Structure and Functions of Pediatric Aerodigestive Programs: A Consensus Statement

We used the Delphi method to obtain consensus over a range of topics related to aerodigestive care. The Delphi method is an iterative, questionnaire-based method of obtaining consensus, which has been adapted to use in health care.^11,–17 This process has particular strengths in situations in which more quantitative evidence is either lacking or cannot be developed easily.

We sought to find common themes and recommendations that would attain a high level of consensus, with consensus defined as a supermajority of >75% or a median response rate that did not change with repeated surveys. We identified a priori 4 broad content areas: (1) definition of an aerodigestive patient, (2) essential construct and functions of an aerodigestive program, (3) identification of aerodigestive research priorities, and (4) evaluation and recognition of aerodigestive programs and future directions. Descriptive statistics were performed by using Excel 2014 (Microsoft Corp, Redmond, WA). This study was reviewed and exempted by the Mayo Clinic Institutional Review Board.

Thirty-three specialists from 11 well-established aerodigestive centers in the United States were recruited to participate based on their national and international recognition in aerodigestive care (12 pediatric pulmonologists, 11 pediatric otolaryngologists, 8 pediatric gastroenterologists, and 2 speech-language pathologists). Participants were invited by the corresponding author based on active participation in long-standing or well-established programs, leadership in program creation, involvement at meetings and conferences, or in publication. A series of 3 questionnaires was administered electronically, with target participation of 100% for each questionnaire. The questionnaires started with more open solicitation of aerodigestive program processes, construct and research priorities. Results were grouped into themes and ranked based on frequency of response. Ranked results and summary statistics were returned to participants. Subsequent questionnaires requested ranking to achieve supermajority. By completion of the third questionnaire, each item had either achieved consensus by a supermajority of 75% or did not change by more than 1 rank, so no further questionnaires were administered.

Despite surveying a range of aerodigestive programs, differing by region, size, and duration of formal operation, consensus was achieved on almost all questions, with a supermajority of >75% agreement or stable median response. Response rates for questionnaires 1 to 3 were 100%, 97%, and 100%, respectively.

A 2-sentence structure for the definition of an aerodigestive patient was chosen. The definition developed and preferred by the majority (75.8%) of respondents is as follows:

“A pediatric aerodigestive patient is a child with a combination of multiple and interrelated congenital and/or acquired conditions affecting airway, breathing, feeding, swallowing, or growth that require a coordinated interdisciplinary diagnostic and therapeutic approach to achieve optimal outcomes.

This includes (but is not limited to) structural and functional airway and upper gastrointestinal tract disease, lung disease because of congenital or developmental abnormality or injury, swallowing dysfunction, feeding problems, genetic diseases, and neurodevelopmental disability.”

For this aim, respondents identified the core aerodigestive team members and the services that should be available within the center. These results are summarized in Table 2. Given that a multidisciplinary team meeting was identified as a key component of the aerodigestive care cycle, participants were asked which disciplines should be present for these meetings. These results are indicated in Table 2. Questioning the roles of care coordinator and nursing, 73% of respondents viewed these roles as the same, whereas 44% also cited the benefits of specialty-specific nursing, especially in providing education and follow-up support for families. Nurse practitioners were identified as the preferred discipline to serve as care coordinator (81%), although advanced practice nurses, registered nurses, and physician assistants were also identified as potential care coordinators. Although general pediatricians may be helpful in these clinics, they have not been traditionally part of aerodigestive teams, with only 2 of the participating programs currently using them in this way. Primary pediatrician roles identified by a supermajority of participants included the following: inpatient generalist consultation, outpatient generalist input, and as primary physician (88%, 81%, and 81%, respectively). Respiratory therapists are used in the programs of participants for education, procedural support, clinical assessments (especially ventilator settings), and performance of pulmonary function testing.

Participants also identified the essential defining functions and features of the care cycle of an aerodigestive program. These results are summarized in Table 3. During further clarification, 84% of respondents rated performance of combined endoscopy with all providers present together at the same time (ear, nose, and throat [ENT], gastroenterology, pulmonology) as essential. This allows all providers to directly observe all portions of the evaluation and maintain a dialogue with each other. They were split on having all shared clinic visits performed together, in the same clinic space, with 55% citing this as “essential” and 45% as “beneficial but nonessential.” Seventy percent of respondents supported a target time frame for completion of initial diagnostic evaluation, defined as the time from the first appointment for diagnostic evaluation until wrap-up from the first diagnostic evaluation. The median time for this evaluation was 7 days with an interquartile range (IQR) of 5 to 17.5 days. It was acknowledged that this is modified by the urgency and specific needs of the patient. Ninety-one percent of participants cited wrap-up visits could occur either in person or over the phone. The experience of seeing the team working together was cited as making a strong impact on caregivers.

Feeding and swallowing disorders are recognized to be highly prevalent in patients with aerodigestive disorders; therefore, clinical swallowing evaluations, fiberoptic endoscopic evaluations of swallowing (FEES), videofluoroscopic swallow studies (VFSS), and provision of direct feeding therapy were all identified as essential components of a program (97%, 97%, 100%, and 100%, respectively). Clinical swallowing evaluations and VFSS were reported as essential for “majority of aerodigestive evaluations.” Speech-language pathologists were identified as the provider of choice for each of these assessments and/or therapies (100% for clinical swallowing evaluation, 81% for FEES, 81% for VFSS, and 91% for feeding therapy), although the role for otolaryngologists in the performance of FEES was supported (78%). A role for occupational therapists for development, instruction, and modeling of feeding plans was well supported (78%), although responses were mixed for their role in clinical swallowing evaluations (70%), VFSS (39%), and FEES (34%). Forty-five percent of participating programs use occupational therapists for feeding and swallowing evaluations (clinical or instrumental).

Operative diagnosis and intervention are recognized as fundamental to aerodigestive care. Overall, respondents cited interventional procedures as performed by pediatric pulmonologists to be beneficial to aerodigestive program function but essential for pediatric gastroenterologists. Consensus for these rankings was achieved based on unchanging median response over serial surveys, although some reached a supermajority of 75% at same rank. The importance of specific procedures cited is listed in Tables 4 and 5 and generally suggest the importance of a high level of procedural skill and expertise for pulmonologists and gastroenterologists, even if all proceduralists do not routinely perform all listed procedures. Respondents strongly endorsed the essential nature of proficiency in open and endoscopic airway reconstruction for otolaryngologists in aerodigestive programs (81%). This includes the following categories: (1) open or endoscopic procedures that directly increase the diameter of the cartilaginous skeleton of the airway, (2) endoscopic treatment of airway obstruction, (3) surgical procedures to treat aspiration, (4) surgical procedures to improve voice, (5) tracheostomy, and (6) foreign body removal. Respondents were then asked to identify a target minimum annual number for surgical categories 1 to 4 for a program to perform to maintain competency (Table 6). There was general agreement across disciplines and programs regarding these targets, with the exception of 1 center recommending higher volumes for open or endoscopic procedures that directly increase the diameter of the airway and endoscopic treatment of airway obstruction. Comparison of median responses from that program to other ENT respondents showed a significant difference in these categories (median 25 vs 6 [IQR 10–35 vs 5–25] and 25 vs 15 [IQR 13.5–27.5 vs 12–25]; Mann–Whitney U test P = .006 and P = .044, respectively).

For this aim, participants were asked to list and then rank research areas that were of the greatest immediate importance and should be prioritized, as well as outcome measures of greatest importance to be used in aerodigestive research. The responses for top research priorities were stable with regards to ranking with the top 10 being cited by 55% to 97% of respondents and the second 10 by 9% to 36%, with rankings not changing from questionnaire 2 to 3 (Table 7). If taken as a group, issues related to aspiration (diagnosis, treatment, microbiome, and sequela) ranked in the top 10 for 91% of respondents. The responses for most important outcome measures were stable with regards to ranking with the top 10 being cited by 67% to 100% of respondents and the second 10 by 30% to 64% (Table 7). Sixty-one percent cited multicenter research as an essential function of aerodigestive programs, whereas 39% cited this as beneficial but nonessential.

Ninety-seven percent of participants stated that current understanding of which patients are appropriate for aerodigestive program and what an aerodigestive program does only exists within large academic centers. Eighty-one percent strongly agree that aerodigestive care is a definable model of care, distinct and distinguishable from routine specialty care for complex pediatric patients. To this point, 91% of respondents agreed (64% strongly agreed) that this definable care model was worthy of program certification, similar to the clinical care center models certified by the Cystic Fibrosis or Primary Ciliary Dyskinesia foundations. Respondents acknowledge that this is a future goal that must be founded on validated, outcome-based care guidelines. Ninety-seven percent of respondents agreed (67% strongly agreed) that aerodigestive care, as a defined subspecialty, was worthy of dedicated funding streams for research, and 88% strongly agreed that diagnostic coding should be modified to incorporate the time and complexity of delivering care in an integrated manner.

The results of this Delphi study indicate a broad consensus among providers from different subspecialties and across multiple geographic regions on the definitions, structure, functions, and priorities for aerodigestive care. In the context of limited published evidence, these results create a framework and an initial foundational definition for the model of pediatric aerodigestive care based on expert consensus.

There is evidence of the value and benefit of a coordinated consistent approach to the care of children with complex chronic conditions. Substantially improved outcomes for children with cystic fibrosis have been achieved through standardization of definitions, multicenter research, development and dissemination of clinical care guidelines, and tracking and reporting of patient outcomes.^18,–20 Similarly, the ImproveCareNow Network has improved outcomes for inflammatory bowel disease via a similar approach.^21,–23 Aerodigestive programs provide diagnostic evaluation, treatment, and often longitudinal coordinated care to complex pediatric patients, similar to pediatric cardiovascular centers, for which there are established, iteratively revised guidelines for diagnosis, treatment, and program composition.²⁴ Although accumulating evidence supports positive impacts of aerodigestive programs, development of a coordinated care model for this population is in its infancy. Further development of guidelines is aspirational and will need basis in evidence and proven outcomes.

This Delphi study leverages the knowledge and experience of a broad panel of subject matter experts; this is a specific strength of this process. Despite the range of disciplines, geography, and program history across the participant group, a supermajority consensus of 75% or stable median was achieved for all but 3 items, suggesting that our findings are robust. The panel remained split on the necessity for shared clinic visits to occur all together in the same physical space and on individual provider certification in aerodigestive care. Also, a recommendation for a target time frame for completion of an aerodigestive evaluation did not reach a supermajority (70%). An additional strength was the high response rate, with only 1 in 33 respondents failing to return 1 of 3 questionnaires.

Major limitations arise from a lack of solid evidence on which to grade these recommendations, relying instead on expert opinion. Until such evidence exists, there is no better alternative. The use of iterative questionnaires without open group discussion, is both a strength and a limitation. In this format there was some impairment in achieving deeper clarity in some responses that remain vague, such as “requirement for respiratory support” as an outcome measure. It is possible that this means something different to different respondents. The strength is that, because responses were pooled and anonymous, there was no single persuasive voice to dominate. We also recognize that the use of a 75% supermajority as the criterion for consensus differs from other Delphi studies. The original descriptions by the Rand Corporation did not specify criteria for consensus, and a wide range of criteria have been used in health care-related studies using this approach, suggesting that this method itself might benefit from standardization.

We believe that development of a robust and well-defined care model requires an initial step to define the population, structure and processes of the model. A trend toward formalizing and developing the aerodigestive care model is evident in the proliferation of programs, inclusion in the US News & World Report Best Pediatric Hospitals survey, the formation of aerodigestive interest groups at subspecialty meetings, development of an aerodigestive society, publication of aerodigestive-specific research, and the continued increase in attendance at aerodigestive conferences. These definitions are necessary for consistency in future research and development within aerodigestive care and, more pragmatically, to guide primary providers in determining which conditions and patients might benefit from care through such a model. We acknowledge that further refinements and development of care guidelines will require more rigorous evidence on many of the elements examined here and others outside of the scope of this study. Given that aerodigestive disorders encompass heterogeneous rare disease states, fulfillment of this aim will require coordinated research between centers, perhaps based on the priorities identified here. One could envision a roadmap to maturation of the aerodigestive care model similar to the development of the chILD Research Network for diffuse lung diseases or efforts led by the Cystic Fibrosis Foundation.^25,–29 The results of this study provide an initial crucial step toward this larger goal by providing definition and framework to the care model and identifying research goals and direction for further maturation.

On the basis of our findings, we suggest that there exists a definable aerodigestive care model, which merits further development and maturation, and which comprises specific personnel, infrastructure, research, and outcomes. Through a growing body of evidence, we suggest that this model delivers consistent, efficacious, cost-effective, outcomes-driven, patient-centered, and family-focused care. In the coming years, with continued effort, this may lead to documented improvement in outcomes, development and dissemination of care guidelines, standardization of approach, accreditation of aerodigestive care centers, an aerodigestive registry, and potentially aerodigestive-specific training opportunities.

 
• ENT

  ear, nose, and throat

 
• FEES

  fiberoptic endoscopic evaluation of swallowing

 
• IQR

  interquartile range

 
• VFSS

  videofluoroscopic swallow study