ABSTRACT
Background: Congenital malformations of the trachea are rare but often life-threatening. Limited data have been published on the outcomes of tracheal reconstruction for congenital tracheal stenosis. We sought to describe the outcomes of patients undergoing tracheal reconstruction over 10 years at our centre. Methods: All paediatric patients who underwent long-segment tracheal or bronchial reconstruction from January 1, 2012, to August 31, 2022, were included. The primary outcome was mortality, and secondary outcomes included reoperation and postoperative morbidity. Patients were followed up to 10 years. Results: Thirty-three patients with ages ranging from 1 day to 12 years (mean 8.5 months) at the time of tracheoplasty or bronchoplasty were included, with 5 patients undergoing off-pump tracheal reconstruction. The most common preoperative comorbidities included patent ductus arteriosus (30.3%), atrial septal defect (27.3%), and prematurity (24.2%). There were no deaths postoperatively within the follow-up period. All patients experienced successful reconstruction with no patients requiring reoperation of the trachea. A total of 14 patients (42.4%) required postoperative balloon dilation, 3 (9.1%) required bronchial repair after tracheal repair, and 2 (6.1%) required bronchoscopic tracheal debridement. Conclusions: This single-centre retrospective study provides a large cohort of congenital tracheal reconstruction patients with a survival rate of 100%, experiencing no mortality during follow-up. The majority of patients had preoperative comorbidities and concomitant congenital cardiac defects. Although tracheal reconstruction continues to be complex with significant postoperative morbidity and mortality, the results of our single-centre study demonstrate the continual advancement of this field and the evolving improvement of postoperative outcomes for these patients.
Contexte: Les malformations congénitales de la trachée sont rares, mais elles menacent souvent le pronostic vital. Peu de données ont été publiées sur les résultats cliniques de la reconstruction trachéale dans les cas de sténose trachéale congénitale. Notre objectif était de décrire les résultats obtenus par les patients ayant subi une reconstruction trachéale dans notre établissement sur une période de 10 ans. Méthodologie: Les données de tous les enfants ayant subi une reconstruction d'un long segment trachéal ou bronchique entre le 1er janvier 2012 et le 31 août 2022 ont été incluses dans l'étude. Le critère principal d'évaluation était la mortalité et les critères secondaires d'évaluation incluaient la réintervention chirurgicale et la morbidité postopératoire. Les patients ont été suivis pendant 10 ans. Résultats: Trente-trois patients âgés de 1 jour à 12 ans (moyenne de 8,5 mois) au moment de la trachéoplastie ou de la bronchoplastie ont été inclus dans l'étude, dont 5 ayant subi une reconstruction trachéale sans circulation extracorporelle (off-pump). Avant l'intervention, les affections concomitantes les plus fréquentes étaient la persistance du canal artériel (30,3 %), la communication interatriale (27,3 %) et la prématurité (24,2 %). Aucun décès postopératoire n'est survenu au cours de la période de suivi. La reconstruction a été un succès chez tous les patients, et aucun d'entre eux n'a dû subir de nouvelle opération trachéale. Une dilatation postopératoire par ballon a été nécessaire chez 14 patients (42,4 %), 3 d'entre eux (9,1 %) ont subi une correction chirurgicale des bronches après la reconstruction trachéale, et 2 (6,1 %) ont subi un débridement trachéal par bronchoscopie. Conclusions: Notre étude rétrospective monocentrique dresse le portrait d'une vaste cohorte de patients ayant subi une reconstruction de la trachée en raison d'une malformation congénitale, parmi laquelle le taux de survie était de 100 %, sans aucun décès au cours de la période de suivi. Par ailleurs, la majorité des patients présentaient, avant l'intervention, des affections et des malformations cardiaques concomitantes. La reconstruction trachéale demeure malgré tout complexe et les taux de mortalité et de morbidité postopératoires restent importants, mais les résultats de notre étude monocentrique témoignent des progrès continuels dans ce domaine et de l'amélioration des résultats postopératoires pour ces patients.
ABSTRACT
OBJECTIVE: To provide recommendations for a comprehensive management approach for infants and children presenting with symptoms or signs of aspiration. METHODS: Three rounds of surveys were sent to authors from 23 institutions worldwide. The threshold for the critical level of agreement among respondents was set at 80 %. To develop the definition of "intractable aspiration," each author was first asked to define the condition. Second, each author was asked to complete a 5-point Likert scale to specify the level of agreement with the definition derived in the first step. RESULTS: Recommendations by the authors regarding the clinical presentation, diagnostic considerations, and medical and surgical management options for aspiration in children. CONCLUSION: Approach to pediatric aspiration is best achieved by implementing a multidisciplinary approach with a comprehensive investigation strategy and different treatment options.
Subject(s)
Otolaryngology , Infant , Child , Humans , Consensus , Surveys and Questionnaires , Delphi TechniqueABSTRACT
OBJECTIVES: To develop consensus statements for the scoring of pediatric drug induced sleep endoscopy in the diagnosis and management of pediatric obstructive sleep apnea. METHODS: The leadership group identified experts based on defined criteria and invited 18 panelists to participate in the consensus statement development group. A modified Delphi process was used to formally quantify consensus from opinion. A modified Delphi priori process was established, which included a literature review, submission of statements by panelists, and an iterative process of voting to determine consensus. Voting was based on a 9-point Likert scale. Statements achieving a mean score greater than 7 with one or fewer outliers were defined as reaching consensus. Statements achieving a mean score greater than 6.5 with two or fewer outliers were defined as near consensus. Statements with lower scores or more outliers were defined as no consensus. RESULTS: A total of 78 consensus statements were evaluated by the panelists at the first survey - 49 achieved consensus, 18 achieved near consensus, and 11 did not achieve consensus. In the second survey, 16 statements reached consensus and 5 reached near consensus. Regarding scoring, consensus was achieved on the utilization of a 3-point Likert scale for each anatomic site for maximal observed obstructions of <50% (Score 0, no-obstruction), ≥ 50% but <90% (Score 2, partial obstruction), and ≥ 90% (Score 3, complete obstruction). Anatomic sites to be scored during DISE that reached consensus or near-consensus were the nasal passages, adenoid pad, velum, lateral pharyngeal walls, tonsils (if present), tongue base, epiglottis, and arytenoids. CONCLUSION: This study developed consensus statements on the scoring of DISE in pediatric otolaryngology using a modified Delphi process. The use of a priori process, literature review, and iterative voting method allowed for the formal quantification of consensus from expert opinion. The results of this study may provide guidance for standardizing scoring of DISE in pediatric patients.
Subject(s)
Endoscopy , Sleep Apnea, Obstructive , Child , Humans , Endoscopy/methods , Pharynx , Polysomnography/methods , Sleep , Sleep Apnea, Obstructive/diagnosisABSTRACT
OBJECTIVE: (1) Compare proportions of collapse, obstruction, or mixed instances on drug-induced sleep endoscopy findings of obese and nonobese children with obstructive sleep disordered breathing. (2) Determine the frequency of collapse in general between both groups. STUDY DESIGN: Retrospective case-control study. SETTING: Tertiary pediatric center. METHODS: Obese (body mass index >95 percentile) children presenting with obstructive sleep disordered breathing (>33 on the pediatric sleep questionnaire) were identified from a prospectively kept surgical database. Only those who had undergone drug-induced sleep endoscopy were eligible. Age and sex pair-matched nonobese children were identified. Only nonsyndromic, neurologically normal, surgically naïve patients were included. The frequency of obstructive, collapse, and mixed pharyngeal patterns was documented in both groups. A comparison of proportions was then undertaken (χ2 test). RESULTS: Over a 5-year period, 73 consecutive children with obesity were identified (40 males; mean of 8.5 ± 3.0 years, 2.8-13.1). They were matched with 73 nonobese children (8.4 ± 3.0 years, 2.6-14.1). The obese group exhibited significantly more pharyngeal collapses (62:47) (p = .0021 odds ratio [OR] 3.358, 95% confidence interval [CI] 1.52-7.42). The proportion of pharyngeal findings on drug-induced sleep endoscopy was significantly different (p = .000129) between the 2 groups; obese (61 mixed: 3 obstruction: 9 collapse) and nonobese (48 mixed: 22 obstruction: 4 collapse). CONCLUSION: The predominance of hypopharyngeal collapse in children with obesity may explain the likelihood of failure of surgery directed at obstructive findings. This may also strengthen the case for drug-induced sleep endoscopy in this group at the initial surgery to guide it rather than after the failure of adenotonsillectomy.
Subject(s)
Airway Obstruction , Pharyngeal Diseases , Sleep Apnea Syndromes , Sleep Apnea, Obstructive , Male , Humans , Child , Retrospective Studies , Case-Control Studies , Polysomnography , Sleep Apnea, Obstructive/surgery , Endoscopy , Sleep Apnea Syndromes/surgery , Sleep , Obesity/complications , Obesity/epidemiology , Pharyngeal Diseases/diagnostic imaging , Pharyngeal Diseases/etiologyABSTRACT
OBJECTIVE: Despite being a more accessible and less resource intensive modality than polysomnography, the utility of sleep oximetry (SO) in pediatric laryngomalacia (LM) is not well understood or validated. We aimed to retrospectively evaluate the utility of overnight home based SO in children with LM by developing and internally validating the Modified Laryngomalacia Oximetry Score (MLOS) scoring system to triage severity and guide clinical decision making. METHODS: We evaluated pediatric patients with a diagnosis of LM at our tertiary referral centre. Data from initial and post-treatment SO including mean oxygen saturation (spO2) nadir and mean oxygen desaturation index (ODI) were aggregated. The MLOS ranging from I-VI (inconclusive to severe) was created by two otolaryngologists to incorporate bradycardia associated desaturation events during SO. Corresponding McGill Oximetry Score (MOS) was also determined. RESULTS: 172 patients were included in final analysis. The average age was 9.2 ± 14.3 months. 98 (57%) of patients were identified as Thompson severity score 1, and 87 (50.6%) of patients underwent supraglottoplasty. The surgical cohort had a significantly higher MLOS and MOS scores of 4 and 2 respectively, and higher mean ODI and spO2 nadir metrics. When evaluating post-supraglottoplasty SO tracings, all parameters improved significantly, including median MLOS score from 4 to 1. Only the mean ODI improved in the non-surgical cohort. Patients with Thompson severity score 2/3 had significantly higher MLOS. CONCLUSION: We present a simple scoring system based on overnight SO, the MLOS, to help triage severity of pediatric LM and guide decision-making. MLOS is associated with worse clinical severity and a need for surgery, and shows significant improvement after surgery.
Subject(s)
Laryngomalacia , Sleep Apnea, Obstructive , Child , Humans , Infant , Laryngomalacia/complications , Laryngomalacia/diagnosis , Laryngomalacia/surgery , Oximetry , Oxygen , Retrospective Studies , Sleep , Sleep Apnea, Obstructive/surgeryABSTRACT
OBJECTIVE: Nasal septum cartilage is a hyaline cartilage that provides structural support to the nasal cavity and midface. Currently, information on its cellular and mechanical properties is widely dispersed and has often been inferred from studies conducted on other cartilage types such as the knee. A detailed understanding of nasal cartilage properties is important for several biological, clinical, and engineering disciplines. The objectives of this scoping review are to (1) consolidate actual existing knowledge on nasal cartilage properties and (2) identify gaps of knowledge and research questions requiring future investigations. DESIGN: This scoping review incorporated articles identified using PROSPERO, Cochrane Library (CDSR and Central), WOS BIOSIS, WOS Core Collection, and ProQuest Dissertations and Theses Global databases. Following the screening process, 86 articles were considered. Articles were categorized into three groups: growth, extracellular matrix, and mechanical properties. RESULTS: Most articles investigated growth properties followed by extracellular matrix and mechanical properties. NSC cartilage is not uniform. Nasal cartilage growth varies with age and location. Similarly, extracellular matrix composition and mechanical properties are location-specific within the NSC. Moreover, most articles included in the review investigate these properties in isolation and only very few articles demonstrate the interrelationship between multiple cartilage properties. CONCLUSIONS: This scoping review presents a first comprehensive description of research on NSC properties with a focus on NSC growth, extracellular matrix and mechanical properties. It additionally identifies the needs (1) to understand how these various cartilage properties intersect and (2) for more granular, standardized assessment protocols to describe NSC.
Subject(s)
Hyaline Cartilage , Nasal Cartilages , Extracellular Matrix , Knee JointABSTRACT
Congenital tracheal stenosis is a rare but life-threatening malformation of the trachea. Surgical reconstruction is high risk, and not frequently performed in neonates born of extreme prematurity and low birth weight. We present the case of an extremely premature 950-gram neonate with severe congenital tracheal stenosis who underwent tracheal reconstruction. Complete repair, with no residual stenosis, was achieved with slide tracheoplasty without the need for cardiopulmonary bypass.
Subject(s)
Plastic Surgery Procedures , Tracheal Stenosis , Constriction, Pathologic/surgery , Humans , Infant , Infant, Newborn , Retrospective Studies , Trachea/abnormalities , Trachea/surgery , Tracheal Stenosis/congenital , Tracheal Stenosis/diagnosis , Tracheal Stenosis/surgery , Treatment OutcomeABSTRACT
OBJECTIVE: The literature on the entity of the anterior larynx (AL) is restricted to little evidence on the difficulty encountered in exposing the larynx for intubation, perioperative morbidity and mortality, and scant reports on its prevalence in general adults and children. Here, we describe the prevalence of AL in a series of children presenting with aerodigestive symptoms and explore its association with functional abnormalities, congenital and structural anomalies or conditions. SETTING: Tertiary paediatric centre. METHODS: We conducted a retrospective case-control study. Using a prospectively collected surgical database, we identified patients diagnosed with AL (Grades IIb-III-IV Modified Cormack-Lehane scale) and sex and age-matched controls who had undergone full airway endoscopy for aerodigestive symptoms. We collected the demographics, presentations, associated diagnoses and investigations. We compared the proportion of patients with large airway abnormalities and dysmorphism and estimated the prevalence of AL. RESULTS: Over a 5.5-year period, 58 children with AL (28 females, mean age 0.38 years) were matched with 58 controls (mean age 0.42 years). Although both groups presented with permutations of stridor, respiratory failure, cyanotic spells, swallowing and feeding difficulties and SDB, AL presented much more with swallowing and feeding problems (33 vs. 20, p < .05). There were significantly more children with dysmorphism in the AL group (29 vs. 9, p < .05). The prevalence of AL was 4.9% (SE 0.0063). CONCLUSIONS: AL was detected in 4.9% of a sample of children who had undergone airway examination for aerodigestive symptoms. It is significantly associated with dysmorphism, and swallowing difficulties that warrant instrumental evaluation.
Subject(s)
Larynx/abnormalities , Case-Control Studies , Child , Child, Preschool , Deglutition Disorders , Female , Humans , Infant , Infant, Newborn , Intubation , Laryngoscopy , Male , Retrospective Studies , Sleep Wake DisordersABSTRACT
OBJECTIVES: There has been increasing interest in the management of oropharyngeal swallowing dysfunction (SwD). Its prevalence, particularly in otherwise healthy infants and toddlers (OHITs), is underappreciated. As the standard diagnostic tests are either invasive or scarce, valid parent-reported outcome (PRO) questionnaires could play a pivotal role in the understanding and managing SwD in this group. This article reviewed the literature on PRO questionnaires pertaining to SwD in OHITs. DATA SOURCE: A librarian searched Prospero, Cochrane Library, Embase, Medline, PsycINFO, HaPI, CINAHL, and SCOPUS until February 2021 using the MeSH terms for deglutition and screening methods. REVIEW METHOD: Questionnaires that examined disease-specific or eating and feeding concerns or difficulties were excluded. Two reviewers independently identified PRO questionnaires for SwD that were used in OHITs and extracted the author names, publication year, questionnaire name, the studied population, and the reported psychometric assessments. A quality assessment was performed based on consensus-based standards for the selection of health measurement instruments (COSMIN) and updated criteria for good measurement properties. RESULTS: Of the 3488 screened articles, we identified only two questionnaires, the pediatric version of the Eating Assessment Tool (PEDI-EAT-10) and the PRO questionnaire for Swallowing Dysfunction in OHITs. The PEDI-EAT-10 authors assessed the validity and reliability on children with cerebral palsy. However, concerns were identified regarding the developmental process and the internal structure validity. The PRO questionnaire for SwD in OHITs meets criteria but has not yet been validated in the population of interest nor its psychometric properties assessed. CONCLUSION: Two instruments were identified. The PED-EAT-10 exhibits methodological flaws, while Edmonton PRO questionnaire for SwD in OHITs awaits construct validation and could fill the current knowledge gap.
Subject(s)
Deglutition , Parents , Child , Child, Preschool , Humans , Infant , Psychometrics , Reproducibility of Results , Surveys and QuestionnairesABSTRACT
BACKGROUND: To evaluate the clinical presentation of choanal atresia (CA) in tertiary centers across Canada. METHODS: Multi-centre case series involving six tertiary care pediatric hospitals across Canada. Retrospective chart review of patients born between 1980 and 2010 diagnosed with CA at a participating center. RESULTS: The health charts of 215 patients (59.6% female) with CA were reviewed and included in this study. The mean age of patients at time of CA presentation was 0.4 months (range 0.1 to 7.2 months) for bilateral CA and 37.8 months (range 0.1 to 164.1 months) for unilateral cases. The most common presenting symptoms for bilateral CA in decreasing order were respiratory distress (96.4%), feeding difficulties (68.2%), and rhinorrhea (65.5%), and for unilateral cases in decreasing order were rhinorrhea (92.0%), feeding difficulties (24.7%), and respiratory distress (18.0%). For the majority of patients (73.2%), the obstruction comprised mixed bony and membranous tissue, with only 10.5% presenting with a purely membranous obstruction. Familial history of CA was confirmed in only 3.3% of cases. One half of patients with CA presented with one or more associated anomalies and 30.6% had a syndrome. CONCLUSIONS: The present investigation is the first national multi-institutional study evaluating the clinical presentation of CA over three decades. The present cohort of CA patients presented with a breadth of co-morbidities with highly variable presentations, with bilateral cases being more severely affected than unilateral cases. Further investigation into hereditary linkages to CA development is warranted.
Subject(s)
Choanal Atresia , Canada , Child , Choanal Atresia/diagnosis , Choanal Atresia/epidemiology , Female , Humans , Infant , Infant, Newborn , Male , Retrospective Studies , Tertiary Care CentersABSTRACT
BACKGROUND: To evaluate the clinical management of choanal atresia (CA) in tertiary centers across Canada. METHODS: Multi-centre case series involving six tertiary care pediatric hospitals across Canada. Retrospective chart review of patients born between 1980 and 2010 diagnosed with choanal atresia to a participating center. RESULTS: The health charts of 215 patients (59.6% female) with choanal atresia (CA) were reviewed. Mean age of initial surgical repair was 0.8 months for bilateral CA, and 48.6 months for unilateral CA. Approaches of surgical repair consisted of endoscopic transnasal (31.7%), non-endoscopic transnasal (42.6%), and transpalatal (25.2%). Stents were used on 70.7% of patients. Forty-nine percent of patients were brought back to the OR for a planned second look; stent removal being the most common reason (86.4%). Surgical success rate of initial surgeries was 54.1%. Surgical technique was not associated with rate of restenosis [χ2 (2) = 1.6, p = .46]. CONCLUSIONS: The present study is the first national multi-institutional study exploring the surgical outcomes of CA over a 30-year period. The surgical repair of CA presents a challenge to otolaryngologists, as the rate of surgical failure is high. The optimal surgical approach, age at surgical repair, use of stents, surgical adjuncts, and need for planned second look warrant further investigation.
Subject(s)
Choanal Atresia , Child , Choanal Atresia/epidemiology , Choanal Atresia/surgery , Endoscopy , Female , Humans , Infant, Newborn , Male , Retrospective Studies , Tertiary Care Centers , Treatment OutcomeABSTRACT
OBJECTIVE: Understanding of maxillary sinus hypoplasia (MSH) and associated sinonasal variants is paramount to the diagnostic and therapeutic success of maxillary sinus and maxillary dental implant surgery. The purpose of this work was to explore the prevalence of MSH, frequency of mucosal thickening, and anatomical variations in the sinonasal complex. METHODS: Retrospective case-control design based on cone beam CT images of dental patients with MSH and matched for age and gender. MSH type and sinonasal variables were recorded. RESULTS: One thousand three hundred seventy cone beam CT scans were analyzed for MSH. MSH prevalence was 6% (n=82), matched with 82 controls= 100 females and 64 males, mean age 37.1±14.1 years. Most MSH were type I, 69.5%, 80.5% unilateral, 65.9% associated with no or mild mucosal thickening. Associated sinonasal anatomical variations were <27% except for deviated/hyperplastic (DH) meatus, 48.8%. Within the MSH group, significant associations were presented between MSH type, mucosal thickening, and DH nasal meatus. MSH group vs controls showed a significant difference in mucosal thickening (OR 5.2, 95% CI 2.0-17.3) and DH meatus (OR 1.6, 95% CI 1.4-2.1). CONCLUSIONS: A hypoplastic maxillary sinus with abnormal or absent uncinate process is associated with advanced mucosal thickening and may present with altered anatomy of the lateral wall of the nasal cavity causing its approximation to the orbital floor. CLINICAL RELEVANCE: Pre-surgical knowledge of altered anatomy in the sinonasal complex is crucial in dental implant or sinus surgery.
Subject(s)
Cone-Beam Computed Tomography , Maxillary Sinus , Adult , Female , Humans , Male , Maxilla , Maxillary Sinus/diagnostic imaging , Middle Aged , Prevalence , Retrospective Studies , Young AdultABSTRACT
OBJECTIVES: Swallowing dysfunction (SwD) is under-reported in otherwise healthy infants and toddlers (OHITs). The identification of parental perceptions of factors that may hinder the diagnosis could help clinicians manage these children in a more expeditious manner. This study investigated the barriers to diagnosing SwD, as reported by the families. DESIGN: Grounded theory study. SETTING: This study was performed in a tertiary care paediatric centre in Canada. PARTICIPANTS: Parents of OHITs were recruited using purposeful sampling. INTERVENTION: We used detailed, semistructured, in-person interviews and the audiotapes and transcriptions were thematically analysed. From the parental insights, we built a framework composed of three themes of barriers. RESULT: Ten parents of OHITs with SwD were interviewed. The children presented with recurrent coughing, choking, cold-like symptoms, recurring/consistent illnesses and feeding difficulties. They were managed with multiple rounds of antibiotics and diagnosed with allergies, asthma or recurrent viral infections before considering SwD. The three emerging themes are false beliefs about SwD among parents and some physicians, parent-related barriers and physician-related barriers. These barriers had severely impacted the parents, impairing work productivity and leading to work-related reprimands and changes in the family dynamics. CONCLUSION: This study suggests that there are several barriers that face the parents of OHITs when seeking a diagnosis of SwD and initiating appropriate management. These barriers likely interact with one another and amplify their effects on the family and the child. A common denominator is a lack of education regarding SwD, its clinical manifestations and the available expertise to manage this condition.
Subject(s)
Deglutition , Parents , Canada , Child , Grounded Theory , Humans , Infant , Perception , Qualitative ResearchABSTRACT
OBJECTIVES/HYPOTHESIS: Create a competency-based assessment tool for pediatric esophagoscopy with foreign body removal. STUDY DESIGN: Blinded modified Delphi consensus process. SETTING: Tertiary care center. METHODS: A list of 25 potential items was sent via the Research Electronic Data Capture database to 66 expert surgeons who perform pediatric esophagoscopy. In the first round, items were rated as "keep" or "remove" and comments were incorporated. In the second round, experts rated the importance of each item on a seven-point Likert scale. Consensus was determined with a goal of 7 to 25 final items. RESULTS: The response rate was 38/64 (59.4%) in the first round and returned questionnaires were 100% complete. Experts wanted to "keep" all items and 172 comments were incorporated. Twenty-four task-specific and 7 previously-validated global rating items were distributed in the second round, and the response rate was 53/64 (82.8%) with questionnaires returned 97.5% complete. Of the task-specific items, 9 reached consensus, 7 were near consensus, and 8 did not achieve consensus. For global rating items that were previously validated, 6 reached consensus and 1 was near consensus. CONCLUSIONS: It is possible to reach consensus about the important steps involved in rigid esophagoscopy with foreign body removal using a modified Delphi consensus technique. These items can now be considered when evaluating trainees during this procedure. This tool may allow trainees to focus on important steps of the procedure and help training programs standardize how trainees are evaluated. LEVEL OF EVIDENCE: 5. Laryngoscope, 131:1168-1174, 2021.
Subject(s)
Clinical Competence/standards , Consensus , Esophagoscopy/education , Internship and Residency/standards , Surgeons/standards , Child , Delphi Technique , Esophagoscopes , Esophagoscopy/instrumentation , Esophagus/diagnostic imaging , Esophagus/surgery , Foreign Bodies/diagnosis , Foreign Bodies/surgery , Humans , Surgeons/education , Surgeons/statistics & numerical data , Surveys and Questionnaires/statistics & numerical dataABSTRACT
OBJECTIVES: There is limited epidemiological information on swallowing dysfunction (SwD) in otherwise healthy infants and toddlers (OHITs). Cost, invasiveness, expertise, and resources constrain the repeatability and utility of instrumental diagnostic tests. A parent-reported outcomes (PRO) tool has the potential to mitigate these disadvantages. Hence, we set out to develop and validate a novel PRO tool to assess SwD in OHITs. STUDY DESIGN: A mixed-method study. SETTING: Tertiary pediatric center. METHODS: We recruited parents of OHITs with SwD and excluded those with a confounding diagnosis (syndromes or neurological impairment). Interviews were conducted and thematically analyzed to extract the relevant domains and items. A similar analytical method was performed on the reports from a systematic review and literature search. Four verification sessions of parents and experts were conducted to maintain rigor. A panel of experts assessed and established the content validity of the items using a modified Delphi technique. RESULTS: We achieved information saturation after interviewing 10 parents and generated 7 domains with 72 items. Over the course of 3 rounds of modified Delphi content validation, the domains were reduced to 3 (swallowing, breathing, and illness) containing 21 items; a content validity index of 82.1% was achieved. CONCLUSION: We validated the content of a new PRO instrument to assess SwD in OHITs. The instrument is composed of 3 primary domains representing 21 items. This tool has the potential to screen for swallowing dysfunction and can assess management outcomes specifically for this population at a community level.
Subject(s)
Deglutition Disorders/diagnosis , Parents/psychology , Patient Reported Outcome Measures , Adult , Age Factors , Child, Preschool , Cohort Studies , Deglutition Disorders/etiology , Deglutition Disorders/therapy , Delphi Technique , Female , Humans , Infant , Male , Reproducibility of Results , Symptom AssessmentABSTRACT
OBJECTIVES: Laryngomalacia (LM) is commonly diagnosed in infants and children with upper aerodigestive symptoms. In the literature, the focus has been on the respiratory impairment, with fewer studies addressing swallowing dysfunction (SwD). The objective of this study is to systematically review the literature for evidence on the prevalence of SwD in children diagnosed with LM. METHODS: A search was conducted on the following databases: OVID Medline, Ovid EMBASE, EBSCO CINAHL, PROSPERO, and Cochrane Library. We included all the studies that reported on children with LM and documented objective swallowing assessment using fiberoptic endoscopic evaluation of swallowing (FEES) or videofluoroscopic swallowing study (VFSS). Two authors independently screened all the studies, assessed the level of evidence in the included studies, and extracted data. Risk of bias assessment and pooled data analysis were performed. RESULTS: The search yielded 512 abstracts. Four studies met the selection criteria representing 425 children. Three studies were retrospective uncontrolled case series and one was a prospective cohort study. In all studies but one, an instrumental assessment of swallowing was selectively performed in patients with clinical indicators of SwD. The pooled estimate (range) of prevalence of SwD was 49% (13.9-90.6%). CONCLUSION: The literature suggests a high prevalence of SwD in children with LM, however the level of evidence is low and generalizability is poor. The wide range of prevalence figures suggests a significant variability in the threshold and indications to assess swallowing in children with LM.
Subject(s)
Deglutition , Laryngomalacia , Child , Humans , Infant , Laryngomalacia/complications , Laryngomalacia/diagnosis , Laryngomalacia/epidemiology , Prevalence , Prospective Studies , Retrospective StudiesABSTRACT
OBJECTIVES: To describe indications for injection augmentation (IA), endoscopic repair (ER) and conservative methods for the management of type 1 laryngeal cleft (LC1) and propose a management algorithm. We also aimed to compare success of IA and ER and determine independent predictors of treatment failure. METHODS: Retrospective study of patients diagnosed with LC1 at a Pediatric Otolaryngology referral centre between 2004 and 2016. All had pre-operative instrumental swallowing evaluation (VFSS/FEES), and were managed with a combination of conservative measures, IA and/or ER. We collected demographics, symptoms, comorbidities, VFSS/FEES results, and operative details. The primary outcome was symptom resolution by parental report. The secondary outcome was predictors of treatment failure. RESULTS: 88 patients were included in the analysis, with mean age 26 ± 25 months. Most presented with choking events (68%) or recurrent pneumonias (48%). In total, there were 55 IA performed and 45 ER. Of the patients who received IA, 19 required subsequent ER. 95% had symptom improvement, 67% had complete resolution. IA had a 56% long-term success rate, whereas that for ER was 85%. Tube feeding at initial evaluation was an independent predictor of treatment failure (HR 11.33 [1.51-84.97], p = 0.018). CONCLUSIONS: LC1 can be effectively managed with a combination of IA and ER with favorable results. Failure to respond to IA does not preclude ER, and both have their role in management. Patients who are tube fed have a higher probability of treatment failure. We propose a management algorithm that includes reasoning for conservative approaches, and reduces exposure to general anesthesia.
Subject(s)
Congenital Abnormalities/surgery , Deglutition Disorders/surgery , Laryngoplasty , Larynx/abnormalities , Airway Obstruction/etiology , Child , Child, Preschool , Comorbidity , Deglutition Disorders/etiology , Endoscopy , Female , Humans , Infant , Injections, Intralesional , Larynx/surgery , Male , Pneumonia/etiology , Retrospective StudiesABSTRACT
OBJECTIVES: To determine the agreement of paediatric otolaryngologists on classifying laryngomalacia (LM). DESIGN: Intra- and interobserver agreement study of two classification systems. SETTING: Three tertiary referral paediatric centres. PARTICIPANTS: Three paediatric otolaryngologists, who were blinded to any clinical details, interpreted the videos of children diagnosed with LM using the Holinger and Olney classifications independently. They rated the videos twice with a washout period of at least 2 weeks. THE MAIN OUTCOME MEASURES: Inter- and intra-observer agreement measured by overall Fleiss kappa and unweighted Cohen's kappa coefficients. The secondary outcome measures were inter- and intra-observer agreement on the individual anatomical subunits of the supraglottis affected by LM, characterised by the subcategories of the classifications. RESULTS: Video records of infants and children <18 years who had an endoscopic diagnosis of LM from 2012 to 2017 were retrospectively chosen for inclusion (n = 73). The overall Fleiss kappa coefficient was 0.25 (95% CI 0.18-0.32) amongst the raters using the Holinger classification and 0.31 (95% CI 0.21-0.42) for the Olney classification. Intra-observer agreement using the Holinger classification was 0.30 (95% CI 0.18-0.42), 0.62 (95% CI 0.23-0.85) and 0.84 (95% CI 0.75-0.94], whilst the Olney classification yielded values of 0.41 (95% CI 0.26-0.56), 0.51 (95% CI 0.29-0.63) and 0.63 (95% CI 0.48-0.78). CONCLUSIONS: The agreement on types of LM between expert observers is modest using the Holinger and Olney classifications. This has significant implications for accurately diagnosing LM and exposes potential obstacles against credible pooling of data and extrapolation of information.
Subject(s)
Laryngomalacia/classification , Adolescent , Child , Child, Preschool , Consensus , Female , Humans , Infant , Infant, Newborn , Male , Video RecordingABSTRACT
OBJECTIVES/HYPOTHESIS: Create a competency-based assessment tool for pediatric tracheotomy. STUDY DESIGN: Blinded, modified, Delphi consensus process. METHODS: Using the REDCap database, a list of 31 potential items was circulated to 65 expert surgeons who perform pediatric tracheotomy. In the first round, items were rated as "keep" or "remove," and comments were incorporated. In the second round, experts were asked to rate the importance of each item on a seven-point Likert scale. Consensus criteria were determined a priori with a goal of 7 to 25 final items. RESULTS: The first round achieved a response rate of 39/65 (60.0%), and returned questionnaires were 99.5% complete. All items were rated as "keep," and 137 comments were incorporated. In the second round, 30 task-specific and seven previously validated global rating items were distributed, and the response rate was 44/65 (67.7%), with returned questionnaires being 99.3% complete. Of the Task-Specific Items, 13 reached consensus, 10 were near consensus, and 7 did not achieve consensus. For the 7 previously validated global rating items, 5 reached consensus and two were near consensus. CONCLUSIONS: It is feasible to reach consensus on the important steps involved in pediatric tracheotomy using a modified Delphi consensus process. These items can now be considered to create a competency-based assessment tool for pediatric tracheotomy. Such a tool will hopefully allow trainees to focus on the important aspects of this procedure and help teaching programs standardize how they evaluate trainees during this procedure. LEVEL OF EVIDENCE: 5 Laryngoscope, 130:2700-2707, 2020.
Subject(s)
Clinical Competence/standards , Pediatrics/standards , Surgeons/standards , Tracheotomy/standards , Child , Consensus , Delphi Technique , Humans , Pediatrics/education , Pediatrics/methods , Single-Blind Method , Surgeons/education , Tracheotomy/educationABSTRACT
BACKGROUND: Inferior turbinoplasty (IT) and adenoidectomy (Ad) are frequently resorted to in children with chronic rhinitis (CR) refractory to medical therapy. The aim of this study is to document the long-term improvement in quality of life (QOL) in children with CR following endoscopic IT with or without Ad. METHODS: A retrospective case series study was conducted. We searched a prospectively kept surgical database for children ≤18 years old who had CR who underwent endoscopic IT with or without Ad between 2009 and 2016 at a tertiary care children's center. Patients with sinonasal pathologies other than CR, had craniofacial syndromes or dysmorphism and had other sinonasal procedures or trauma were excluded. Collected data included demographics, secondary diagnoses, duration of follow-up, and complications of procedures. The Glasgow Children's Benefit Inventory (GCBI) was administered by phone to assess QOL improvement. RESULTS: One hundred sixty-five eligible subjects were identified. Eighty-nine subjects met the inclusion criteria. Data was collected for the 60 subjects that were reached. Forty-two patients had IT only while 18 had IT and Ad. The mean age was 10.7 ± 2.7 years, with 31 males and 29 females. The median duration of follow-up (25th, 75th percentile) was 38.1 months (24.6, 55.8). The median GCBI score (25th, 75th percentile) was 22.9 (6.3, 39.6) revealing an overall positive benefit in all domains. There was only one complication. CONCLUSIONS: This study validates prior findings regarding improvement of QOL and safety of IT with or without Ad for children with CR and indicates it is maintained in the long term.