Cardiopulmonary Testing before Pediatric Adenotonsillectomy for Severe and Very Severe Obstructive Sleep Apnea Syndrome.

OBJECTIVES/HYPOTHESIS: Adenotonsillectomy is first-line treatment for pediatric obstructive sleep apnea syndrome (OSAS) when not otherwise contraindicated. There is concern severe OSAS increases risk of comorbid cardiopulmonary abnormalities, such as ventricular hypertrophy or pulmonary hypertension, which preoperative testing could detect. Our objective is to determine if there is a severity of pediatric OSAS where previously undetected cardiopulmonary comorbidities are likely. STUDY DESIGN: Retrospective chart review. METHODS: We performed a retrospective review of 358 patients ≤21 years with severe OSAS who underwent adenotonsillectomy at a tertiary hospital June 1, 2016 to June 1, 2018. We extracted demographics, comorbidities, polysomnography, and preoperative tests. Wilcoxon rank-sum and logistic regression estimated associations of OSAS severity (based on obstructive apnea-hypopnea index [OAHI], hypoxia, hypercarbia) with preoperative echocardiograms and chest X-rays (CXRs). RESULTS: Mean age was 5.9 (±3.6) years and 52% were male. Mean OAHI and oxygen saturation nadir were 30.3 (±23.8) and 80.7% (±9.2), respectively. OAHI ≥60 was associated with having a preoperative echocardiogram (OR, 3.8; 95% CI, 1.7-8.5) or CXR (OR, 3.0; 95% CI, 1.4-6.8) compared to OAHI 10-59. There were no significant associations between OSAS severity and test abnormalities. The presence of previously diagnosed cardiopulmonary comorbidities was associated with abnormalities on echocardiogram (OR, 36; 95% CI, 4.1-320.1) and CXR (OR, 4.1; 95% CI, 1.2-14.4). CONCLUSIONS: Although pediatric patients with very severe OSAS (OAHI ≥60) underwent more pre-adenotonsillectomy cardiopulmonary tests, OSAS severity did not predict abnormal findings. Known cardiopulmonary comorbidities may be a better indication for cardiopulmonary testing than polysomnographic parameters, which could streamline pre-adenotonsillectomy evaluation and reduce cost. LEVEL OF EVIDENCE: 4 Laryngoscope, 131:2361-2368, 2021.

Structure and Functions of Pediatric Aerodigestive Programs: A Consensus Statement.

Aerodigestive programs provide coordinated interdisciplinary care to pediatric patients with complex congenital or acquired conditions affecting breathing, swallowing, and growth. Although there has been a proliferation of programs, as well as national meetings, interest groups and early research activity, there is, as of yet, no consensus definition of an aerodigestive patient, standardized structure, and functions of an aerodigestive program or a blueprint for research prioritization. The Delphi method was used by a multidisciplinary and multi-institutional panel of aerodigestive providers to obtain consensus on 4 broad content areas related to aerodigestive care: (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. After 3 iterations of survey, consensus was obtained by either a supermajority of 75% or stability in median ranking on 33 of 36 items. This included a standard definition of an aerodigestive patient, level of participation of specific pediatric disciplines in a program, essential components of the care cycle and functions of the program, feeding and swallowing assessment and therapy, procedural scope and volume, research priorities and outcome measures, certification, coding, and funding. We propose the first consensus definition of the aerodigestive care model with specific recommendations regarding associated personnel, infrastructure, research, and outcome measures. We hope that this may provide an initial framework to further standardize care, develop clinical guidelines, and improve outcomes for aerodigestive patients.

Financial and Health Impacts of Multidisciplinary Aerodigestive Care.

OBJECTIVES: (1) Analyze upstream and downstream activity before and after enrollment with the Multidisciplinary Pediatric Aerodigestive Care Team (MPACT). (2) Identify potential demand for MPACT services with ICD-9 data. STUDY DESIGN: Retrospective review of financial claims data. SETTING: Tertiary care children's center. SUBJECTS: Pediatric patients (0-18 years old) enrolled with MPACT (pediatric otolaryngology, gastroenterology, pulmonary, speech-language pathology). METHODS: Case mix data from fiscal years (FYs) 2010-2013 were analyzed for primary, secondary, and tertiary ICD-9 codes in 4 aerodigestive diagnostic categories (ADCs): dysphagia, chronic cough, gastroesophageal disease, and chronic pulmonary disease/asthma. Inclusion criteria included patients <18 years old, seen by MPACT, with FY2010-FY2013 case mix data and ≥2 ADCs. Unique outpatient and inpatient encounters and associated charges were evaluated to determine upstream and downstream activity trends. RESULTS: Of the 126 patients meeting inclusion criteria, 55 (44%) had ≥3 ADCs, and 11 (9%) had 4. These 126 patients received outpatient care during 3068 unique encounters. Outpatient total charges were $282,102 before and $744,542 after MPACT intervention. Eighty-six (68%) patients received inpatient care during 423 unique encounters. Inpatient charges were $4,257,137 before and $2,872,849 after MPACT enrollment. Overall, a net reduction of $921,848 in total charges, $7316 per MPACT patient, was noted. FY2010-FY2014 data identified an additional 1728 pediatric patients with ≥2 ADCs not enrolled in MPACT. CONCLUSION: A cohort of children with aerodigestive disease experienced a shift from inpatient to outpatient care with an overall 20% reduction in patient charges when the years before and after MPACT enrollment were compared. Available ICD-9 data suggest potential demand for MPACT services.

Airway obstruction after lingual frenulectomy in two infants with Pierre-Robin Sequence.

Pierre-Robin Sequence (PRS) is defined as the triad of micrognathia, glossoptosis, and cleft palate and affects approximately 1/8500 births. Airway obstruction is common in infants with PRS and results from glossoptosis leading to pharyngeal obstruction. Any procedure that increases the severity of glossoptosis, such as lingual frenulectomy, may increase the risk of obstruction or aggravate existing obstruction. This report discusses two cases of significant airway decompensation after lingual frenulectomy requiring surgical intervention in infants with PRS. We suggest that lingual frenulectomy be contraindicated in infants with PRS or suspected PRS due to the possible increased risk of airway obstruction.

Interdisciplinary pediatric aerodigestive care and reduction in health care costs and burden.

IMPORTANCE: Children with complex respiratory and gastrointestinal disorders frequently require care from multiple pediatric subspecialists. Interdisciplinary pediatric aerodigestive clinic centers have been established at several pediatric tertiary care centers in the United States. Their effectiveness is unknown. OBJECTIVE: To determine whether an interdisciplinary approach to pediatric aerodigestive disorders reduces health care costs and burden. DESIGN, SETTING, AND PARTICIPANTS: Retrospective medical record review for the first 125 unique pediatric patients (median age, 1.51 years) seen at the Pediatric Aerodigestive Center (PAC) for aerodigestive disorders between June 2010 and August 2013 for a total of 163 outpatient clinical encounters. The PAC is an academic pediatric tertiary care center where specialists in gastroenterology, otolaryngology, pulmonology, and speech-language pathology provide interdisciplinary evaluation during a single clinic encounter and combined operative management when indicated. INTERVENTIONS: Interdisciplinary evaluation and treatment of pediatric aerodigestive disorders. MAIN OUTCOMES AND MEASURES: Number of operative procedures and estimated hospital charges related to combining clinic encounters and operative procedures. RESULTS: During the initial visit, each of the 125 patients received a mean (SD) of 2.9 (0.8) of a potential 4 services. Estimating per-encounter visit costs for gas, parking, and facility fees, we found that the average cost savings per family per PAC visit was $182. Evaluation under anesthesia was recommended for 85 patients (68%), resulting in 267 operative procedures performed by participating services during 158 encounters with general anesthesia. Thus, 109 episodes of anesthesia were avoided (41% reduction), reducing the risks of anesthesia and related care costs ($1985 per avoided episode). CONCLUSIONS AND RELEVANCE: Coordination of interdisciplinary care in the PAC resulted in a reduction of hospital charges realized through reduction in clinic- and anesthesia-related visits. Reductions in episodes of anesthesia may also reduce neurocognitive risks associated with multiple anesthetic exposures. Other nontangible benefits associated with the coordination of care, such as caregiver satisfaction, warrant further study.

Hospitalizations in children with preexisting tracheostomy: a national perspective.

OBJECTIVES/HYPOTHESIS: To describe the reasons for hospitalization and characteristics of children with preexisting tracheostomy and to compare hospital utilization between children with and without tracheostomy. STUDY DESIGN: Retrospective cohort study. METHODS: Children with tracheostomy were selected in the Healthcare Cost and Utilization Project Kids' Inpatient Database 2009 using International Classification of Diseases, Ninth Revision, Clinical Modification codes. We compared hospital utilization with the children's clinical characteristics (e.g., chronic condition number and type). We also assessed hospitalizations for tracheostomy complications and ambulatory care sensitive conditions (ACSCs) that could be potentially influenced by high-quality outpatient and community care delivery. RESULTS: In 2009, there were 21,541 hospitalizations for children with tracheostomy totalling $1.4 billion (U.S.). On average, children with tracheostomy had five chronic conditions (standard deviation 1.4). Eighty-one percent (n = 17,448) had one or more complex chronic conditions (CCCs), and 67.1% (n = 14,379) had a gastrostomy. Among children with one or more CCCs, mean hospital charges were greater for hospitalizations of children with tracheostomy compared to without ($69,999 vs. $64,017, P = 0.008). Twenty-one percent (n = 4,421) of all hospitalizations of children with tracheostomy were due to an ACSC (14.5%, n = 3,122) or a tracheostomy complication (6.0%, n = 1,299). Bacterial pneumonia (9.6% of all hospitalizations, n = 2,059) was the most common ACSC. CONCLUSIONS: Children with tracheostomy are a vulnerable group of children with multiple CCCs who experience lengthy and costly hospitalizations. Many hospitalizations are due to an ambulatory care sensitive condition or a tracheostomy complication. Further investigation is needed to determine whether some of these hospitalizations may be avoidable with improved outpatient and community tracheostomy care. LEVEL OF EVIDENCE: 2b.

The association between laryngeal cleft and tracheoesophageal fistula: myth or reality?

OBJECTIVES/HYPOTHESIS: Laryngeal cleft (LC) associated with tracheoesophageal fistula (TEF) with or without esophageal atresia (EA) has rarely been described. The purpose of this study is to review our experience, clinical features, management, delay in diagnosis, and complications in children with these anomalies. STUDY DESIGN: Retrospective chart review at pediatric tertiary referral center. METHODS: Patients diagnosed with LC alone or LC and TEF over a 10-year period were included. Data including demographics, type of TEF and LC, comorbidities, symptoms, management, complications and swallowing outcomes were analyzed. RESULTS: There were 161 pediatric patients diagnosed with LC alone and 22 with LC and TEF. In patients with LC and TEF, aspiration was the most common presenting symptom (n = 11, 50%). Seventeen patients (77%, mean age 4 years 7 months) underwent endoscopic repair and five patients (23%) with type I clefts did not require surgery. Two patients required revision surgery. For patients with LC alone, the mean age at repair was 3.70 years (4 months-19.9 years) compared to 4.69 years (8 months-17.83 years) for patients with LC and TEF (P = 0.0187). The postoperative swallowing studies from 15 patients showed no aspiration. Mean follow-up was 4 years and 6 months. CONCLUSION: The diagnosis and management of LC in patients with TEF is often delayed. If a child presents with persistent aspiration after TEF repair, a complete airway endoscopy should be performed to evaluate for vocal fold mobility and cleft. Endoscopic repair is the recommended approach for those patients requiring surgical intervention. LEVEL OF EVIDENCE: 4.

Acquired Glottic Web Formation in a 3-Year-Old Girl: Case Report and Literature Review.

Pediatric glottic webs are most commonly congenital in origin. Acquired webs in children are typically traumatic in nature, primarily from external trauma or intubation. We report a unique variation of this post-traumatic etiology: a web centered at the junction of the anterior one-third and posterior two-thirds of the vocal folds, possibly formed after phonotraumatic stress from severe coughing. Our case suggests that careful attention should be paid to persistent voice changes after acute illness, as this may be a sign of laryngeal web in the setting of phonotraumatic stress.

Mortality and major morbidity after tonsillectomy: etiologic factors and strategies for prevention.

OBJECTIVE/HYPOTHESIS: To report data on death or permanent disability after tonsillectomy. STUDY DESIGN: Electronic mail survey. METHODS: A 32-question survey was disseminated via the American Academy of Otolaryngology-Head and Neck Surgery electronic newsletter. Recipients were queried regarding adverse events after tonsillectomy, capturing demographic data, risk factors, and detailed descriptions. Events were classified using a hierarchical taxonomy. RESULTS: A group of 552 respondents reported 51 instances of post-tonsillectomy mortality, and four instances of anoxic brain injury. These events occurred in 38 children (71%), 15 adults (25%), and two patients of unstated age (4%). The events were classified as related to medication (22%), pulmonary/cardiorespiratory factors (20%), hemorrhage (16%), perioperative events (7%), progression of underlying disease (5%), or unexplained (31%). Of unexplained events, all but one occurred outside the hospital. One or more comorbidities were identified in 58% of patients, most often neurologic impairment (24%), obesity (18%), or cardiopulmonary compromise (15%). A preoperative diagnosis of obstructive sleep apnea was not associated with increased risk of death or anoxic brain injury. Most events (55%) occurred within the first 2 postoperative days. Otolaryngologists who reported performing <200 tonsillectomies per year were more likely to report an event (P < .001). CONCLUSIONS: This study, the largest collection of original reports of post-tonsillectomy mortality to date, found that events unrelated to bleeding accounted for a preponderance of deaths and anoxic brain injury. Further research is needed to establish best practices for patient admission, monitoring, and pain management. LEVEL OF EVIDENCE: N/A.

Tracheotomy-related catastrophic events: results of a national survey.

OBJECTIVES/HYPOTHESIS: To gather qualitative and semiquantitative information about catastrophic complications during and following tracheotomy. STUDY DESIGN: National survey distributed to American Academy of Otolaryngology-Head and Neck Surgery members via the Academy weekly email newsletter during April and May 2011. METHODS: A total of 478 respondents provided estimates of the number of four specific tracheotomy-related complications (innominate artery fistula, esophageal fistula, acute tracheotomy occlusion, and obstructing granuloma), all catastrophic events, and events resulting in death or permanent disability encountered during their careers. There were 253 respondents who provided 405 free-text descriptions of specific events. RESULTS: The respondents experienced approximately one catastrophic event every 10 years and one event resulting in death or permanent disability every 20 years. More than 90% occurred more than 1 week after surgery. Categories of physicians who experienced more events per year included academic physicians and laryngologists. Pediatric otolaryngologists had twice as many innominate artery fistulas per year of practice as others. Qualitative (free-text) descriptions of the most serious events demonstrated that more of these events involved loss of airway and volume bleeds, usually from innominate or carotid artery erosion. Many of the events due to airway loss involved potentially correctable deficits in family education, nursing care, home care, and other structural factors. CONCLUSIONS: Even when we allow for selection bias, these data suggest that a substantial number of tracheotomy complications leading to death or permanent disability occur at a national level. The vast majority of events occur more than 1 week after the procedure. Many of the described events were caused by factors that should be amenable to prospective system improvement strategies.

Unverifiable publications in otolaryngology residency applications.

OBJECTIVE: To determine the prevalence of unverifiable ("ghost") publications in applications to an otolaryngology residency program through the Electronic Residency Application Service (ERAS), correlate with applicant characteristics, and determine if incidence changed after the addition of PubMed (PMID) numbers in 2008. STUDY DESIGN AND SETTING: Cross-sectional study of residency applications before and after inclusion of PMID numbers at an academic otolaryngology program. SUBJECTS AND METHODS: Applications for 2007 and 2008 were reviewed. Publications were verified against Medline, Google Scholar, PubMed, ISI Web of Science, and Google. Ghost publications were defined as journals, books, abstracts, or posters that could not be verified as presented, published, or including the applicant author. RESULTS: In total, 489 applications were reviewed: 243 before PMID numbers were requested and 246 after. Of 2300 listed publications, 125 (5%) were not actual publications and 460 (20%) were in pending status. Forty-five percent (775/1715) could not be verified: 660 of 953 (69%) abstracts/posters, 18 of 47 (38%) chapters, and 97 of 715 (14%) journal articles. Abstracts/posters and book chapters were hardest to verify. The proportion of overall reported publications that could be verified was lower following the addition of PMID to the ERAS application (P = .0003), and the proportion of verifiable journal articles was unchanged from 86.0% to 86.9% (P = .62). Unlike previous findings, gender and medical school ranking were not associated with ghost publications. CONCLUSION: A substantial number of publications, especially book chapters and posters/abstracts, listed on otolaryngology residency applications could not be verified. The addition of the PMID to applications did not reduce the number of ghost journal publications.

Development and pilot testing of an operative competency assessment tool for pediatric direct laryngoscopy and rigid bronchoscopy.

OBJECTIVES: To develop and pilot test an objective assessment of technical skills instrument for evaluation of residents' surgical performance of pediatric direct laryngoscopy and rigid bronchoscopy, with emphasis on feasibility, validity, and interrater agreement. STUDY DESIGN: Prospective, unblinded educational quality-improvement project combining paired and unpaired observations. METHODS: Objective Structured Assessment of Technical Skills (OSATS) evaluation instruments were developed from detailed faculty input. Two instruments were created: a surgical checklist and a global assessment of surgical performance. The instruments were tested in a simulation bronchoscopy course and in the operative suite. Paired observations were used to determine interrater agreement, whereas multiple evaluations were used to calculate construct validity and internal consistency. RESULTS: Forty-four assessments were completed for 19 residents. Seven faculty members evaluated residents as they performed laryngoscopy and bronchoscopy in an animal simulation laboratory and in the operating room. The evaluation tool was found to be feasible. The interrater agreement for both instruments was significant at 80.4% to 84.6% (P ≤.0001). Construct validity was confirmed with increasing mean global and task specific scores by postgraduate year (P <.0001). Internal consistency, measured with Cronbach alpha, was high at 0.968. CONCLUSIONS: This pilot study suggests that a reliable and valid instrument for objective evaluation of surgical competency can be developed for pediatric direct laryngoscopy and rigid bronchoscopy. This instrument can be used for formative and summative feedback of operative performance. In addition, it was easy to use and valid in a limited evaluation; however, larger studies are required to validate its utility.

Ludwig's angina following frenuloplasty in an adolescent.

Ludwig's angina is a rapidly progressive cellulitis of the submandibular space and has the potential for significant upper airway obstruction. Most reported cases follow an odontogenic infection. We present the case of a 13-year-old girl who underwent a frenuloplasty to correct speech disturbances and subsequently developed a life-threatening infection of the floor of mouth. Immediate intubation, surgical decompression and antibiotic therapy successfully resolved the episode. To our knowledge, this is the first report of an iatrogenic Ludwig's angina attributable to a frenuloplasty performed for ankyloglossia. We briefly review the literature on ankyloglossia, pediatric Ludwig's angina and postoperative infections.

Innate and adaptive mediators in cystic fibrosis and allergic fungal rhinosinusitis.

BACKGROUND: Surfactant-associated proteins (SP) A and D are both innate immunity mediators and produced in normal and diseased sinus mucosa. Cystic fibrosis (CF) is associated with Th1 adaptive inflammation whereas allergic fungal rhinosinusitis (AFRS) is associated with Th2 adaptive inflammation. The purpose of this study is to show and quantify the presence of SP A, SP D, tumor necrosis factor (TNF) alpha, (a Th1 marker), and eotaxin (a Th2 marker) in normal and diseased sinus mucosa. METHODS: Intraoperative sinus mucosal biopsy specimens from human volunteers were obtained during endoscopic sinus surgery for CF (n = 4), AFRS (n = 10), and normal controls (CTLs; n = 4). Specimens were evaluated for presence and quantity of SP A, SP D, and TNF-alpha using Western blot with semiquantitative immunoblot analysis. Eotaxin was quantified using ELISA immunoassay. Results were standardized and reported as picograms of mediator per microgram of total protein. RESULTS: SP A, SP D, and TNF-alpha levels in CF tissue extracts were 2-10 times higher than levels in AFRS tissue (with SP D and TNF-alpha reaching statistical significance) but CF tissue was not significantly higher than CTL tissue. SP A, SP D, and TNF-alpha were not significantly elevated in AFRS. Eotaxin showed elevated levels in CF and AFRS when compared with CTLs (p = 0.03 and 0.003, respectively). CONCLUSION: SP D and TNF-alpha are significantly increased in CF compared with AFRS, suggesting activation of both innate immunity and Th1-mediated inflammation and potential correlation between SPs and downstream adaptive immune responses.

Laryngopharyngeal dysfunction after the Norwood procedure.

OBJECTIVE: We sought to evaluate the incidence and significance of recurrent laryngeal nerve and swallowing dysfunction after a Norwood procedure compared with that after biventricular aortic arch reconstruction. METHODS: From April 2003 through December 2004, 36 neonates underwent a Norwood procedure; 33 of 36 had postoperative fiberoptic laryngoscopy and modified barium swallow. Study results were used to guide the transition from nasogastric tube to oral feeding and placement of gastrostomy tubes. During the same time period, 18 neonates underwent aortic arch reconstruction as part of a biventricular repair. RESULTS: After a Norwood procedure, laryngoscopy showed left true vocal fold (cord) paralysis in 3 (9%) of 33 patients. The results of a modified barium swallow were abnormal in 16 (48%) of 33 patients, with aspiration in 8 (24%) of 33 patients. Of the 3 patients with vocal fold paralysis, 2 had a normal modified barium swallow result, and 1 had aspiration. Gastrostomy tubes were placed in 6 (18%) of 33 patients, all with an abnormal modified barium swallow result. Hospital stay was longer in patients with an abnormal modified barium swallow result: 34 +/- 13 versus 22 +/- 7 days (P < .01). After biventricular repair with aortic arch reconstruction, left true vocal fold paralysis occurred in 4 (25%) of 16 patients; results of a modified barium swallow were abnormal in 10 (59%) of 17 patients, with aspiration in 6 (35%) of 17 patients (all nonsignificant vs patients undergoing the Norwood procedure). Follow-up laryngoscopy in 4 patients with vocal fold paralysis showed no change in 3 of 4 patients and improvement in 1 patient. Follow-up modified barium swallow showed resolution of aspiration in 11 (85%) of 13 patients. Hospital survival was 32 (89%) of 36 patients for the Norwood procedure and 18 (100%) of 18 patients for biventricular repair. There has been 1 sudden death before second-stage palliation. CONCLUSIONS: After a Norwood procedure, swallowing dysfunction occurs in 48% of patients, with aspiration in 24%, and results in increased length of hospital stay. Left recurrent laryngeal nerve injury, seen in 9% of patients, is an uncommon cause of swallowing dysfunction. Postoperative aspiration generally resolves over time, whereas vocal fold paralysis does not. Systematic evaluation of swallowing function allows appropriate tailoring of feeding regimens and might contribute to decreased hospital and interstage mortality.