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Original Research

Otolaryngology–
Head and Neck Surgery

Expert Consensus Statement: Pediatric 1–14


Ó American Academy of
Otolaryngology–Head and Neck
Drug-Induced Sleep Endoscopy Surgery Foundation 2021
Reprints and permission:
sagepub.com/journalsPermissions.nav
DOI: 10.1177/0194599820985000
http://otojournal.org
Cristina M. Baldassari, MD1, Derek J. Lam, MD, MPH2,
Stacey L. Ishman, MD, MPH3, Boris Chernobilsky, MD4,
Norman R. Friedman, MD5, Terri Giordano, DNP6,
Claire Lawlor, MD7, Ron B. Mitchell, MD8,
Heather Nardone, MD9, James Ruda, MD10, Habib Zalzal, MD11,
Adrienne Deneal, MHS12, Nui Dhepyasuwan, MEd12,
and Richard M. Rosenfeld, MD, MPH, MBA13

Abstract apnea, flexible fiberoptic endoscopy, upper airway endo-


Objective. To develop an expert consensus statement on scopy, expert consensus
pediatric drug-induced sleep endoscopy (DISE) that clarifies
controversies and offers opportunities for quality improve- Received September 2, 2020; accepted November 27, 2020.
ment. Pediatric DISE was defined as flexible endoscopy to
examine the upper airway of a child with obstructive sleep

O
apnea who is sedated and asleep. bstructive sleep apnea (OSA) affects approximately
1.2% to 5.7% of children1 and is characterized by
Methods. Development group members with expertise in repetitive cessations or reductions in airflow during
pediatric DISE followed established guidelines for developing sleep with oxygen desaturation or arousals. Sequelae of
consensus statements. A search strategist systematically untreated pediatric OSA may include neurocognitive deficits,
reviewed the literature, and the best available evidence was behavioral problems, hyperactivity, poor school performance,
used to compose consensus statements regarding DISE in and decreased quality of life (QOL). Adenotonsillectomy
children 0 to 18 years old. Topics with significant practice (AT) is most commonly the first-line surgical treatment,2 but
variation and those that would improve the quality of up to 30% of children will have persistent OSA after AT,
patient care were prioritized. especially if obese, syndromic, or with other comorbid condi-
Results. The development group identified 59 candidate con- tions.3 To help guide decision making regarding the treatment
sensus statements, based on 50 initial proposed topics, that
focused on addressing the following high-yield topics: (1) 1
indications and utility, (2) protocol, (3) optimal sedation, (4) Eastern Virginia Medical School / Children’s Hospital of The King’s
Daughters, Norfolk, Virginia, USA
grading and interpretation, (5) complications and safety, and 2
Oregon Health and Science University, Portland, Oregon, USA
(6) outcomes for DISE-directed surgery. After 2 iterations 3
Cincinnati Children’s Hospital Medical Center / University of Cincinnati
of the Delphi survey and removal of duplicative statements, College of Medicine, Cincinnati, Ohio, USA
4
26 statements met the criteria for consensus; 11 statements NYU Langone Health, New York, New York, USA
5
were designated as no consensus. Several areas, such as the Children’s Hospital Colorado / University of Colorado Anschutz Medical
Campus, Aurora, Colorado, USA
role of DISE at the time of adenotonsillectomy, were identi- 6
Children’s Hospital of Philadelphia, Philadelphia, Pennsylvania, USA
fied as needing further research. 7
Children’s National Medical Center / George Washington University
School of Medicine and Health Sciences, Washington, DC, USA
Conclusion. Expert consensus was achieved for 26 statements 8
UT Southwestern Medical Center, Dallas, Texas, USA
pertaining to indications, protocol, and outcomes for pedia- 9
Nemours / Alfred I. duPont Hospital for Children, New Castle County,
tric DISE. Clinicians can use these statements to improve Delaware, USA
10
quality of care, inform policy and protocols, and identify Nationwide Children’s Hospital, Columbus, Ohio, USA
11
areas of uncertainty. Future research, ideally randomized West Virginia University, Morgantown, West Virginia, USA
12
American Academy of Otolaryngology–Head and Neck Surgery
controlled trials, is warranted to address additional contro- Foundation, Alexandria, Virginia, USA
versies related to pediatric DISE. 13
SUNY Downstate Health Sciences University, New York, New York, USA

Corresponding Author:
Keywords Cristina M. Baldassari, MD, Department of Otolaryngology–Head and Neck
Surgery, Eastern Virginia Medical School, 600 Gresham Drive, Suite 1100,
drug-induced sleep endoscopy, drug-induced sedation endo- Norfolk, VA 23507, USA.
scopy, bronchoscopy, nasopharyngoscopy, obstructive sleep Emails: cristina.baldassari@chkd.org, Baldassc@gmail.com
2 Otolaryngology–Head and Neck Surgery

of OSA in children, drug-induced sleep endoscopy (DISE) to examine the airway of children with OSA while in drug-
has become increasingly popular as a method to evaluate the induced sleep, (2) recruit the expert development group, (3)
dynamic obstruction in the upper airway that occurs during vet potential conflicts of interest among proposed develop-
sleep. ment group members, (4) perform a systematic literature
DISE is flexible fiberoptic endoscopy that is performed review, (5) determine the scope and population of interest
during sedated sleep that facilitates passage of the endoscope for the ECS, (6) develop topic questions and consensus for
transnasally into the pharynx without awakening the patient. statements for each topic question, (7) develop and imple-
First described by Croft and Pringle in 1991,4 DISE has been ment modified Delphi Method surveys, (8) revise the ECSs
widely adopted and is frequently utilized to evaluate adults in an iterative fashion based on survey results, and (9) aggre-
with OSA who are being considered for surgical therapy. gate the data for analysis and presentation. The pertinent
The VOTE classification (velum, oropharynx, tongue base, details of these steps are briefly described.
epiglottis) is the most widely used scale to report DISE find-
ings in adults.5 Many studies have been published describing Determination of DISE as the Topic of an ECS and
patterns of obstruction observed during DISE and associa- Development Group Recruitment and Vetting
tions with baseline OSA severity.6-9 In addition, there is a DISE was proposed for an ECS by the AAO-HNSF Sleep
growing body of literature describing the impact of DISE on Disorders Committee. After deliberation, the AAO-HNSF
treatment decision making in adult OSA10-13 and the utility GTF approved and prioritized the suggestion; development
of DISE in predicting surgical outcomes.14-17 group leadership was selected, and administrative support
The widespread utilization of DISE by sleep surgeons and was allocated. Development group membership was strategi-
the significant advances that have been made in DISE-related cally developed to ensure appropriate representation of all
research have led to increased convergence in the practice of relevant stakeholder groups and organizations within otolar-
adult DISE. In contrast, the interest in DISE as part of the yngology. The stakeholders were contacted regarding the
treatment paradigm for pediatric OSA has occurred far more consensus statement project and the requirements and
recently, principally in the past 5 years. DISE has been uti- desired qualifications for development group membership;
lized to identify sites of obstruction in children prior to or each group then nominated its own representative content
concurrent with AT (‘‘surgically naı̈ve’’) and also in those expert to participate.
who have persistent post-AT OSA. However, evidence The ECS development group included representatives
demonstrating its utility and effectiveness is sparse; most liter- from the American Society of Pediatric Otolaryngology, the
ature in this field consists of small case series. Society of Otorhinolaryngology and Head-Neck Nurses, the
Although DISE has been shown to affect decision making International Surgical Sleep Society, and the Society of Ear
in surgically naı̈ve children, leading to a change in surgical Nose and Throat Advancement in Children, as well as appro-
procedure in some cases,18 it remains unclear whether the priate committees within the AAO-HNSF including the
outcomes of DISE-directed interventions are superior to out- Board of Governors, the Sleep Disorders Committee, the
comes of more conventional approaches. Multiple studies Pediatric Otolaryngology Committee, and the Section for
have demonstrated substantial practice variation with respect Residents and Fellows-in-Training. The methodologist and
to DISE in children.19-21 Areas of controversy include the representatives from the Society of Otorhinolaryngology and
appropriate indications, optimal sedation regimen, endo- Head-Neck Nurses and Section for Residents and Fellows-
scopy protocol, and interpretation of DISE findings. in-Training were nonvoting members of the development
Given the knowledge gap that exists regarding the role of group.
DISE in the management of pediatric OSA, the American All development group members are in active clinical
Academy of Otolaryngology–Head and Neck Surgery (AAO- practice, are content experts in DISE, and agreed in advance
HNSF) Guidelines Task Force (GTF) selected this topic for of appointment to participate in all verbal discussions (per-
expert consensus statement (ECS) development. The objective formed via teleconference) and votes. Once the development
of this ECS is to identify areas of consensus from a develop- group was assembled, complete disclosure of potential con-
ment group of experts in the field regarding the appropriate flicts of interest were reported and vetted, including recusal
indications, perioperative protocols, and interpretation of of panelists receiving support from Inspire Medical Systems,
DISE in children. Our primary aim was to develop statements Inc during potential discussions regarding hypoglossal nerve
that would address areas of controversy, with the goal of stimulation. Conflicts of interest were consistent with the
reducing practice variation and improving the quality of care Council of Medical Specialty Societies’ Code for Interactions
for pediatric patients with OSA. With Companies,23 which requires that the chair and a major-
ity of the participants do not have a direct conflict with the
Methods deliberations. The development group chair and assistant
This ECS was developed according to an a priori protocol22 chair led the development of the consensus statements and the
(previously used by AAO-HNSF to successfully develop Delphi process with input from a senior consultant/methodol-
multiple other consensus statements) with the following ogist from AAO-HNSF leadership and GTF and with admin-
steps: (1) define the subject of the ECS as use of endoscopy istrative support from an AAO-HNSF staff liaison.
Baldassari et al 3

Literature Review and Determination of the Scope was divided into 2 Delphi rounds. All answers were deidenti-
of the Consensus Statement fied and remained confidential to development group mem-
bers; however, names were collected by staff to ensure proper
Two systematic literature reviews were performed by an
follow-up, if needed.
information specialist, using keywords identified by the
According to the outcomes of the top ranked topic list
development group, to identify current evidence regarding
choices and resulting discussion, the development group
pediatric DISE. The literature searches were conducted from
chair and assistant chair developed the first Delphi survey,
October to December 2019 and included all relevant publica-
which consisted of 59 statements. Prior to dissemination to
tions in English from PubMed, Embase, Cumulative Index
the development group, the Delphi surveys were reviewed
to Nursing and Allied Health Literature, Cochrane Database
by the methodologist for content and clarity. Questions in
of Systematic Reviews, Web of Science, Agency for
the survey were answered with a 9-point Likert scale, where
Healthcare Research and Quality, ECRI National Guideline
1 = strongly disagree, 3 = disagree, 5 = neutral, 7 = agree,
Clearinghouse, Canadian Medical Association Infobase, UK
and 9 = strongly agree. The surveys were distributed, and
National Institute for Health and Care Excellence, TRIP
responses were aggregated, distributed back to the develop-
Database, UK National Library of Guidelines, Scottish
ment group, discussed via teleconference, and revised, if
Intercollegiate Guidelines Network, New Zealand Guidelines
warranted. The purpose of the teleconference was to provide
Group, Australian National Health and Medical Research
an opportunity to clarify any ambiguity, propose revisions,
Council, Guidelines International Network, and BIOSIS. The
or drop any statements recommended by the development
following terms were used in the search:
group.
Criteria for consensus were established a priori as
(drug-induced sleep endoscopy OR drug-induced sedation
follows22:
endoscopy) OR ((DISE OR bronchoscopy OR nasal endo-
scopy OR nasendoscopy OR nasopharyngoscopy OR flexible
Consensus: statements achieving a mean score of 7.00
fiberoptic endoscopy OR endoscopy) AND (snoring OR
or higher and having no more than 1 outlier,
sleep OR sleep apnea OR sleep apnea, obstructive OR sleep-
defined as any rating 2 or more Likert points from
disordered breathing OR airway)) AND (infant OR infants
the mean in either direction.
OR child OR children OR adolescent OR adolescents OR
Near consensus: statements achieving a mean score of
juvenile OR juveniles OR pediatric OR pediatrics OR youth)
6.50 or higher and having no more than 2 outliers.
No consensus: statements that did not meet the criteria
The target audience of the ECS was defined as otolaryn-
of consensus or near consensus.
gologists or other endoscopists performing DISE for children
with OSA. The target population was defined as patients
Two iterations of the Delphi survey were performed. All
aged 0 to 18 years. Once the target population and scope of
group members completed all survey items. The develop-
practice were determined, the development group used the
ment group extensively discussed (via teleconference) the
results of the literature reviews, combined with their expert
results of each item after the first Delphi survey. Most items
opinion and stakeholder needs, to propose topics and ques-
that reached consensus were accepted; however, 10 consen-
tions for which knowledge gaps, uncertainty, or opportuni-
sus statements were revised for improved clarity. Items that
ties for quality improvement existed. The main focus was
did not meet consensus were discussed to determine if word-
DISE-related areas with significant practice variation that
ing or specific language was pivotal in the item not reaching
would most benefit from consensus from expert clinicians.
consensus. The second iteration of the survey was used to
These topics were used as the basis for formulating the ini-
reassess items for which there was consensus or near consen-
tial statements, which were then evaluated through the
sus or for items for which there were suggestions for signifi-
Delphi Survey Method.
cant alterations in wording that could have affected survey
results. All items reaching consensus were accepted except
Delphi Survey Method Process and Administration for 4 statements removed due to redundancy. The factors
A modified Delphi Survey Method was utilized to assess leading to the remaining items not reaching consensus were
consensus for the proposed statements,22 with multiple anon- not attributed to wording or other modifiable factors but
ymous surveys completed to minimize bias within the devel- rather a true lack of consensus.
opment group and facilitate consensus.24 Web-based software The final version of the ECSs was grouped into several
(www.surveymonkey.com) was used to administer confiden- specific areas: indications and utility for pediatric DISE, pro-
tial surveys to development group members. A potential topic tocol for DISE, optimal sedation for DISE, grading/interpre-
list of 50 questions was put together by the development tation of DISE, outcomes of DISE-directed surgery, and
group during the first call, and each development group complications and safety. The final manuscript was drafted
member was invited to provide 1 draft statement for each of with participation and final review from each development
his or her top 5 ranked topic list choices. The survey period group member.
4 Otolaryngology–Head and Neck Surgery

Table 1. Indications and Utility for Pediatric DISE: Statements That Reached Consensus.
No. Statements that reached consensus Mean Outliers

4 Polysomnography is recommended prior to DISE to confirm presence and severity of OSA. 8.88 0
1b DISE is indicated for children with OSA who have small tonsils. 7.50 1
14b DISE is useful in children with persistent OSA to provide information about planning 7.75 1
additional sleep surgery.
6 Children should undergo DISE before being considered for additional pharyngeal surgery 8.13 1
for persistent OSA.
7 DISE should be performed in children with persistent OSA prior to performing additional 7.63 1
surgery for OSA.
10 There is limited utility for DISE in children with an apnea-hypopnea index \2. 7.75 1
13 The role of DISE at the time of adenotonsillectomy is limited in children with OSA and 8.25 0
adenotonsillar hypertrophy who are not at risk for persistent OSA.
20b Healthy children with OSA and small tonsils who have partial or complete obstruction at 8.50 0
the tonsils observed during DISE would benefit from a palatine tonsillectomy.
18 DISE performed concurrently with adenotonsillectomy in children at high risk of persistent 7.25 1
OSA identifies sites of additional obstruction beyond the tonsils and adenoids.
Abbreviations: DISE, drug-induced sleep endoscopy; OSA, obstructive sleep apnea.

Table 2. Protocol for Pediatric DISE: Statements That Reached Consensus.


No. Statements that reached consensus Mean Outliers

24b Photo and video documentation of pediatric DISE are useful to facilitate education, 8.38 0
collaboration, and research.
22 Jaw thrust and lateral positioning maneuvers may be utilized during pediatric DISE to augment 9.00 0
the exam.
23 Evaluation of the airway below the glottis should be carried out at the time of DISE in children 8.38 1
with signs or symptoms suggesting tracheal or bronchial pathology.
32 Information obtained from DISE is most useful when no supplemental oxygen is being given. 8.13 1
37 Topical nasal decongestion should not be used during DISE as it alters nasal anatomy and 7.38 1
airflow dynamics that may mask the true obstructive breathing pattern.

Abbreviation: DISE, drug-induced sleep endoscopy.

Results Indications and Utility for Pediatric DISE


A total of 9 statements reached consensus regarding the indi-
The literature search yielded 123 articles, with 62 remaining cations and utility of pediatric DISE (Table 1). There were
after the titles and abstracts were screened for relevance. 5 statements that did not reach consensus (Table 7). The
Additional exclusions at this stage included commentaries, development group emphasized the importance of obtaining
letters to the editor, editorials, studies in animals, and a polysomnogram (PSG) prior to performing DISE in chil-
research limited to adults. The remaining 62 articles were dren and that DISE has limited utility in children with an
reviewed independently by the chair and assistant chair and apnea-hypopnea index (AHI) \2.
classified per the Oxford Centre for Evidence-Based Medicine’s There was extensive discussion regarding appropriate
2011 levels of evidence.25 Based on the evidence levels, indications for DISE. Consensus was reached that DISE was
there were 11 level 1 articles, 1 level 2 article, 4 level 3 arti- indicated in the following scenarios: (1) children with OSA
cles, 45 level 4 articles, and 1 level 5 article. Thus, most rel- and small tonsils, (2) children with persistent OSA following
evant articles were small case series. AT, and (3) at the time of AT for children at high risk of per-
A total of 59 consensus statements were developed for sistent OSA. The development group identified the following
assessment. After 2 iterations of the Delphi survey and factors as being associated with a high risk of persistent
removal of duplicative and similar statements, 26 statements OSA following AT: (1) severe baseline disease, defined as
met the standardized definition for consensus (Tables 1-6), an obstructive AHI .10 events/hour; (2) obesity; (3) craniofacial
and 11 did not (Table 7). The expert statements were orga- syndromes, including Down syndrome; and (4) neuromuscular
nized into the specific subject areas.
Baldassari et al 5

Table 3. Optimal Sedation for Pediatric DISE: Statements That Reached Consensus.
No. Statements that reached consensus Mean Outliers

26 Either propofol or dexmedetomidine is an optimal form of sedation for use in pediatric DISE. 7.75 0
27 The level of sedation during pediatric DISE should be titrated to audible snoring, an obstructive 8.25 0
breathing pattern, or both while allowing for flexible endoscopy without patient reactivity or
awakening.
30 Inhalational agents may be used for induction and to gain intravenous access during DISE but 8.50 0
should be discontinued for the diagnostic portion of the study.
31 The surgeon and anesthetist should agree on the sedation protocol prior to DISE. 9.00 0
Abbreviation: DISE, drug-induced sleep endoscopy.

Table 4. Grading/Interpretation of Pediatric DISE: Statements That Reached Consensus.


No. Statements that reached consensus Mean Outliers

39 During pediatric DISE, the following parameters should be observed and documented for each 8.75 0
anatomic level: (a) site, (b) pattern or shape, and (c) severity of obstruction.
40 During pediatric DISE, the following anatomic sites should be assessed and documented: nasal 8.86 0
airway (including nasal cavities, nasopharynx), palate/velum, pharyngeal airway (including lateral
oropharyngeal wall and tongue base), and supraglottic larynx.
48b Cine MRI can be a useful adjuvant to DISE in children to determine the thickness of lingual tonsils 7.50 1
and assess the contribution of the tongue base to palatal obstruction observed during DISE.
Abbreviations: DISE, drug-induced sleep endoscopy; MRI, magnetic resonance imaging.

Table 5. Complications and Safety of Pediatric DISE: Statements That Reached Consensus.
No. Statements that reached consensus Mean Outliers

25 DISE can be performed safely outside the operating room if anesthesia support is available. 7.88 1
52b Children with OSA undergoing DISE may be discharged the same day. 8.38 0
50 The preoperative polysomnogram will guide the severity of hypoxemia and oxygen desaturations 7.88 1
that should be tolerated during DISE.
Abbreviations: DISE, drug-induced sleep endoscopy; OSA, obstructive sleep apnea.

Table 6. Outcomes of DISE-Directed Surgery in Children: Statements That Reached Consensus.


No. Statements that reached consensus Mean Outliers

44 Children undergoing DISE-directed surgery for OSA experience improvement in symptomatology 8.00 0
and quality of life.
45 Children undergoing DISE-directed surgery for OSA experience improvement in apnea-hypopnea 7.63 0
index and oxygen saturation nadir.

Abbreviations: DISE, drug-induced sleep endoscopy; OSA, obstructive sleep apnea.

disorders. The group was not able to identify how ‘‘small’’ ton- risk for persistent OSA can identify sites of additional obstruc-
sils should be defined. tion beyond the tonsils and adenoids. However, whether DISE
There was lengthy discussion regarding the controversial can change surgical decision making at the time of AT in chil-
topic of the appropriateness of performing DISE at the time of dren at risk for persistent OSA achieved only near consensus.
AT in children with OSA. Ultimately, the development group There was consensus that the role of DISE at the time of
agreed that DISE performed at the time of AT for children at AT is limited in children with OSA and adenotonsillar
6 Otolaryngology–Head and Neck Surgery

Table 7. Statements That Were Classified as No Consensus.


No. Statement Mean Outliers

Indications and utility for pediatric DISE


47b DISE is indicated at the time of adenotonsillectomy in surgically naı̈ve children who are at risk for 5.63 5
persistent OSA.
11b Performance of DISE at the time of adenotonsillectomy in children at high risk of persistent 6.50 4
disease may guide future OSA management.
3 DISE is indicated at the time of adenotonsillectomy for children with OSA who have a high risk of 6.00 4
persistent sleep apnea, such as those with trisomy 21 or obesity.
9 Children with mild OSA and symptoms can be assessed with DISE. 5.75 3
17 Children at high risk of persistent OSA who are undergoing a planned adenotonsillectomy benefit 6.00 3
from DISE if the surgeon and family are prepared to proceed with adjuvant surgery at the time of
the adenotonsillectomy.
Protocol for DISE
33 Supplemental oxygen should be used during DISE if intraprocedure levels of oxygen saturation fall 6.63 5
below the nadir seen on PSG.
34 DISE results can be altered by supplemental oxygen. 7.13 3
36 Topical nasal decongestants should be avoided during assessment of the nasal airway but can be 5.13 5
used to facilitate advancement of the endoscope for the rest of the DISE evaluation after the
nasal examination is complete.
12 Children with persistent OSA should attempt positive airway pressure therapy prior to DISE. 5.50 3
Optimal sedation for DISE
38 Ketamine should be avoided when DISE is performed in children because it increases muscle tone, 5.38 5
which can stiffen the airway and mask a child’s true obstructive breathing pattern.
Outcomes of DISE-directed surgery
19 For children at high risk of persistent OSA, DISE findings at the time of adenotonsillectomy may 7.13 3
direct surgery to address additional sites of obstruction at the time of adenotonsillectomy

Abbreviations: DISE, drug-induced sleep endoscopy; OSA, obstructive sleep apnea; PSG, polysomnogram.

hypertrophy who are not at risk for persistent OSA. In chil- suggest tracheal or bronchial pathology. The development
dren with persistent OSA following AT, DISE is useful in group agreed that nasal decongestants and supplemental
assessing candidacy for additional sleep surgery. The devel- oxygen should be avoided during pediatric DISE.
opment group considered children to have persistent OSA if
they have severe persistent OSA (obstructive AHI .10 Optimal Sedation for DISE
events/hour) or mild to moderate OSA (obstructive AHI A total of 4 statements met consensus regarding the optimal
between 2 and 10 events/hour) with associated daytime and/ sedation for DISE (Table 3), while 1 did not reach consensus.
or nighttime OSA symptomatology. The development group These statements address the use of inhalational anesthesia,
recommended that children undergo DISE prior to perform- the choice of intravenous sedative agent, the level of sedation,
ing additional surgery for persistent OSA, including pharyn- and communication with the anesthetist during pediatric
geal surgery. Pharyngeal surgery refers to procedures that DISE.
address lateral oropharyngeal wall collapse and tongue base The development group felt that there was an opportunity
obstruction, such as expansion pharyngoplasty and midline for quality improvement regarding sedation for pediatric
glossectomy, respectively. DISE as there is a significant amount of practice variation in
this area. Propofol and dexmedetomidine were identified as the
Protocol for DISE optimal sedation agents for use in pediatric DISE. Inhalational
Nine statements were created pertaining to the protocol for agent use should be minimized during DISE; if used during
performing pediatric DISE, of which 5 reached consensuses induction, these agents should be discontinued prior to the diag-
(Table 2). The importance of photo and video documentation nostic portion of the procedure. The development group could
during DISE was highlighted. In addition, there was consen- not reach consensus regarding the use of ketamine during
sus on the utility of positioning and maneuvers such as jaw DISE, due to concern that it increases muscle tone and masks a
thrust during pediatric DISE to augment the examination. child’s true pattern of upper airway obstruction.
Evaluation of the subglottis and lower airway during DISE The ideal level of sedation for pediatric DISE is reached
was deemed unnecessary unless patients have symptoms to when the child has audible snoring and/or an obstructive
Baldassari et al 7

breathing pattern as well as a lack of responsiveness to the may affect the execution of pediatric DISE includes identifi-
introduction of the flexible endoscope. cation of a positional component of OSA, as well as the
degree of hypoxemia.
Grading/Interpretation of DISE Since the development of DISE in adults in the 1990s, the
All 3 proposed statements regarding grading and interpreta- indications and utility of DISE have evolved for children
tion of pediatric DISE met consensus (Table 4). The impor- with OSA.21 The most common reason to perform pediatric
tance of a systematic approach to the DISE examination was DISE has been to direct additional surgical intervention in
emphasized, including the evaluation of the location, pattern, children with persistent OSA following AT. Indeed, children
and severity of obstruction. The anatomic sites that should with persistent OSA benefit from DISE to identify sites of
be documented during pediatric DISE are the nasal cavity obstruction that may be addressed surgically.19,21,31,32
and nasopharynx, the velum, the pharynx (including lateral Common sites of obstruction noted upon DISE in children
walls and tongue base), and the supraglottic larynx. Cine with persistent disease include the tongue base, adenoids
magnetic resonance imaging (MRI) can be a useful adjuvant (secondary to regrowth), inferior turbinates, velum, and lat-
to DISE in children in that it can provide additional informa- eral oropharynx.19,32-34 In a review21 of the pediatric DISE
tion regarding lingual tonsil thickness and tongue base con- literature, 100% of 162 children undergoing DISE for the
tribution to palatal obstruction. indication of persistent OSA had at least 1 site of obstruction
identified.
Complications and Safety The development group reached consensus that DISE-
All 3 statements related to complications and safety reached directed surgery for children with persistent OSA can improve
consensus (Table 5). While DISE is typically performed in PSG parameters such as AHI and symptomatology more than
the operating room, the procedure may be safely performed surgery performed without this information. Indeed, there are
outside this setting, provided there is support from anesthesia some emerging data that DISE may be superior to in-office
clinicians. Children, even those with severe disease and clinical examination in identifying sites of obstruction in chil-
comorbid medical conditions, can typically be discharged dren with persistent OSA. In a series of 22 children35 with
the same day that DISE is performed. Pre-DISE PSG pro- persistent OSA, DISE resulted in a change of the surgical
vides valuable information regarding a child’s oxygenation plan in 73% of patients, with the most common modifications
that can be used to guide the degree of desaturation allowed being performance of turbinate reduction and pharyngoplasty.
during the procedure. There is significant controversy regarding when DISE
should be performed in children concurrently with AT (‘‘sur-
Outcomes of DISE-Directed Surgery gically naı̈ve’’) to assess other sites of upper airway obstruc-
Both statements reached consensus regarding outcomes of tion. The development group agreed that DISE can be
DISE-directed surgery (Table 6). These statements high- appropriately used in surgically naı̈ve children to evaluate
lighted the improvements that occur in PSG measures, such children with OSA and small tonsils. However, the develop-
as AHI and oxygen saturation nadir, as well as QOL and ment group was unable to agree on the definition of small
symptomatology following DISE-directed surgery. tonsils, due to some members defining small tonsils as size 1
(based on Brodsky 1-4 scoring)36 while others included size
Discussion 1 and 2. The literature supports the idea that DISE is useful
in children with OSA and small tonsils to identify potential
Indications and Utility for Pediatric DISE sites of obstruction beyond the naso- and oropharynx.37 One
In agreement with the survey of pediatric DISE practices recent study38 found that 60% of children with size 1 tonsils
published in 2017,20 there was a strong consensus among (n = 65) undergoing DISE did not have lateral pharyngeal
group members to obtain a PSG prior to DISE. The rationale wall collapse. Similarly, a review by Galluzzi39 found that
for this recommendation is that multiple studies have 30% of surgically naı̈ve children undergoing DISE had no
demonstrated that clinical assessment is a poor predictor of evidence of obstruction caused by adenoids and/or tonsils.
the presence of OSA and its severity.1,26-29 In the Childhood Data are lacking for whether performing DISE-directed
Adenotonsillectomy Trial, otolaryngologists accurately pre- surgery improves outcomes, such as supraglottoplasty for
dicted the presence of OSA only 50% of the time in healthy sleep-dependent laryngomalacia in children who do not have
children between the ages of 5 and 9 years.30 adenotonsillar obstruction. In contrast, if DISE does reveal
There are several advantages to confirming the diagnosis lateral pharyngeal wall obstruction in children with small
and severity of OSA by PSG prior to performing pediatric tonsils, the development group agreed that tonsillectomy
DISE. Because a child who has infrequent obstructive should be performed. This is supported by the study by Chen
respiratory events during PSG is unlikely to exhibit a repeti- and He,40 which noted significant improvement in the
tive obstructive breathing pattern during DISE, DISE was respiratory disturbance index following DISE-directed ton-
not felt to be appropriate for children with an AHI \2 sillectomy for children with grade 1 tonsils.
events/hour. In addition, these children may be managed The development group agreed that the role of DISE at
with watchful waiting or anti-inflammatory medications. the time of AT is limited in children with enlarged tonsils
Other useful information that can be obtained from PSG that and adenoids who are not at risk for persistent OSA. In such
8 Otolaryngology–Head and Neck Surgery

patients, DISE has repeatedly identified adenotonsillar demonstrated a fourfold increase in OSA by anesthetizing
hypertrophy as the most common site of obstruction.41 AT is the nose.46
also well known to result in significant improvements in The development group also agreed that pre-DISE PSG is
symptomatology and PSG parameters in the majority of oth- recommended and that the information obtained during
erwise healthy children.2 Furthermore, in a report of 88 chil- DISE was most useful when supplemental oxygen was
dren, DISE did not change the surgical plan in .95% of avoided. Surgeon and anesthesia provider awareness of the
surgically naı̈ve children with OSA.35 baseline asleep oxygen saturation data from a pre-DISE PSG
For children at high risk for persistent disease, the devel- is useful as it may allow for permissive oxygen desaturation
opment group agreed that DISE performed at the time of AT during DISE provided the patient’s other cardiorespiratory
can identify sites of additional obstruction beyond the tonsils parameters, including heart rate, are stable. While anesthesia
and adenoids. However, there is limited evidence regarding providers typically use supplemental oxygen to maintain
the utility of performing DISE at the time of AT in these oxygen saturation .90% during surgery, DISE is ideally
children. Previous research has demonstrated that children performed when no supplemental oxygen is administered.
with severe disease often have multilevel obstruction on There were concerns expressed that respiratory control
DISE.42 Some group members asserted that surgically instability (high loop gain) can contribute to obstructive
addressing additional sites of obstruction identified during breathing patterns and that administration of supplemental
DISE at the time of AT may improve treatment outcomes. oxygen will mask loop gain’s effect on the upper airway.47
For example, Ulualp43 reported on a series of children with However, definitive data are lacking regarding if or how
severe OSA who underwent modified expansion pharyngo- supplemental oxygen can alter patterns of obstruction during
plasty at the time of AT after DISE demonstrated lateral oro- DISE.
pharyngeal wall collapse. Larger improvements in the AHI In terms of airway evaluation below the glottis during
were noted in children who had additional palatal surgery DISE, the consensus among group members was that tra-
performed when compared with patients who were treated cheoscopy and bronchoscopy should not routinely be per-
with AT alone (control). formed as part of DISE evaluation unless there are
Another rationale for performing DISE at the time of AT additional clinical findings to suggest lower airway pathol-
in children at high risk for persistent disease was to guide ogy. A recent review48 of 335 patients who underwent tra-
future OSA management. However, the development group cheoscopy and bronchoscopy during DISE found that while
was able to reach only near consensus on this point. Group 5% of children had sites of obstruction in the lower airway,
members expressed concern that removal of the tonsils and only 0.3% of these children required additional intervention
adenoids may change airflow dynamics and necessitate for these findings. One member of the development group
repeat DISE prior to any additional surgical intervention in did express concern that not performing bronchoscopy could
children found to have persistent obstruction. miss cases of tracheomalacia and bronchomalacia that might
be contributing to obstruction.
Protocol for DISE Group members universally agreed that providers per-
In 2017, a multi-institutional survey of pediatric DISE forming pediatric DISE should consider evaluating the
experts was published that demonstrated agreement among impact of jaw thrust and lateral positioning maneuvers on
participants in regard to preoperative evaluation and DISE airway dynamics and obstruction. While positioning devices
protocols but not in the sedation technique.20 Similarly, the and oral appliances are more commonly used to manage
development group identified several technical aspects of OSA in adults, there is evidence that they can be acceptable
DISE that providers should be aware of when performing treatment options for children with persistent OSA.49,50
this procedure. They agreed that topical nasal decongestion Photo or video documentation of pediatric DISE was rec-
should not be used during DISE, as it alters nasal anatomy ommended to enable education, collaboration, and research.
and airflow dynamics that may mask the true obstructive Adoption of a standardized approach for DISE documenta-
breathing pattern. Because the nasal passages account for tion would facilitate prospective multi-institutional research
.50% of upper airway resistance and the Starling resister studies that can advance our knowledge of pediatric DISE
model predicts that improving airflow through the nasal pas- outcomes. Friedman and Parikh51 recently developed a DISE
sages can decrease the collapsing forces in the hypophar- documentation protocol that establishes fixed examination
ynx,44 there was concern from the development group that points for brief video recording. Establishment of standard
nasal medications may result in alterations in the true hypo- examination points allows for a consistent and reproducible
pharyngeal obstructive breathing patterns. optical view, while reference point standardization ensures
Controversy remains regarding the use of nasal anes- that the focal distance from the telescope to the structure
thetics during pediatric DISE, and no consensus was reached will be similar for each DISE procedure.
on this topic. A 2019 publication suggested that topical
anesthesia has no effect on DISE; however, this pilot study Optimal Sedation for DISE
included adults with primary snoring as well as OSA.45 An An ideal anesthetic for pediatric DISE maintains spontaneous
investigation that compared topical lidocaine with placebo ventilation, mimics normal asleep airway tone, is repeatable,
Baldassari et al 9

and does not exacerbate underlying medical conditions. The infusion), and (3) the depth of anesthesia desired. Many cen-
development group agreed that either propofol or dexmedeto- ters have developed a specific DISE anesthetic protocol,20
midine was the optimal form of sedation for DISE. Both and some have dedicated DISE anesthesia teams in an effort
agents have been found to have similar effects on upper to facilitate safety and consistency.
airway morphology as evaluated by cine MRI.52 The benefits Decisions regarding the appropriate agent and depth of
of propofol include quick onset of action and fast redistribu- anesthesia are of major importance, as overdosing can result
tion and metabolism, resulting in rapid awakening when dis- in significant oxygen desaturations and premature termina-
continued. Its cost is also low. The major drawback of tion of the procedure. Alternatively, insufficient sedation
propofol is the potential for dose-dependent airway collapse. may result in poor patient tolerance and unnecessarily pro-
In a systematic review, 4 of 12 studies showed a decrease in long the DISE procedure. Therefore, there was consensus
the upper airway cross-sectional area with increasing doses of that the optimal sedation level is one where the child is snor-
propofol.52 Three additional studies demonstrated a decrease ing and/or showing an obstructive breathing pattern and is
in genioglossus electromyography activity with propofol- not responding to the introduction of the flexible endoscope.
based induction.53-55 Dexmedetomidine’s effect on airway While some adult studies have investigated BIS (bispectral
collapsibility is less than that of propofol, and several studies index) to measure depth of anesthesia with upper airway
report that this drug has minimal effect on upper airway obstruction, there is no widely accepted BIS score for pedia-
cross-sectional area.52 Benefits of dexmedetomidine include tric DISE, and its use with dexmedetomidine is known to be
maintaining spontaneous ventilation, airway patency, and less accurate than it is when propofol is use.61 For these rea-
airway tone, even at higher doses.56 However, its slow redis- sons, the patient’s depth of anesthesia is assessed clinically
tribution and metabolism result in prolonged time to awaken- by lack of responsiveness to stimulation.
ing. Another side effect is bradycardia and hypotension, and it
is more expensive than propofol. However, dexmedetomidine Grading/Interpretation of DISE
has been shown to result in a physiologic state similar to The VOTE classification system for adult DISE, described
NREM sleep. Of note, neither dexmedetomidine nor propofol by Kezirian et al in 2011,5 has been widely adopted by adult
replicates stage R (REM) sleep. sleep surgeons. This system documents the pattern and
Another agent that has been utilized for DISE is keta- severity of obstruction at 4 anatomic sites: the velum, oro-
mine. The development group was unable to reach consensus pharynx, tongue base, and epiglottis. However, it has not
regarding its use, as some noted its utility in counteracting been widely utilized in pediatric DISE likely because it fails
the dexmedetomidine side effects while others expressed to account for additional sites of obstruction noted among
concerns that it increases upper airway tone and subse- children with OSA, such as the nasal cavity and the supra-
quently masks a child’s true obstructive breathing pattern.57 glottis. Thus, investigators including Boudewyns, Chan-
This is supported by a study by Eikermann and colleagues58 Parikh (C-P score), Lam (Sleep Endoscopy Rating Scale),
that demonstrated that ketamine results in a much greater Bachar, and Fishman have developed pediatric DISE grading
increase in genioglossus activity as compared with propofol. scales.62-67 These grading scales allow for a standardized
Inhalational agents are often used for anesthetic induction means of reporting DISE findings with the goal of predicting
and to gain intravenous access during pediatric procedures, surgical outcomes.
including DISE. However, the development group agreed The C-P score has shown good internal and external
that inhalational agents should be discontinued for the diag- validity,64 and a follow-up series demonstrated correlation
nostic portion of the study because they have been shown to with AHI severity and lowest oxygen saturation on PSG.68
cause upper airway obstruction in a dose-dependent manner. Unlike the C-P score, the Sleep Endoscopy Rating Scale
Several studies have also suggested exaggerated upper incorporates the nasal cavity, and it yields a single total
airway collapse with inhalational agents.56 A study of sevo- score for pediatric DISE, which has been shown to correlate
flurane demonstrated a dose-dependent effect on 2 of the with OSA severity and response to AT.37,65 To date, how-
major upper airway dilator muscles, the genioglossus and ever, a universal grading scale has yet to be adopted.
palatoglossus.59 Litman et al reported decreased pharyngeal Whereas the development group did not identify a single
dilator muscle activity with halothane and sevoflurane, with superior DISE scoring system, the group highlighted the
sevoflurane causing relatively more upper airway obstruc- importance of consistently documenting the site, pattern, and
tion.60 Due to these concerns, the development group agreed severity of obstruction. It also agreed that this information
that efforts should be made to minimize inhalation agent should be assessed/documented for the following anatomic
usage during pediatric DISE. sites by all providers performing DISE in children: nasal
A key component to a successful DISE procedure is the airway (including nasal cavities, nasopharynx), palate/
communication between anesthesia and otolaryngology pro- velum, pharyngeal airway (including lateral oropharyngeal
viders while sharing the airway. Therefore, the development wall and tongue base), and supraglottic larynx. Further
group agreed that prior to commencing DISE, the surgeon research and collaboration are needed to develop a pediatric
and anesthesia provider should discuss the following DISE DISE grading system that can be widely adopted and utilized
sedation-related elements: (1) the agent or agents to be used, to allow for standardized research data collection and ulti-
(2) the form in which they will be administered (bolus, mately improved evaluation and treatment algorithms.69
10 Otolaryngology–Head and Neck Surgery

Cine MRI, which uses fast gradient echo sequence with Outcomes of DISE-Directed Surgery
axial and midline sagittal images, has also been utilized to Much of the data regarding DISE-directed surgical outcomes
assess sites of obstruction in children with OSA while the are small, single-institution case series. Thus, definitive data
child is spontaneously breathing under sedation. Advantages demonstrating that DISE-directed surgery is superior to stan-
include its high-resolution, dynamic airway evaluation that dard surgical therapy for OSA are lacking. Despite this,
allows for visualization of multiple anatomic sites at once. there was consensus that children undergoing DISE-directed
Despite this, it has not been widely adopted across many surgery experience improvement in the severity of their
pediatric institutions nationally.19 This is likely due to safety OSA as assessed by PSG parameters.75-77 This is supported
concerns regarding loss of the airway while obtaining the by a systematic review of 120 pediatric patients that found
MRI in sedated children with significant OSA, as well as the improvement in AHI and nadir oxygen saturation following
cost and burden of additional testing. However, there are cer- DISE-directed surgery.75 DISE-directed surgery has also
tain elements of the airway evaluation, including the thick- been shown to reduce the AHI in certain subgroups of pedia-
ness of the lingual tonsils and the contribution of the tongue tric patients with OSA, including infants,18,78 children with
base to palatal obstruction, that may be more readily appar- small tonsils,40,79,80 children with persistent OSA following
ent during cine MRI than DISE. In a small case series70 that an AT,81 children with Down syndrome,34,82-84 and children
examined findings in children undergoing both diagnostic with hypotonia.85 Despite this, persistent OSA has been
modalities, DISE and cine MRI found the same sites of reported following DISE-directed surgery.34,75,84,85 It
obstruction in 10 of 15 patients. DISE identified an addi- remains unclear whether the residual disease burden in these
tional site of obstruction not noted on cine MRI in 3 children would have been more severe if DISE had not been
patients. utilized to direct their treatment. To definitively understand
the role of DISE in pediatric OSA management, further
Complications and Safety research is needed to compare outcomes between children
Overall, DISE is safe and well tolerated in children with treated with and without DISE-directed surgery.
OSA, even those with comorbid conditions, such as obesity, Pediatric OSA can have a profound negative impact on
hypotonia, and Down syndrome.42,71-73 Potential complica- children’s QOL, with parents often reporting significant day-
tions include intraoperative loss of airway from laryngospasm time and nighttime symptomatology such as hyperactivity
or bronchospasm, oversedation, hypotension, bradycardia, and and frequent nighttime awakenings.86 Thus, validated instru-
respiratory depression.21 Thus, DISE should be performed in ments that assess disease-specific QOL and symptoms are
a setting that allows for cardiopulmonary monitoring, intrave- now often utilized in conjunction with PSG when assessing
nous access, availability of emergency airway equipment, and treatment outcomes. The development group reached con-
presence of a skilled team of anesthesia personnel. While it is sensus that children undergoing DISE-directed surgery for
frequently performed in the operative environment, DISE has OSA experience improvement in symptomatology and QOL.
also been safely and successfully performed outside this Evidence supporting this can be found in the study by Park
setting. et al,85 which demonstrated a significant improvement in
Group members deemed it appropriate for DISE to be OSA-18 QOL scores in children with hypotonia after DISE-
performed in settings other than the operating room provided directed surgery. Subjectively, DISE-directed surgery has
that there is access to the elements noted earlier. For exam- also been found to improve patient-reported symptoms such
ple, providers have safely performed DISE in the radiology as snoring, pauses in breathing, gasping for air, mouth
department following cine MRI. In a review of 118 children breathing, daytime somnolence, sleepwalking, sleep talking,
undergoing DISE in an MRI induction suite, there were no teeth grinding, enuresis, and feeling tired.87 Future prospec-
unplanned admissions or major complications.72 Performing tive studies on pediatric DISE-directed surgery should
DISE outside the operating room may also result in cost include validated QOL and symptom scores when assessing
reductions.72 outcomes.
The development group noted that children with OSA
may be discharged the same day as the DISE procedure, but Strengths and Limitations
it did not make specific recommendations regarding the A major strength of this consensus statement is the metho-
duration of time that they should be observed in the posta- dology of the consensus statement process. It is rigorous and
nesthesia care unit. Children can be discharged the same day follows an explicit, a priori, tested protocol to focus on qual-
as DISE, provided that they meet appropriate discharge cri- ity improvement, reduce bias, and ensure consistency regarding
teria. Such criteria may vary slightly among institutions. A judgments of consensus, near consensus, and no consensus.
common clinical scale used to determine discharge eligibil- In addition, the participants are content experts in the field
ity is the modified Aldrete scale that assesses patient charac- with extensive clinical and research experience with DISE.
teristics such as consciousness, respiration, and motor The primary limitation of this research was the lack of high-
activity.74 quality, prospective studies to inform the group members’
Baldassari et al 11

opinions. In most studies that were reviewed, there was concern writer, panel member; Terri Giordano, writer, panel member;
for bias because most were small cases series based on chart Claire Lawlor, writer, panel member; Ron B. Mitchell, writer,
review without control or comparison groups. Additionally, as panel member; Heather Nardone, writer, panel member; James
the methodology protocol for ECS development involves only Ruda, writer, panel member; Habib Zalzal, writer, panel member;
otolaryngology stakeholders, direct input from other disciplines Adrienne Deneal, writer, AAO-HNSF staff liaison; Nui
Dhepyasuwan, writer, AAO-HNSF staff liaison; Richard M.
such as anesthesia is lacking.
Rosenfeld, writer, senior adviser .
The development group identified numerous topics that
merit further research, including whether DISE performed at Disclosures
the time of AT in children at risk for persistent disease
Competing interests: Cristina M. Baldassari, CORE Grant
improves outcomes. Additional research is needed to deter- Funding, Inspire Grant Funding; Stacey L. Ishman, Inspire
mine an optimal sedation protocol and ideal grading system Medical—consultant, National Institutes of Health grant support
for pediatric DISE. Future studies should also clarify how (National Heart, Lung, and Blood Institute / National Institute of
best to define small tonsils and assess outcomes in children Dental and Craniofacial Research); Boris Chernobilsky, Inspire
with small tonsils in which DISE identifies alternate sites of Medical Systems, Inc—stock; Adrienne Deneal, salaried
obstruction. Nationwide adoption of a standardized protocol employee of American Academy of Otolaryngology–Head and
and grading system would allow for significant advances to Neck Surgery Foundation; Nui Dhepyasuwan, salaried employee
be made in research on DISE-directed surgical outcomes. of American Academy of Otolaryngology–Head and Neck
Surgery Foundation.
Conclusions Sponsorships: American Academy of Otolaryngology–Head and
Neck Surgery Foundation provided staff support and reimbursed
A development group of otolaryngologists and a head and development group members for travel expenses.
neck nurse developed a consensus statement on pediatric Funding source: None.
DISE based on available evidence and expert opinion utilizing
an established protocol. Numerous topics related to pediatric References
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