallowing impairment was demonstrated in all
phases of swallowing for all cleft types. Oral phase impairment ranged from 27–67% pre-intervention to
19–75% post-intervention, triggering impairment from 24–42% pre-intervention to 24–75% post-
intervention, and pharyngeal phase impairment (laryngeal penetration and aspiration) from 57–100%
pre-intervention to 40–100% post-intervention. Laryngeal penetration and aspiration on thin and thick
liquids, silent aspiration, PAS, and FOIS scores are reported. Significant improvements in swallowing
function (p < 0.05) were documented in all of the conservatively and surgically managed sub-groups.
Conclusions: The MBS study is a useful tool for evaluating swallowing function in patients with laryngeal
cleft and provides information beyond the lack or presence of aspiration. Understanding impairments in
all phases of swallowing may be beneficial for perioperative management.
ß 2016 Elsevier Ireland Ltd. All rights reserved.

1. Introduction laryngeal clefts may present with airway and/or swallowing

Laryngeal clefts are rare congenital anomalies that result from
failure of fusion of the tracheoesophageal septum or two lateral
growth centers of the posterior-cricoid cartilage during embryo-
logical development [1–3]. The annual incidence ranges from 1 in
10,000 to 1 in 20,000 live births, with a male predominance
[4]. Most cases are sporadic while others associated with
syndromes such as Opitz-Frias or Pallister-Hall, or congenital
anomalies such as tracheoesophageal fistulas [5]. Patients with
* Corresponding author. Tel.: +1 519 685-8242, Fax: +1 519 685-8185.
E-mail addresses: julie.strychowsky@lhsc.on.ca (J.E. Strychowsky),
pamela.dodrill@childrens.harvard.edu (P. Dodrill), emoritz@wesleyan.edu
(E. Moritz), jennifer.perez@childrens.harvard.edu (J. Perez),
reza.rahbar@childrens.harvard.edu (R. Rahbar).
impairments, which can lead to recurrent aspiration pneumonia,
respiratory distress, and failure to thrive [1–3]. Therefore, appro-
priate and timely diagnosis is paramount.
Management of laryngeal clefts ranges from conservative
monitoring to surgical intervention. Both groups generally require
feeding therapy input to address potential swallowing impairments.
Feeding therapy techniques may involve skill building activities (e.g.
oral-motor therapy) and/or teaching the patient or their care-givers
how to use compensatory strategies, such as the use of modified
liquids/foods (e.g. thickened liquids, pureed foods), modified feeding
equipment (e.g. slower or faster flow bottle nipples, open cups vs
straw cup), modified feeding positioning (e.g. upright vs reclined)
and modified feeding strategies (e.g. having the feeder actively pace
the feed, having the child take single sips from a straw vs sequential
sips). It is the role of the Otolaryngologist, with input from the

http://dx.doi.org/10.1016/j.ijporl.2015.12.025
0165-5876/ß 2016 Elsevier Ireland Ltd. All rights reserved.
 J.E. Strychowsky et al. / International Journal of Pediatric Otorhinolaryngology 82 (2016) 38–42
speech-language pathologist, to determine whether a patient would
best be managed through feeding therapy alone or if they also
require surgical repair of their cleft.
There is a lack of standardized evaluation for the diagnosis of
laryngeal cleft [1–3,6–9]. Diagnostic practices include history and
physical examination, chest X-ray, swallowing assessment (modi-
fied barium swallow [MBS] and/or fiberoptic endoscopic evalua-
tion of swallowing [FEES]), flexible fiberoptic laryngoscopy, and
the gold standard of operative endoscopy to gently palpate the
interarytenoid area. Other diagnostic adjuncts include measure-
ment of lipid laden macrophage levels obtained by bronchial
alveolar lavage [10] and interarytenoid injection [11–13]. The MBS
(also known as video fluoroscopic swallow study, VFSS) is the most
widely utilized tool for the assessment of swallowing disorders in
children [14,15]; however, the exact role in the diagnosis and
monitoring of laryngeal clefts remains controversial.
The purpose of this study was to describe the range of
swallowing dysfunction that may be present in patients with
laryngeal clefts both pre- and post-intervention (conservative
management versus surgery).
2. Materials and methods
A retrospective review of a prospective database on all patients
diagnosed with laryngeal cleft at Boston Children’s Hospital was
performed. Institutional review board approval was obtained. All
patients who were diagnosed with laryngeal cleft from 2002 to
2014 by the senior author (R.R.) and had MBS studies performed
were included. A speech language pathologist (P.D.) reviewed all of
the MBS studies to classify types of swallowing impairments and
determined Penetration–Aspiration (PA) scale [16] scores and
Functional Oral Intake Scale (FOIS) [17] scores for each MBS study.
The original reports from the MBS studies were used for data
collection and when this data was insufficient, the radiologic
examination was reevaluated.
MBS swallowing impairments were classified as follows: oral
phase impairment, swallow triggering impairment, and pharyn-
geal phase impairment (including laryngeal penetration on thin
liquids, laryngeal penetration on thickened liquids, aspiration on
thin liquids, aspiration on thickened liquids, and silent aspiration).
Laryngeal penetration was defined as food or liquid penetrating the
laryngeal inlet above the level of the vocal folds, whereas
aspiration was defined as food/liquid passing below the vocal
folds. For those who underwent surgery, the MBS study that
preceded surgical repair was analyzed. In addition, where
available, post-operative MBS studies at or around 4 months
post-surgery were analyzed for comparison (a four-month time-
frame was used to allow for adequate surgical healing and
assumed swallowing retraining). For those who did not undergo
surgical management, the first MBS available was used as a
baseline measure. A subsequent study at or around 4 months post-
initial assessment was used for a similar comparison.
PA and FOIS scores were determined based on the findings and
recommendations at the time of each MBS study. Rosenbek and
colleagues developed the PA scale in 1996 [16]. It is an 8-point
validated multidimensional assessment tool for swallowing
impairment that relies on the classification of the depth to which
material passes into the airway and whether or not it is expelled
(Table 1). The FOIS pediatric scale was adapted by Crary and
colleagues [17] from an existing adult tool [18]. It is a 7-point
ordinal scale that documents the functional intake of food and
liquid in patients (Table 2).
Data from patient medical records was extracted by one
member of the research team (E.M.) and included: patient
demographics, type of cleft, medical comorbidities, symptomatol-
ogy, feeding history, and number of clinic visits and MBS studies.
39
Table 1
Laryngeal Penetration–Aspiration (PA) Scale [16].
1 Material does not enter airway
2 Material enters the airway, remains above the vocal folds, and is ejected
from the airway
3 Material enters the airway, remains above the vocal folds, and is not
ejected from the airway
4 Material enters the airway, contacts the vocal folds, and is ejected from
the airway
5 Material enters the airway, contacts the vocal folds, and is not ejected
from the airway
6 Material enters the airway, passes below the vocal folds, and is ejected
into the larynx or out of the airway
7 Material enters the airway, passes below the vocal folds, and is not
ejected from the trachea despite effort
8 Material enters the airway, passes below the vocal folds, and no effort is
made to eject
The diagnosis of laryngeal cleft was confirmed by palpation of
the interarytenoid area during direct laryngoscopy by the senior
author (R.R.). The type of laryngeal cleft was defined by the
Benjamin–Inglis classification system, which describes type 1 as an
interarytenoid defect to the level of the vocal folds, type 2 as partial
extension through the posterior cricoid cartilage, type 3 as
extension completely through the posterior cricoid cartilage and
possible extension into the cervical trachea, and type 4 as
extension into the intrathoracic trachea [19].
Statistical analysis was performed (P.D. and K.K.) using the SPSS
Statistics for Windows (Version 19, SPSS Inc., Chicago, IL). Chi-
square analysis was used to analyze trends in dichotomous
measures (e.g. presence/absence of oral phase impairment,
pharyngeal phase impairment, laryngeal penetration, aspiration).
Kruskal Wallis and Mann Whitney U analyses were used to
compare non-parametric measures (e.g. PA scale and FOIS scores).
t-Tests and ANOVAs were used to compare parametric measures
(e.g. number of MBS studies, ORL visits, pneumonias). A p-
value < 0.05 was considered statistically significant.
3. Results
One hundred seventy-five patients underwent laryngeal cleft
repair during the study period (type 1, n = 111; type 2, n = 54; type
3, n = 9; type 4, n = 1), while 50 patients with laryngeal cleft were
managed conservatively with feeding therapy and compensatory
strategies (type 1 only). Pre-operative MBS studies were available
for 138 surgical patients (79%) (type 1, n = 98; type 2, n = 37; type 3,
n = 3; type 4, n = 0) and all conservatively managed patients.
Patient demographics and comorbidities are detailed in Table 3.
MBS swallowing impairments varied by cleft sub-type and for
pre- versus post-surgery (Table 4). All laryngeal cleft sub-types
demonstrated some degree of oral phase impairment, triggering
impairment, and pharyngeal phase impairment (including laryn-
geal penetration, aspiration, and silent aspiration). Interestingly,
within these groups, some patients did not demonstrate any
Table 2
Functional oral intake scale (FOIS) for infant/toddler [17] Table 14-8.
1 Nothing by mouth
2 Tube dependent, with minimal attempts at liquids/food
3 Tube dependent, with consistent intake of liquids/food
4 Total oral diet, but requiring modified liquids  compensations*
4.5 Total oral diet, but requiring modified solids  compensations*
5 Total oral diet, without special preparation (i.e. developmentally
appropriate), but with compensations*
6 Total oral diet (developmentally appropriate), with no restrictions
*
Compensations = special feeding equipment (e.g. special nipples/cups), special
therapy strategies (e.g. pacing), or special positioning (e.g. side-lying for infants,
head support for older children).
40 J.E. Strychowsky et al. / International Journal of Pediatric Otorhinolaryngology 82 (2016) 38–42

Table 3 population since the analyses are underpowered to detect a

Patient demographics and comorbidities by cleft type.
difference.
No repair Cleft 1 Cleft 2 Cleft 3 Mean PAS and FOIS scores improved for all cleft sub-types with
Demographics management (Table 5). Note that for the PA scale, a smaller number
Age at 1st MBS (mean in years) 2.0 3.0 3.5 2.8 post-surgery reflects improved swallowing, while for the FOIS
Age at pre-op MBS used in analysis NA 3.5 4.4 1.3 score, a larger number post-surgery reflects a less restricted and
(mean in years) more age-appropriate diet.
Age at surgery (mean in years) NA 4.0 4.5 1.4a
The mean number of MBS studies and Otorhinolaryngology
Time from 1st ORL visit to surgery NA 1.9 0.86 0.34
(mean in years) (ORL) visits pre- and post-operatively by cleft type is reported in
Table 4. Of note, those children who did not undergo surgery still
Comorbidities
Pneumonia (%) 47 58 78 83
required several ORL visits and MBS studies to monitor their
G-tube (%) 14 26 41 83 progress.
TEF (%) 9 6 19 50
EA (%) 5 3 13 50 4. Discussion
GER meds (%) 77 82 91 100
Seizure meds (%) 4 4 9 0
CP (%) 4 4 6 0 To our best knowledge, this is the first study to evaluate the
Syndrome (%) 9 16 28 50 utility of the MBS for the management of laryngeal clefts in
Neuro-developmental (%) 16 15 13 0 pediatric patients. It is the first detailed analysis that illustrates the
GI (%) 86 91 94 100 etiopathogenesis of swallowing impairments among patients with
Respiratory (%) 72 65 82 50
Cardiac (%) 12 16 13 33
laryngeal clefts, attributing dysfunction to all phases of swallowing
and not simply aspiration. We have demonstrated that these
CP, cerebral palsy; EA, esophageal atresia; GER, gastroesophageal reflux; GI,
impairments are present at baseline and may persist beyond
gastrointestinal; MBS, modified barium swallow; NA, not applicable; ORL,
otolaryngology; TEF, tracheoesophageal fistula. surgical correction and/or feeding therapy. We have also shown
a
Not all patients had a pre-operative MBS. that MBS measures of swallowing function improve after laryngeal
cleft repair and/or feeding therapy.
We propose that the utilization of the MBS study for patients
laryngeal penetration or aspiration on baseline MBS: 36% of those with laryngeal clefts is two-fold: (1) to guide surgical decision-
with type 1cleft who received conservative management only, 21% making, and (2) to guide feeding therapy. Surgical decision-making
of those with type 1 cleft who underwent surgical management, is usually based on the degree of laryngeal cleft and function as
and 46% of those with type 2 cleft. However, all patients with type qualified by the presence of feeding difficulties and respiratory
3 laryngeal clefts (100%) demonstrated aspiration at baseline. complications. Whereas type 2 to 4 clefts are primarily repaired
Significant improvements in swallowing function (p < 0.05) due to the presence of the anatomical abnormality alone, type
were observed in all of the surgically managed sub-groups 1 clefts are usually repaired when conservative management fails.
(aspiration in type 1 = 64% vs 32%, type 2 = 38% vs 24%, type This decision is rarely straightforward and is reflected in our data;
3 = 100 vs 50%), as well as in the sub-group who underwent patients with type 1 laryngeal clefts had a greater number of pre-
conservative management (46% vs 3%). However, some degree of operative MBS studies and a longer duration of time from initial
swallowing impairment remained across all cleft types post- assessment to surgery.
intervention. This is likely due to the high prevalence of Some patients underwent surgery despite a normal MBS study.
comorbidities across all sub-groups of children with laryngeal The decision to proceed to surgery is independent of the MBS study
clefts (see Table 3), in particular cardiac disease (12% conservative results and can be attributed to the presence of respiratory
management sub-group, 16% type 1, 13% type 2, 33% type 3), complications in the setting of a diagnosis of laryngeal cleft on
neuro-developmental disorders (16% conservative management interarytenoid palpation. This is an inherent weakness to the MBS
sub-group, 15% type 1, 13% type 2, 0% type 3), and gastro- study since it is an assessment at one point in time. Patients may
esophageal reflux (77% conservative management sub-group, 82% have intermittent aspiration that is not captured during the MBS
type 1, 91% type 2, 100% type 3). Although statistical analyses are study.
presented for patients with type 3 laryngeal clefts, these results The pre-operative MBS study can demonstrate the patient’s
need to be interpreted in the context of the small patient baseline swallowing impairment to help guide the patient and

Table 4
MBS outcomes for pre- versus post-intervention by cleft type.

Pre-intervention Post-intervention

No repair Cleft 1 Cleft 2 Cleft 3 No repair Cleft 1 Cleft 2 Cleft 3

n = 50 n = 98 n = 37 n=3 n = 32 n = 72 n = 38 n=4

Oral phase impairment (%) 36 27 38 67 19* 21 18* 75

Triggering impairment (%) 40 42 24 33 41 36 24 75
Pharyngeal phase impairment (%) 64 79 57 100 56 61* 40* 100
Laryngeal penetration on thin liquids (%) 64 79 54 100 53 60* 40* 75
Laryngeal penetration on thickened liquids (%) 28 48 32 100 13* 24* 11* 50
Aspiration on thin liquids (%) 46 64 38 100 3* 32* 24* 50
Aspiration on thickened liquids (%) 18 34 22 100 3* 13* 8* 50
Silent aspiration (%) 24 53 33 33 3* 29* 16* 25
PA scale (mean score) 4.7 5.7 4.0 6.7 2.7* 4.1* 3.0 4.8
FOIS (mean score) 4.1 4.1 4.2 2.3 4.8* 4.8 4.8 3.3
# MBS 2.8 2.3 1.4 1.2 1.0 1.2 1.2
# ORL clinic visits 5.1 3.7 3.7 1.8 3.5 4.2 7.5

FOIS, functional oral intake scale score; MBS, modified barium swallow; n, number; ORL, otolaryngology; PA, laryngeal Penetration–Aspiration Scale score.
*
p < 0.05.
 J.E. Strychowsky et al. / International Journal of Pediatric Otorhinolaryngology 82 (2016) 38–42 41

Table 5
PA and FOIS scores pre- and post-surgery by laryngeal cleft type.

Pre-intervention Post-intervention

Mean (SD) Mean (SD) Mean difference (95%CI) p

PA scale score
No repair (n = 50) 4.7 (3.1) 2.7 (1.8) 2.0
Type 1 (n = 62) 6.6 (2.5) 4.1 (2.9) 2.5 ( 1.6, 3.3) <0.001
Type 2 (n = 26) 3.9 (3.1) 2.5 (2.5) 1.4 ( 0.1, 2.7) 0.038
Type 3 (n = 2) 7.0 (1.4) 5.0 (1.4) N/A N/A

FOIS score
No repair (n = 32) 4.1 (1.35) 4.8 (1.4) 0.7
Type 1 (n = 63) 3.8 (1.0) 4.8 (1.2) 1.1 (0.8, 1.3) <0.001
Type 2 (n = 26) 4.4 (1.6) 5.2 (1.3) 0.8 (0.3, 1.3) 0.002
Type 3 (n = 2) 3.0 (1.4) 4.0 (2.8) N/A N/A

PA scale score = laryngeal Penetration–Aspiration Scale, ranging from 1 (best) to 8 (worst score), FOIS score = functional oral intake scale score, ranging from 1 (worst) to 6
(best score), p-Value from paired t-test.

caregiver’s expectations for swallowing function after feeding
therapy and when warranted, surgical correction. If the im-
pairment is attributed to laryngeal penetration and/or aspiration
alone, the use of thickening agents or repair of the cleft alone may
be successful. However, if a patient demonstrates oral phase
impairment, delayed triggering of the swallow, or functional
pharyngeal phase impairment, these elements of impaired
swallowing may persist despite rigorous surgical repair or therapy.
Underlying neurological impairment or syndromic associations
may raise suspicion for dysfunction in these areas of swallowing.
The MBS study can help to direct feeding therapy both pre- and
post-operatively. Regardless of laryngeal cleft type, a team
approach to feeding, including a feeding specialist, is paramount.
The presence or absence of laryngeal penetration and/or aspiration
can help direct appropriate feeding compensations and/or wean-
ing protocols. We have also demonstrated that many patients with
laryngeal clefts demonstrate silent aspiration (i.e. aspiration with
no overt clinical signs). This could be easily missed on clinical
examination or FEES assessment. The presence of silent aspiration
on MBS study may preclude the decision to wean thickeners or
other compensations.
There is only one study in the published literature that
evaluated swallowing function in children after laryngeal cleft
repair [20]. Osborn and colleagues performed a 10-year retrospec-
tive review of 60 children and reported FEES and videofluoroscopic
swallow study (VFSS) findings. However, the authors did not report
data for all phases of swallowing, only penetration and aspiration.
At their last follow-up post-surgery, 57% demonstrated normal
swallowing function, 20% demonstrated laryngeal penetration, and
23% demonstrated aspiration.
Our study has some weaknesses given the inherent limitation of
a retrospective study. We were also only able to review MBS
studies performed at our institution. Some patients were also
from out of town and although their surgery was in our center,
subsequent MBS studies may have been completed elsewhere.
Additionally, many children managed at Boston Children’s
Hospital have multiple medical comorbidities and therefore may
be associated with a higher prevalence of swallowing impairment
as a result of these comorbidities. However, this is the first study to
demonstrate that swallowing impairment may be present in all
phases of swallowing among patients with laryngeal clefts and not
simply limited to penetration and aspiration.
5. Conclusions
The MBS study is a useful tool for evaluating swallowing
function and dysfunction in patients with laryngeal cleft
independent of lack or presence of aspiration. Understanding
the overall swallowing impairments in all of the phases of
swallowing (oral phase, swallow triggering, and pharyngeal
impairments) may be beneficial for the surgical decision-making
and perioperative management of these patients. The findings
from the MBS study should be reviewed by the clinical team
(Otolaryngologist and speech-language pathologist) and dis-
cussed with the patients and their caregivers to help guide their
management expectations.
We observed significant improvements in swallowing function
in all of the surgically managed sub-groups at our facility, as well as
in the sub-group who underwent conservative management.
However, it should be noted that no sub-group experienced
complete resolution of swallowing issues. This is likely due to the
high prevalence of comorbidities across all sub-groups of children
with laryngeal clefts and the multifactorial etiology of their
swallowing impairment.
Acknowledgements
The authors would like to thank Dr Kosuke Kawai for his help
with the statistical analyses and Kayla Hernandez and Kara Larson,
speech-language pathologists, for their guidance and support.
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