Factors associated with laryngeal injury after intubation in children: a systematic review.

PURPOSE: The purpose of this study is to evaluate all potential factors associated with laryngeal injury after endotracheal intubation in the pediatric population. METHODS: A systematic literature search was conducted in Medline, Embase, Cochrane, web of science and Google scholar up to 20th of March 2023. We included all unique articles focusing on factors possibly associated with intubation-injury in pediatric patients. Two independent reviewers determined which articles were relevant by coming to a consensus, quality of evidence was rated using GRADE criteria. All articles were critically appraised according to the PRISMA guidelines. The articles were categorized in four outcome measures: post-extubation stridor, post-extubation upper airway obstruction (UAO) necessitating treatment, laryngeal injury found at laryngoscopy and a diagnosed laryngotracheal stenosis (LTS). RESULTS: A total of 24 articles with a total of 15.520 patients were included. The incidence of post-extubation stridor varied between 1.0 and 30.3%, of post-extubation UAO necessitating treatment between 1.2 and 39.6%, of laryngeal injury found at laryngoscopy between 34.9 to 97.0% and of a diagnosed LTS between 0 and 11.1%. Although the literature is limited and quality of evidence very low, the level of sedation and gastro-esophageal reflux are the only confirmed associated factors with post-extubation laryngeal injury. The relation with age, weight, gender, duration of intubation, multiple intubations, traumatic intubation, tube size, absence of air leak and infection remain unresolved. The remaining factors are not associated with intubation injury. CONCLUSION: We clarify the role of the potential factors associated with laryngeal injury after endotracheal intubation in the pediatric population.

Hyoid bone morphology in patients with isolated robin sequence - A case-control study utilizing 3D morphable models.

Background: Abnormalities of the hyoid bone are associated with impairment of oropharyngeal functions including feeding, swallowing, and breathing. Few studies have characterized anatomic abnormalities of the hyoid in patients with Robin sequence (RS), e.g. a less mineralized and voluminous hyoid. The purpose of this study was to compare normal hyoid bone morphology and hyoid bone morphology in children with isolated RS. Methods: Three-dimensional (3D) reconstructions of the hyoid bone were obtained from CT-imaging of children with RS and unaffected controls. A 3D morphable model was constructed using Principal Component Analysis (PCA). Partial least squares - Discriminant Analysis (PLS-DA) and multivariate analysis of variance (MANOVA) were used to characterize and compare hyoid shape differences between patients with RS and an age-matched control group. Results: The study included 23 subjects with RS (mean age 9.8 ± 10.3 months) and 46 age-matched control samples. A less voluminous hyoid was observed for the RS group with a larger lateral divergence of the greater horns compared to controls (MANOVA, p-value<0.001). The first shape variable from the PLS-DA model showed a significant correlation for the observed variance between the two groups (Spearman R = -0.56, p-value<0.001). The control samples and 151 CT-scans of subjects up to age 4 years were used to create a 3D morphable model of normal hyoid shape variation (n = 197, mean age 22.1 ± 13.1 months). For the normal 3D morphable model, a high degree of allometric shape variation was observed along the first principal component. Conclusions: The 3D morphable models provide a comprehensive and quantitative description of variation in normal hyoid bone morphology, and allow detection of distinct differences between patients with isolated RS and controls.

Feeding and swallowing outcomes following mandibular distraction osteogenesis: an analysis of 22 non-isolated paediatric cases.

Patients with mandibular hypoplasia and upper airway obstruction are at an increased risk of feeding and swallowing difficulties. Little has been described regarding these outcomes following mandibular distraction. The aim of this study was to evaluate the effect of mandibular distraction on feeding and swallowing function. A retrospective study was performed on 22 patients with non-isolated mandibular hypoplasia and severe upper airway obstruction treated with mandibular distraction. Median age at first mandibular distraction was 3.1 years (interquartile range 2.3-6.0 years) and the median follow-up time was 3.5 years (interquartile range 2.0-9.4 years). Prior to mandibular distraction, feeding difficulties were present in 18 patients. Swallowing difficulties were present in 20 patients, all of whom had problems in the oral phase of swallowing, while 11 patients had additional problems in the pharyngeal phase. Following mandibular distraction, at the time of follow-up, feeding difficulties persisted in 13 patients. Swallowing difficulties in the oral phase remained present in all 20 patients, while pharyngeal phase problems persisted in seven patients. In conclusion, feeding and swallowing difficulties are highly prevalent in non-isolated patients and often persist following mandibular distraction. Moreover, these can be the reason that decannulation cannot be accomplished. Hence, awareness and close follow-up by a specialized speech therapist is of paramount importance.

Indications and clinical outcome in pediatric tracheostomy: Lessons learned.

OBJECTIVE: Indications for tracheostomy have changed over the last decades and clinical outcome varies depending on the indication for tracheostomy. By gaining more insight in the characteristics and outcome of the tracheostomized pediatric population, clinical care can be improved and a better individual prognosis can be given. Therefore, we studied the outcome of our pediatric tracheostomy population in relation to the primary indication over the last 16 years. METHODS: We retrospectively included children younger than 18 years of age with a tracheostomy tube in the Erasmus Medical Center, Sophia children's hospital. The primary indication for tracheostomy, gender, age at tracheostomy, age at decannulation, comorbidity, mortality, closure of a persisting tracheocutaneous fistula after decannulation, surgery prior to decannulation and the use of polysomnography were recorded and analyzed. RESULTS: Our research group consisted of 225 children. Reasons for a tracheostomy were first divided in two major diagnostic groups: 1) airway obstruction group (subgroups: laryngotracheal obstruction and craniofacial anomalies) and 2) pulmonary support group (subgroups: cardio-pulmonary diseases and neurological diseases). Children in the airway obstruction group were younger when receiving a tracheostomy (3.0 months vs. 31.0 months, p < 0.05), they were tracheostomy dependent for a longer time (median 21.5 months vs. 2.0 months, p < 0.05) and they required surgery more often (74.5% vs. 8.3%, p < 0.05) than the children in the pulmonary support group. The decannulation rate of children with a laryngotracheal obstruction is high (74.8%), but low in all other subgroups (craniofacial anomalies; 38.5%, cardio-pulmonary diseases; 34.6% and neurological diseases; 52.9%). Significantly more children (36.7%) died in the pulmonary support group due to underlying comorbidity, mainly in the cardio-pulmonary diseases subgroup. Surgery for a persisting tracheocutaneous fistula was performed in 34 (37.8%) children, with a significant relationship between the duration of the tracheostomy and the persistence of a tracheocutaneous fistula. No cannula related death occurred during this study period. CONCLUSION: Main indications for a tracheostomy were airway obstruction and pulmonary support. Children in the airway obstruction group were younger when receiving a tracheostomy and they were tracheostomy dependent for a longer period. Within the airway obstruction group, the decannulation rate for children with laryngotracheal stenosis was high, but low for children with craniofacial anomalies. In the pulmonary support group, the decannulation rate was low and the mortality rate was high. Surgery for a persisting tracheocutaneous fistula was frequently needed.

Functional outcomes in patients with facial dysostosis and severe upper airway obstruction.

An increased risk of upper airway obstruction (UAO) is seen in up to 95% of patients with facial dysostosis. Secondary to respiratory problems are feeding difficulties and increased nutritional requirements. Little has been described regarding these outcomes in this patient population. Hence, a retrospective cohort study was performed to gather data on functional outcomes. Eighteen patients with facial dysostosis and severe UAO were included. The median follow-up time was 3.42 years. A tracheostomy tube was placed in 13 patients, of whom 10 subsequently underwent mandibular distraction. Three of the five patients without a tracheostomy underwent mandibular distraction as the primary surgical treatment; the remaining two patients were treated conservatively with oxygen supplementation. At presentation, 13 patients had feeding difficulties. Overall malnutrition was present in 16 patients during follow-up. At the end of follow-up, severe UAO was present in 12 patients, feeding difficulties in seven patients, and malnutrition in four patients, while two patients died. In conclusion, patients with facial dysostosis have a high prevalence of severe UAO, feeding difficulties, and malnutrition. Importantly, mandibular distraction has limited success in treating severe UAO in these patients. Close follow-up by a specialized craniofacial team is of paramount importance to manage the long-term consequences.

Post-extubation stridor after prolonged intubation in the pediatric intensive care unit (PICU): a prospective observational cohort study.

PURPOSE: Prolonged endotracheal intubation may lead to laryngeal damage, with stridor being the most relevant clinical symptom. Our objective was to determine the incidence of post-extubation stridor and their clinical consequences in children within a tertiary referral center and to identify contributing factors. METHODS: 150 children, aged 0-16 years, intubated for more than 24 h were prospectively enrolled until discharge of the hospital. Potential relevant factors, thought to mediate the risk of laryngeal damage, were recorded and analyzed. RESULTS: The median duration of intubation was 4 days, ranging from 1 to 31 days. Stridor following extubation occurred in 28 patients (18.7%); 3 of them required reintubation due to respiratory distress and in 1 child stridor persisted for which a surgical intervention was necessary. In multivariate analyses, we found the following independent predictors of stridor: intubation on the scene, the use of cuffed tubes and lower age. CONCLUSION: Despite a high incidence for post-extubation stridor, only few children need reintubation or surgical intervention as a result of post-extubation lesions. Intubation on the scene, the use of cuffed tubes and young age are associated with a significant increased risk of post-extubation stridor. Awareness of these factors gives the possibility to anticipate on the situation and to minimize laryngeal injury and its possible future consequences.

Long-term quality of life in children after open airway surgery for laryngotracheal stenosis.

OBJECTIVES: The purpose of this study is to evaluate the long-term health related quality of life (HRQoL) in a cohort of children surgically treated for laryngotracheal stenosis (LTS). STUDY DESIGN: Prospective cohort study. METHODS: Parents of children between 4 and 18 years at follow-up completed the Child Health Questionnaire Parent Form (CHQ-PF50). Children between 11 and 18 years at follow-up completed the Child Health Questionnaire Child Form (CHQ-CF87). Biographical and pre-operative data were extracted from the hospital records. Post-operative measurements consisted of the Bruce treadmill test and pulmonary function testing (PFT). RESULTS: Fifty-four parents completed the CHQ-PF50; twenty-one children completed the CHQ-CF87. The CHQ-PF50 was significantly worse than the norm population on the subscales physical functioning, role functioning: emotional/behavior, general health perceptions, family activities, parental impact: emotional, and time. CHQ-CF87 was significantly worse than the norm population on physical functioning and better on mental health. After multivariate analysis, presence of co-morbidities and glottic stenosis are the most important pre-operative factors for worse scores on general health. As post-operative measurements, the Bruce treadmill test and peak expiratory flow (PEF) correlate well with HRQoL physical subscales. CONCLUSIONS: At long-term follow-up after treatment for LTS, deficits in HRQoL may still exist. Presence of co-morbidities and glottic stenosis are important negative factors for long-term HRQoL. The Bruce treadmill test and peak expiratory flow on pulmonary function testing correlate well with physical subscales on HRQoL. A long-term multidisciplinary follow-up with assessment of HRQoL is advised in patients treated for LTS. LEVEL OF EVIDENCE: 2B, individual prospective cohort study.

Awake Flexible Fiberoptic Laryngoscopy to diagnose glossoptosis in Robin Sequence patients.

BACKGROUND: Robin Sequence (RS) is usually defined as the combination of micrognathia, glossoptosis and upper airway obstruction. No objective criteria to diagnose RS exist. To compare management strategy results, a single RS definition using objective criteria is needed. The most frequently used primary diagnostic tool for glossoptosis is awake Flexible Fiberoptic Laryngoscopy (aFFL). OBJECTIVES: To determine the reliability of the aFFL videos as an independent diagnostic tool itself, rather than on the complete evaluation of a patient. DESIGN, SETTING, PARTICIPANTS: All RS individuals from an existing cohort with an available aFFL video were included retrospectively. Thirty age-matched patients without pathologic findings on aFFL were used as controls. aFFL videos were scored by six otolaryngologists as: a. Marked glossoptosis, b. Mild glossoptosis, c. Severity unknown, d. No glossoptosis, e. Insufficient video quality. Videos were anonymised and rated twice, in altered sequences, after a washout period of minimally 2 weeks. MAIN OUTCOME MEASURES: Inter-rater and intrarater agreement. RESULTS: Twenty-six videos of 16 RS patients and 30 videos of controls were included. Inter-rater agreement was fair in the whole group (κ: 0.320) and RS group (κ: 0.226), and fair to moderate in determining presence of glossoptosis (total group κ: 0.430; RS κ: 0.302; controls κ: 0.212). The intrarater agreement for the presence of glossoptosis in RS was moderate (κ: 0.541). CONCLUSIONS: aFFL offers fair to moderate inter-rater agreement, with moderate intrarater agreement, in evaluating glossoptosis in RS. Using aFFL as the single tool in choosing management strategies in RS seems insufficient. There is need for a more reliable, patient friendly diagnostic tool or an internationally accepted aFFL scoring system, to diagnose glossoptosis in RS.

Atypical presentation of a newborn with Apert syndrome.

INTRODUCTION: Apert syndrome is a rare syndrome characterized by a consistent phenotype including bilateral coronal suture synostosis with an enlarged anterior fontanel, midface hypoplasia, and complex symmetric syndactyly of hands and feet. CASE REPORT: We present a boy with Apert syndrome caused by the pathogenic c.755C > G p.Ser252Trp mutation in the FGFR2 gene with atypical characteristics, including premature fusion of the metopic suture with a small anterior fontanel, hypotelorism, and a massive posterior fontanel. Directly after birth, he showed papilledema, epilepsy, and central apneas. CONCLUSION: We present a newborn with Apert syndrome with atypical craniofacial presentation.

Characteristics and surgical outcome of 98 infants and children surgically treated for a laryngotracheal stenosis after endotracheal intubation: excellent outcome for higher grades of stenosis after SS-LTR.

INTRODUCTION: To describe the characteristics and surgical outcome of 98 infants and children treated for an acquired laryngeal stenosis after intubation for respiratory support. MATERIAL AND METHODS: We retrospectively reviewed our data from the last 18 years (1994-2013) concerning infants and children with an acquired laryngotracheal stenosis who were treated in our hospital with a laryngotracheal reconstruction or a cricotracheal resection. Outcome was defined by decannulation ratio. RESULTS: Of the 98 infants and children who were studied, 54% were preterm, 18% neonates, 13% infants and 14% children. Ninety-one SS-LTR's, two DS-LTR's and five CTR's were performed as primary surgery; three revision operations were performed (DS-LTR). Seventy-seven children had a tracheostomy prior to surgery; decannulation ratio was 93% after primary surgery and 95% after inclusion of revision surgery. For SS-LTR, the decannulation ratio was 93%, including grade III stenosis with comorbidities. Male sex and glottic involvement of the stenosis are correlated to failure of decannulation. Intubation in the term neonatal period is correlated to complicated post-operative course after SS-LTR. CONCLUSIONS: Excellent results of surgery for acquired laryngotracheal stenosis can be obtained with a high decannulation rate. Even for higher grades of stenosis with comorbidities and glottic involvement, an SS-LTR is an effective surgical treatment for acquired laryngeal stenosis.

Impact of the consideration of serrated polyps to the interval of colonoscopic surveillance in the NHS Bowel Cancer Screening Programme.

AIM: Most international post polypectomy surveillance guidelines do not recommend surveillance for serrated polyps. In the present study the additional impact of serrated polyps on surveillance intervals from international adenoma surveillance guidelines was investigated. METHOD: Endoscopic and pathology records were audited of participants in the NHS Bowel Cancer Screening Programme (guaiac faecal occult blood test, gFOBT) in 2011. Surveillance intervals were calculated for current guidelines and also for serrated polyps based on previously described aggressive and conservative strategies. RESULTS: In total, 389 patients were included of whom 141 (36.2%) were high risk (advanced adenoma: adenoma ≥ 10 mm, villous elements, high grade dysplasia, or adenoma ≥ 3 in number) needing surveillance at ≤ 3 years. Thirty-three (8.5%) had significant serrated polyps, of whom 18 (4.6% of the total) had significant serrated lesions and simultaneous advanced adenoma or ≥ 3 adenomas. Adopting an aggressive surveillance strategy, the mean overall absolute additional proportion of all such patients in the surveillance group at 3 years or less was 4.0% (3.9% - 4.1%; 4.2% women; 3.8% men). These proportions varied according to endoscopist from 2.3% to 4.7%. For more conservative strategies the increase was only 1%. CONCLUSION: The impact of including serrated polyps in current guidelines would result in a small increase in surveillance intervals for FOBT based bowel cancer screening. About half of those who might need surveillance for serrated polyps would already receive surveillance for being in a high risk adenoma group.