Paediatric anterior larynx: presentations, associations and a review of the literature.

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.

Prevalence of maxillary sinus hypoplasia and association with variations in the sinonasal complex: a cone beam CT study.

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.

Validity of laryngomalacia classification systems: A multi-institutional agreement study.

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.

Postnatal Diagnosis of a Baby With Multiple Rare Congenital Anomalies Including Syngnathia, Brain Dysmorphism, and Skin Pigmentation.

Syngnathia is a rare congenital disorder of jaw fusion with a paucity of literature from developed countries. We present a case of an infant noted to have multiple anomalies at birth including syngnathia, microcephaly with a variant of brain abnormality between holoprosencephaly and syntelencephaly, optic nerve hypoplasia, ear canal anomalies, hemi-vertebrae, and suspected hypomelanosis of Ito. To our knowledge, this patient with syngnathia and multiple anomalies is the first to be reported, but whether they are a coincidence, a pathogenetic association, or a new syndrome remains unknown. This case is discussed with a brief review of the literature.

Agreement between cone-beam computed tomography and nasoendoscopy evaluations of adenoid hypertrophy.

INTRODUCTION: The goals of this study were to evaluate (1) the reliability and accuracy of cone-beam computed tomography (CBCT) for assessing adenoid size compared with nasoendoscopy and (2) the influence of clinical experience on CBCT diagnosis. METHODS: Adenoid size was graded on a 4-point scale for CBCT and nasoendoscopy by a pediatric otolaryngologist. Reliability was assessed with intraobserver and interobserver agreement. Accuracy was assessed with agreement between CBCT and nasoendoscopy, plus sensitivity and specificity analyses. The CBCT assessments were completed by a team of 4 evaluators: an oral and maxillofacial radiologist, an airway orthodontist who participates in the multidisciplinary team, an academic orthodontist whose primary research is in 3-dimensional imaging, and a highly experienced private practice orthodontist comfortable with CBCT imaging. Each evaluator was specifically chosen to represent a unique set of clinical and radiographic experiences. All evaluators were blinded to the subject's identity and clinical history, and they evaluated the images in a unique random order and evaluated each image 3 times separated by a minimum of 7 days. The same computer hardware and software were used. RESULTS: Thirty-nine consecutively assessed, nonsyndromic subjects (ages, 11.5 ± 2.8 years) were evaluated. The CBCT demonstrated excellent sensitivity (88%) and specificity (93%), strong accuracy (ICC, 0.80; 95% CI, ± 0.15), and good reliability, both within observers (ICC, 0.85; 95% CI, ± 0.08) and between observers (ICC, 0.84; 95% CI, ± 0.08). The clinical experience of the CBCT evaluator did not have a statistically significant effect. CONCLUSIONS: CBCT is a reliable and accurate tool for identifying adenoid hypertrophy.

Congenital Tracheal Stenosis Patients Undergoing Modified Slide Tracheoplasty: Single-Centre Technique and Long-Term Morbidity and Mortality.

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.

International pediatric otolaryngology group (IPOG) consensus on approach to aspiration.

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.

Craniofacial morphology in pediatric patients with persistent obstructive sleep apnea with or without positive airway pressure therapy: a cross-sectional cephalometric comparison with controls.

INTRODUCTION: Compression on the midface with nasal mask-delivered positive airway pressure (PAP) therapy in growing patients might contribute to midface retrusion. The objective of this study was to investigate the association between long-term PAP use and craniofacial morphologic pattern in children with persistent obstructive sleep apnea. METHODS: Images generated with cone-beam volumetric imaging were used to complete lateral cephalometric analyses of anteroposterior projection of the midface region. The study group included 12 subjects (10 boys, 2 girls; mean age, 9.0 years) who used PAP therapy for at least 6 months and at least 6 hours per night. Measurements from this group were compared with those of a control group of 11 subjects (5 boys, 6 girls; mean age, 9.6 years) with obstructive sleep apnea who did not have PAP. Measurements were taken at 1 time point. RESULTS: No significant differences were identified between the groups for any cephalometric variable. Multivariate linear regression analysis also did not identify a significant association between the number of months of PAP therapy and the cephalometric variables. Cephalometric data for both groups were pooled for comparison with appropriate published normative values for age and sex. Anterior cranial base length, overall anteroposterior length of the maxillary base, and mandibular body length were significantly shorter than normal in the subjects compared with published normative values. CONCLUSIONS: No association was demonstrated between midface projection and PAP use in growing patients. When compared with normative data for anterior cranial base, children with obstructive sleep apnea had shorter maxillary and mandibular lengths.

Patterns of Pharyngeal Obstruction and Collapse in Obese and Nonobese Children on Drug-Induced Sleep Endoscopy.

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.

International Pediatric Otolaryngology Group (IPOG) consensus on scoring of pediatric Drug Induced Sleep Endoscopy (DISE).

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.

Branchiooculofacial syndrome and bilateral ectopic thymus: report of a family.

Branchiooculofacial syndrome (BOFS) is a rare autosomal-dominant condition characterized by branchial cleft sinus and ophthalmologic and craniofacial abnormalities that can range from mild to severe forms. Ectopic thymus, an uncommon condition with just over 100 cases reported in the literature, is seen in BOFS. We report a family with BOFS affecting the mother and two daughters, showing the dominant inheritance and variable phenotypical expression of BOFS.

Eosinophilic esophagitis in children needing emergency endoscopy for foreign body and food bolus impaction.

OBJECTIVE: The objective of this study was to examine the prevalence of eosinophilic esophagitis (EoE) in children presenting with esophageal foreign body (FB)/food bolus impaction. METHODS: A retrospective chart review for all children who underwent endoscopic esophageal FB extraction at the Stollery Children's Hospital between January 2005 and December 2008 was performed. The prevalence of EoE and characteristics of children who had EoE were documented. RESULTS: A total of 140 children (mean age, 4.98 [SD, 4] years; range, 0.3-16.9 years; 81 boys) had esophageal FB/food impaction and needed endoscopic removal over the study period. Eleven children (mean age, 9.38 [SD, 5.35] years; range, 1.9-16.2 years; 8 boys) were diagnosed with EoE (8%). Seven (39%) of 18 children with food impaction were diagnosed with EoE. Four (3%) of 122 children with esophageal foreign bodies other than food had EoE. CONCLUSIONS: Considerable number of children with esophageal FB/food bolus impaction may have EoE. Routine sampling of esophageal mucosa at different levels during esophageal FB extraction may be considered. Well-designed prospective studies with multiple-level esophageal biopsies taken from all children with FB/food impaction are needed to confirm our results.

A novel sleep oximetry scoring tool for pediatric laryngomalacia.

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.

Properties of the Nasal Cartilage, from Development to Adulthood: A Scoping Review.

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.

Tracheal Reconstruction for Congenital Tracheal Stenosis: A 950-Gram Neonate.

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.

Esophageal foreign body extraction in children: flexible versus rigid endoscopy.

BACKGROUND: Esophageal foreign body (FB) impaction is a common emergency in children. The goal of this study was to compare rigid versus flexible endoscopy in esophageal FB extraction in children. METHODS: In a retrospective cohort study with consecutive data, children with esophageal FB impaction who were admitted between January 2005 and December 2008 to the Stollery Children's Hospital, Edmonton, Canada, were included. Nature of the procedure for FB removal (flexible vs. rigid endoscopy), duration of the procedure, complications, and associated pathology were documented. RESULTS: A total of 140 children were included (81 boys; mean age, 59.8 ± 48.6 (range, 4-203) months). More than half (54%) of patients were aged 3 years or younger. Coins were the most common foreign body (77.9%). Flexible endoscopy was used in 89 patients, rigid in 49, and both in 2 patients. The mean duration of the endoscopic procedure was 10.50 ± 12.2 minutes for FE (95% confidence interval (CI), 7.94-13.08) and 16.49 ± 21.1 minutes for RE (95% CI, 13.75-22.45; p = 0.04). Biopsies were taken in 19% of patients undergoing FE and in 6% of RE (p = 0.04). CONCLUSIONS: Both rigid and flexible endoscopy techniques appear to be equally safe and effective in esophageal foreign body extraction. However, performing flexible endoscopy for esophageal foreign body takes a substantial shorter duration compared with rigid endoscopy. Flexible endoscopy would probably allow a better and more thorough examination and, hence, biopsying esophageal mucosa compared with rigid endoscopy.

What are parents' perceptions related to barriers in diagnosing swallowing dysfunction in children? A grounded theory approach.

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.

Systematic review of validated parent-reported questionnaires assessing swallowing dysfunction in otherwise healthy infants and toddlers.

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.