Foreign body aspiration in children: our pediatric tertiary care experience.

PURPOSE: Inhalation of a foreign body is a real emergency in pediatric age and requires prompt diagnosis and treatment to reduce mortality. The objective of this study is to analyze clinical and radiological details, types, and localization of foreign bodies in patients conducted or to our hospital with suspected inhalation. METHODS: We conducted a retrospective analysis of all cases of foreign body inhalation admitted to our Pediatric Emergency Room between January 2009 and June 2022. RESULTS: 171 patients were included in the study. In 83 patients, the FB was detected. The mean age of presentation was 2.3 years (SD: ± 2). Cough (73%) and unilateral reduced breath sound (51%) were the most common clinical symptom and clinical sign. The most frequent localization was the right main bronchus (43%). The foreign bodies retrieved were vegetable (83%), of which peanut was the most common. Chest radiographs were normal in 25%. The mean duration of hospitalization was 5 days (± 2.9). Complications such as pneumothorax were seen in two cases. CONCLUSIONS: Foreign body inhalation represents a true pediatric emergency and still a challenge in clinical practice. The best way to manage it is an early diagnosis and removal by fully trained staff.

Vocal Cord Paralysis and Feeding Difficulties as Early Diagnostic Clues of Congenital Myasthenic Syndrome with Neonatal Onset: A Case Report and Review of Literature.

Herein, we present a newborn female with congenital vocal cord paralysis who required a tracheostomy in the neonatal period. She also presented with feeding difficulties. She was later diagnosed with a clinical picture of congenital myasthenia, associated with three variants of the MUSK gene: the 27-month follow-up was described. In particular, the c.565C>T variant is novel and has never been described in the literature; it causes the insertion of a premature stop codon (p.Arg189Ter) likely leading to a consequent formation of a truncated nonfunctioning protein. We also systematically collected and summarized information on patients' characteristics of previous cases of congenital myasthenia with neonatal onset reported in the literature to date, and we compared them to our case. The literature reported 155 neonatal cases before our case, from 1980 to March 2022. Of 156 neonates with CMS, nine (5.8%) had vocal cord paralysis, whereas 111 (71.2%) had feeding difficulties. Ocular features were evident in 99 infants (63.5%), whereas facial-bulbar symptoms were found in 115 infants (73.7%). In one hundred sixteen infants (74.4%), limbs were involved. Respiratory problems were displayed by 97 infants (62.2%). The combination of congenital stridor, particularly in the presence of an apparently idiopathic bilateral vocal cord paralysis, and poor coordination between sucking and swallowing may indicate an underlying congenital myasthenic syndrome (CMS). Therefore, we suggest testing infants with vocal cord paralysis and feeding difficulties for MUSK and related genes to avoid a late diagnosis of CMS and improve outcomes.

Pediatric Bilateral Vocal Cord Immobility: New Treatment With Preservation of Voice.

OBJECTIVES: Pediatric bilateral vocal cord immobility (BVCI) represents a severe life-threatening condition that often causes severe dyspnea. Endoscopic arytenoid lateral abduction (EALA) is a relatively new, secure, minimal-invasive surgical technique. The present prospective observational study aims to evaluate the effects of EALA in terms of respiratory function, voice quality, and swallowing capabilities. METHODS: Twenty-one pediatric patients with BVCI underwent EALA. Eleven out of 21 patients had tracheostomy at the time of surgery. Pre and postoperative functional assessments included endoscopic evaluation, maximum phonation time, pediatric Voice Handicap Index (pVHI), GIRBAS Scale criteria, and Montreal Children's Hospital Feeding scale (MCH-Feeding scale). peak tidal inspiratory flow or peak inspiratory flow (PIF) and number of desaturations/hour (ODI/h) were evaluated in patients without tracheostomy. RESULTS: Postoperative endoscopy showed glottic airway improvement in all patients. Average time for decannulation was 4.6 weeks. One patient has not yet been decannulated. No major complications occurred. In patients without tracheostomy, we observed a significant improvement of ODI/h and PIF after surgery (p < 0.05) as expected. PVHI, MCH-Feeding scale, and GIRBAS score significantly worsened 1 month after surgical intervention (p < 0.05). One year after surgery, however, all values, except for B and A parameters of the GIRBAS score, returned to levels comparable to those preoperative. CONCLUSIONS: EALA represents a simple, safe and effective solution in pediatric patients with BVCI, avoiding tracheostomy, allowing early decannulation, preserving swallowing function, and maintaining good quality voice. LEVEL OF EVIDENCE: 4 Laryngoscope, 133:2325-2332, 2023.

3D bioprinting in airway reconstructive surgery: A pilot study.

OBJECTIVES: Open surgery is a reliable choice for congenital subglottic stenosis, that represents the third most common congenital anomaly of the larynx. One of the procedures performed is anterior laryngotracheal reconstruction (LTR) with anterior rib graft. The objective of this preliminary study was to evaluate the potential of 3D printing technology for the realization of laryngo-tracheal scaffold in Polycaprolactone (PCL) implanted in vivo in ovine animal model. METHODS: A 3D computer model of a laryngeal graft and a tracheal graft was designed and printed with PCL through 3D additive manufacturing technology. The scaffolds were seeded with autologous mesenchymal stem cells and cultured in vitro for up to 14 days. Anterior graft LTR with 3D printed scaffolds was performed on 5 sheep. The animals underwent endoscopic examinations at the first, 3rd, 6th, and 12th weeks after surgery and before sacrifice. The integration of the material was evaluated by the pathologist. RESULTS: Two animals showed a favourable postoperative course and were sacrificed at 6 months postoperatively. In these cases, we observed endoscopically a complete integration of the cellularized PCL scaffold into the peri-implant tissues, and the pathologist found the growth of respiratory epithelium on the scaffold's inner surface. Other two animals showed a difficult post-operative recovery characterized by respiratory distress resulting in early sacrifice on postoperative days 31 and 33. In these animals we found a poor integration of the grafts into the tracheal structure, and a better integration of the laryngeal scaffold. The last animal developed a wound abscess and was sacrificed 80 days after surgery. We observed, in this case, a poor scaffold integration and an acute inflammatory reaction. CONCLUSIONS: From the preliminary data obtained we found that the excessive stiffness of the material, along with the anatomical features of the sheep, is a major limitation of this study. It will be necessary in the future to create a new biocompatible, more flexible and elastic graft, to achieve greater integration into surrounding tissues. Bioconstructed grafts could simplify surgery for the treatment of laryngo-tracheal stenosis, particularly in the treatment of long tracheal stenoses, which have, at the moment, very complex surgical options. LEVEL OF EVIDENCE: NA.

Pediatric Voice Handicap Index (pVHI): A Tool for Evaluating the Reliability and Validity of Voice Therapy in Children With Benign Vocal Fold Nodules.

OBJECTIVES: benign vocal fold lesions (BVFLs) represent the most common etiology of voice disorders in paediatric population, cause of dysphonia by preventing full vocal fold closure and modifying vibratory characteristics. Of those pathologies, vocal fold nodules represent almost 63% of the cases in children between 0-14 years. Management may include a combination of phonosurgery, voice therapy and pharmacological treatment, but almost 95% of otolaryngologists recommend voice therapy as primary treatment. The Pediatric Voice Handicap Index (pVHI) is nowadays widely used and accepted in the evaluation and monitoring of paediatric patients with dysphonia. The aim of our study was to demonstrate the reliability and validity of PVHI in the evaluation of the effects of voice therapy as treatment of vocal fold nodules in the paediatric population. PATIENTS AND METHOD: in this retrospective chart review twenty-seven (27) dysphonic patients with bilateral vocal fold nodules were treated with behavioral voice therapy approaches, and the PVHI surveys was administered to each patient parent's before and after therapy. The age of the patients ranged from 4 to 14 years (mean age 8.7 y-o); 17 (63%) were male and 10 (37%) females. RESULTS: according to our results, behavioral voice therapy effectively improved pVHI scores from pre- to post-treatment in paediatric patients with bilateral vocal fold nodules.

Changes in Breathing Patterns after Surgery in Severe Laryngomalacia.

BACKGROUND: Most of the studies regarding the surgical treatment of severe laryngomalacia (LM) have been aimed at describing the efficacy of the treatment in terms of improvement of clinical symptoms or anatomical findings. There are no studies specifically aimed at analyzing the changes in breathing patterns following surgical treatment for severe LM. OBJECTIVE: To review the breathing pattern changes before and after corrective surgery in infants with severe LM. STUDY DESIGN: A series of retrospective cases at a tertiary referral children's hospital. METHODS: Retrospective chart review of 81 infants who underwent supra-glottoplasty (SGP) for severe laryngomalacia between 2011 and 2020 at Bambino Gesù Children's Hospital of Rome, Italy. Among the patients, 47 (58%) were male and 34 (42%) were female. Twenty-one patients (26%) had one or more comorbidities condition. The data collected included age, symptoms, a polysomnography/pulse oximetry study, growth rate, the findings from flexible endoscopy, pre- and post-supra-glottoplasty (SGP) pulmonary function tests (PFTs) and, when indicated, 24 h pH-metry. Breathing patterns were studied during restful, normal sleep, using an ultrasonic flow-meter (Exhalyzer, Viasys) which measured: Tidal Volume (Vt), Respiratory Rate (RR), time to peak expiratory flow/expiratory time ratio (tPTEF/Te, an index of the patency of the lower airways) and mean expiratory/mean inspiratory flow ratio (MEF/MIF, an index of the patency of the upper airways) evaluated before surgical procedure (T1) and 3-6 weeks after (T2). Pre- and post-operative mean data were calculated and comparisons made with a Student T-test. RESULTS: The surgical procedure was well tolerated by all infants and no intraoperative or post-operatory long-term complications were noted. In T1, breathing patterns were characterized by low tidal volume and high tPTEF/Te and MEF/MIF ratios, suggesting a severe reduction in the patency of the upper airways in all patients. After surgery (T2), all the previously mentioned variables significantly improved, reaching normal values for the child's age. CONCLUSIONS: Supra-glottoplasty, as already described in several studies, is a safe and efficient procedure to treat severe laryngomalacia during infancy. The improvement in breathing patterns after surgery was reliably confirmed by a lung function test in our study, which showed the diagnostic value of testing respiratory functionality in the laryngomalacia and comparing them to clinical and endoscopic data. Moreover, considering the results obtained, we also propose the use of this available, dependable test to verify its therapeutic effects (post-surgery) and to monitor future respiratory development in these infants. Moreover, we believe that further studies will provide detailed grading guidelines for gravity of the LM, based on these functional lung tests.

Management of Upper Airway Infantile Hemangiomas: Experience of One Italian Multidisciplinary Center.

Airway infantile hemangiomas (IHs) can represent a life-threatening condition since the first months of life. They may be isolated or associated to cutaneous IHs, and/or part of PHACES syndrome. Diagnosis, staging, and indication to treatment are not standardized yet despite the presence in the literature of previous case series and reviews. The diagnosis might be misleading, especially in the absence of cutaneous lesions. Airway endoscopy is the gold standard both for diagnosis and follow-up since it allows evaluation of precise localization and entity of obstruction and/or stricture. Proliferation of IH in the infant airways manifests frequently with stridor and treatment is required as soon as possible to prevent further complications. The first line of therapy is oral propranolol, but duration of treatment is not yet well-defined. All considered, we report the experience of our multidisciplinary center from 2009 to date, on 36 patients affected by airway IHs, and successfully treated with oral propranolol. Thus, the authors propose their experience for the management of airway IHs, specifically early diagnosis, when to perform endoscopy, how to interpret its findings, and when to stop the treatment.

Operative Management of Neonatal Lymphatic Malformations: Lesson Learned From 57 Consecutive Cases.

Aim of the study: Lymphatic malformations (LMs) are rare entities, sometimes difficult to treat, that may be life-threatening when intricately connected to airway structures. Invasive treatments are occasionally required, with sclerotherapy considered the treatment of choice and surgery as a second-line approach. The aim of the present study was to evaluate our multidisciplinary team experience in treating newborns affected by LMs requiring operative management, while defining early outcomes. Methods: Retrospective review of all consecutive patients admitted for LMs requiring operative management between January 2000 and January 2019. Patients were mainly characterized based on anatomical district of the LM (and further stratified based on the development of respiratory distress), need for tracheostomy, number of sclerotherapies, indication for surgery, and residual disease beyond the 1st year. Morbidity and mortality were also evaluated. Fisher exact test and Mann-Whitney test were used as appropriate. Statistical significance was set at p < 0.05. Results: Fifty-seven patients were included in the study, 36 with cervicofacial and/or mediastinal LMs and 21 with LMs of other anatomical districts. Due to the risk of developing respiratory distress at birth, patients with cervicofacial and/or mediastinal LMs were divided into two groups (8/36 group A vs. 28/36 group B). Group A patients are at higher risk for tracheostomy (7/8 group A vs. 1/28 group B, p = 0.0001) and more often require surgical reduction of the residual lymphatic abnormality (5/8 group A vs. 4/28 group B, p = 0.013). They also require sclerotherapies more often, but the difference is not statistically significant (8/8 group A vs. 19/28 group B, p = 0.15). Patients with cervicofacial/mediastinal LMs frequently suffer from persistent residual disease beyond the 1st year of life, significantly more often in group A (7/8 group A vs. 12/28 group B, p = 0.043). Conclusion: LMs are rare conditions with potential life-threatening behavior. Their intrinsic clinical complexity requires a multidisciplinary approach to the affected patients. Planning a long-term follow-up is essential because of the late-term problems those patients may experience.

Surgical Options for Pediatric Bilateral Vocal Cord Palsy: State of the Art.

Management of pediatric bilateral vocal cord palsy (BVCP) is a controversial and challenging topic. It may represent a severe obstructive condition usually associated with respiratory distress, and, in such condition, tracheostomy has been considered the gold standard for a long time. Many surgical options have been described and used to increase the glottic space in BVCP (1), with ongoing research of less invasive techniques. The challenge and current trend in our department and in many major pediatric centers is to avoid tracheotomy through an early treatment. Many techniques introduced in the last decade reduced the number of tracheotomies and increased the decannulation rate. Furthermore, we observed a recent increase in attention to preserve the quality of the voice with new techniques, such as endoscopic arytenoid abduction lateropexy which is in our opinion an important innovation to improve glottic space with satisfactory voice results. We present a review of the literature about the evolution of the treatment options for pediatric BVCP during the years.

Reflux and dental disorders in the pediatric population: A systematic review.

OBJECTIVES: To investigate the role of laryngopharyngeal reflux (LPR) or gastroesophageal reflux disease (GERD) in the development of dental disorders in pediatric population. METHODS: PubMed, Scopus Cochrane database were assessed for subject headings using the PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-analyses) recommendations. Relevant studies published between January 1990 and January 2020 describing the association between reflux and dental disorders in children were retrieved. Three authors reviewed the LPR diagnosis method; inclusion criteria and outcomes. The bias analysis was performed through the tools of the Oxford Centre for Evidence-Based Medicine evidence levels. RESULTS: The electronic search identified 126 publications, of which 11 clinical studies and 2 basic science researches met our inclusion criteria. There is an important heterogeneity between studies about diagnostic method and clinical outcome evaluation. All studies based the reflux diagnosis on GERD criteria. No author considered hypopharyngeal nonacid reflux episodes through hypopharyngeal-esophageal intraluminal multichannel impedance pH monitoring (HEMII-pH). The results of studies support a higher prevalence of dental erosion in children with GERD compared with healthy individuals. Controversial findings were found about the potential association between reflux and caries, and the modification of both saliva composition and production in reflux children. CONCLUSION: The association between reflux and dental disorder is still uncertain. Future studies considering pharyngeal acid and nonacid reflux episodes through HEMII-pH are needed to confirm this hypothesis. The pepsin detection in saliva would be an additional way for detecting LPR in children with dental disorders.

Dehiscence of Bronchial Anastomosis After Lung Transplantation: A Successful Unconventional Treatment.

Anastomotic airway complications, including the dehiscence of the bronchial anastomosis, are a severe cause of morbidity after lung transplantation. We present a case of dehiscence treated by placing an uncovered metal stent into the main bronchus. We usually use this procedure for bronchial stenosis, but in this case, the stent favored the growth of granulation tissue and so the closure of the dehiscence. This procedure was minimally invasive and may be an alternative to an open repair, without precluding open repair in case of failure.

Laryngotracheal Abnormalities in Esophageal Atresia Patients: A Hidden Entity.

Importance: Presence of laryngotracheal abnormalities is associated with increased morbidity and higher mortality rate in esophageal atresia patients. Objective: Determine the prevalence of laryngotracheal abnormalities (LTA) in a prospectively collected cohort of patients treated for esophageal atresia and/or tracheoesophageal fistula (EA/TEF). Analysis of the impact of those airway anomalies in early post-operative outcomes was performed. Patients and Methods: This was a review of a prospectively collected database, including patients from January 2008 to December 2017. Patients enrolled in the present study were treated in a high-volume referral center. Present study included all newborn-infants consecutively treated for EA/TEF. All patients were evaluated by flexible laryngotracheoscopy performed under local anesthesia in spontaneous breathing. In case of airway malformation suspected during flexible endoscopy, a rigid endoscopy was performed to complete airway assessment. If post-operative respiratory symptoms (noisy breathing, respiratory difficulty, failure to extubate, or difficulty feeding) were noted, a second laryngotracheoscopy was performed. Primary study outcome was to evaluate the prevalence of LTA in EA/TEF infants, characterizing of LTA, and their impact on early post-operative outcomes. Those primary study outcomes were planned before data collection began. Results: During the study period 207 patients with EA/TEF were treated. LTA had a period prevalence of 40.1% (83/207). Although no differences were recorded in terms of demographics and clinical presentation, LTA+ infants more frequently required tracheostomy (12/52, 23% vs. 0/124, 0%; p 0.0001) and were at increased risk of death (12/83, 14% vs. 5/124, 4%; p 0.009) in comparison with EA/TEF without LTA. Conclusions: Present data suggest a high prevalence of congenital LTA in patients affected by EA. Most of the abnormalities are congenital and a high proportion of patients with LTA require a tracheostomy. Mortality significantly correlates with the presence of LTA. Systematic airway endoscopic preoperative evaluation has to be pushed forward to minimize LTA-related morbidity and mortality.

Biofilm's Role in Chronic Cholesteatomatous Otitis Media: A Pilot Study.

Cholesteatoma is a destructive lesion involving the temporal bone, which may induce severe complications due to its expansion and erosion of adjacent structures. Bacterial biofilm plays a crucial role in the pathogenesis of many otolaryngologic inflammatory/infectious chronic diseases. In this pilot study, we investigated, by means of cultural examination and with scanning electron microscope, the presence of bacterial biofilm in a series of samples from the epitympanic and mastoid region in patients affected by cholesteatoma and from the promontory region in patients with healthy mucosa who were undergoing to stapes surgery. The preliminary data support the association between biofilm and cholesteatoma (81.3% of the cases) and allow us to hypothesize that keratinized matrix of cholesteatoma may represent the ideal substrate for biofilm colonization and survival; this finding is consistent with the clinical course of aural cholesteatoma, characterized by recurrent exacerbations and recalcitrant course.