-
Annals of Translational Medicine Nov 2020Laryngomalacia is the most common cause of stridor in neonates and infants, and supraglottoplasty is the mainstay of surgical treatment. Although low-temperature plasma...
Efficacy and toxicities of low-temperature plasma radiofrequency ablation for the treatment of laryngomalacia in neonates and infants: a prospective randomized controlled trial.
BACKGROUND
Laryngomalacia is the most common cause of stridor in neonates and infants, and supraglottoplasty is the mainstay of surgical treatment. Although low-temperature plasma radiofrequency ablation (LTP-RFA) using coblation technology has been used for treating laryngomalacia, it is still lack of high-quality clinical evidence. Therefore, we conduct this prospective randomized study to clearly define the role of LTP-RFA for the treatment of laryngomalacia in neonates and infants.
METHODS
Between Jan 2017 and Dec 2019, a total of 89 children with laryngomalacia were included for analysis. All patients were initially stratified according to the severity of laryngomalacia. Patients with severe laryngomalacia were randomly assigned to receive LTP-RFA or traditionally surgical supraglottoplasty, while patients with moderate laryngomalacia were assigned to LTP-RFA or observation. The primary end point was the efficacy and toxicities of LTP-RFA by assessing the changes of clinical score and visual analogue scale (VAS) symptom score. The total score was the combination of clinical score with VAS score.
RESULTS
Of the 89 children, 40 children presented with severe laryngomalacia, and the remaining 49 children were diagnosed as moderate laryngomalacia. The median age was 68 days (range, 19 to 337 days). For children with severe laryngomalacia, our results showed that LTP-RFA treatment significantly reduced the operative time (5.55±1.66 18.7±5.31 min, P<0.001), length of hospital stay (6.71±1.15 7.95±1.55 days, P=0.008) and the amount of intraoperative hemorrhage (1.71±1.79 4.90±1.82, P<0.001) when compared to traditionally surgical supraglottoplasty, while the treatment efficacy was comparable between LTP-RFA and traditionally surgical supraglottoplasty in terms of changed total score (P=0.322), changed clinical score (P=0.135) and changed VAS symptom score (P=0.559). Additionally, for children with moderate laryngomalacia, LTP-RFA treatment significantly improved the symptom evaluated by total score (P<0.001), clinical score (P<0.001) and VAS symptom score (P<0.001) in comparison with the observation group. Post-operative pneumonia was observed in 10 patients. No surgical related death was reported.
CONCLUSIONS
The present study indicated that LTP-RFA was an effective treatment option for both severe and moderate laryngomalacia in neonates and infants with a low intraoperative complication. Long-term outcomes of LTP-RFA for laryngomalacia would be reported in further studies.
PubMed: 33313111
DOI: 10.21037/atm-20-3164 -
Medicine Feb 2018Supraglottoplasty is the mainstay of surgical treatment for laryngomalacia. A novel supraglottoplasty surgical technique is needed to achieve better efficacy. The...
BACKGROUND
Supraglottoplasty is the mainstay of surgical treatment for laryngomalacia. A novel supraglottoplasty surgical technique is needed to achieve better efficacy. The purpose of this study was to introduce modified microscopic radiofrequency ablation supraglottoplasty (MMRAS) for the treatment of congenital laryngomalacia and to evaluate the outcome and effectiveness of this novel approach.
METHODS
Seventeen children with severe laryngomalacia who underwent MMRAS were studied retrospectively. Supraglottoplasty of type III laryngomalacia was different from classical method. All the patients were kept intubated for 5 days after surgery to achieve a better epiglottal position and to avoid reconglutination of aryepiglottic folds. The patients' demographic information, symptoms, comorbidities, type of laryngomalacia, synchronous airway lesions and final outcomes were examined.
RESULTS
The median age at the time of surgery was 3.36 months (3 months 10 days). Operative indications included feeding difficulties, noisy breathing or respiratory distress (or both), and sleep-related symptoms. The MMRAS success rate was 82.4%. Most patients were extubated successfully on postoperative day 5. The major postoperative complication was pulmonary infection which occurred in 3 cases (17.6%) and required anti-infective therapy. No perioperative deaths and no long-term complications occurred. Failures were observed in 3 (17.6%) of 17 cases, 2 patients presented with a neurological disease and required tracheostomy, 1 patient relapsed because of postoperative adhesions and later underwent revision supraglottoplasty.
CONCLUSIONS
From these results, we conclude that MMRAS is an effective and safe treatment for symptomatic laryngomalacia and has the potential to provide better breathing, feeding, and sleeping outcomes in children with severe laryngomalacia. Postoperative intubation for 5 days may result in better therapeutic outcomes. Multicenter cooperative studies of comparison between MMRAS and conventional approaches would lend further evidence-based support for this surgical method.
Topics: Catheter Ablation; Epiglottis; Female; Humans; Infant; Infant, Newborn; Laryngomalacia; Male; Microsurgery; Plastic Surgery Procedures; Retrospective Studies; Treatment Outcome
PubMed: 29443747
DOI: 10.1097/MD.0000000000009850 -
Pediatric Clinics of North America Aug 2013Extraesophageal reflux disease, commonly called laryngopharyngeal reflux disease (LPRD), continues to be an entity with more questions than answers. Although the role of... (Review)
Review
Extraesophageal reflux disease, commonly called laryngopharyngeal reflux disease (LPRD), continues to be an entity with more questions than answers. Although the role of LPRD has been implicated in various pediatric diseases, it has been inadequately studied in others. LPRD is believed to contribute to failure to thrive, laryngomalacia, recurrent respiratory papillomatosis, chronic cough, hoarseness, esophagitis, and aspiration among other pathologies. Thus, LPRD should be considered as a chronic disease with a variety of presentations. High clinical suspicion along with consultation with an otolaryngologist, who can evaluate for laryngeal findings, is necessary to accurately diagnose LPRD.
Topics: Child; Humans; Laryngopharyngeal Reflux; Laryngoscopy; Risk Factors
PubMed: 23905824
DOI: 10.1016/j.pcl.2013.04.011 -
Respirology Case Reports Jul 2018Congenital laryngomalacia is the most common cause of stridor in infants and usually resolves without therapy by 12-18 months of age. However, a recent study found...
Congenital laryngomalacia is the most common cause of stridor in infants and usually resolves without therapy by 12-18 months of age. However, a recent study found that laryngomalacia may leave structural and functional traces with increased risk of later respiratory symptoms, suggesting that late-onset laryngomalacia may represent long-term consequences of milder or even undiagnosed forms. Unusual cases demonstrated that inspiratory stridor developed subsequent to upper respiratory tract infections. The lack of airway hyperresponsiveness in adulthood also raised questions regarding the diagnosis of childhood asthma. Laryngomalacia should be distinguished from severe asthma.
PubMed: 29619222
DOI: 10.1002/rcr2.316 -
Children (Basel, Switzerland) Dec 2021Most 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...
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 -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.
PubMed: 34943316
DOI: 10.3390/children8121120 -
Genes May 2023Progressive corneal opacification can result from multiple etiologies, including corneal dystrophies or systemic and genetic diseases. We describe a novel syndrome...
Progressive corneal opacification can result from multiple etiologies, including corneal dystrophies or systemic and genetic diseases. We describe a novel syndrome featuring progressive epithelial and anterior stromal opacification in a brother and sister and their mildly affected father, with all three family members having sensorineural hearing loss and two also with tracheomalacia/laryngomalacia. All carried a 1.2 Mb deletion at chromosome 13q12.11, with no other noteworthy co-segregating variants identified on clinical exome or chromosomal microarray. RNAseq analysis from an affected corneal epithelial sample from the proband's brother revealed downregulation of , , , , , and within the microdeletion interval, with no notable effect on the expression of nearby genes. Pathway analysis showed upregulation of collagen metabolism and extracellular matrix (ECM) formation/maintenance, with no significantly down-regulated pathways. Analysis of overlapping deletions/variants demonstrated that deleterious variants in were found in patients with laryngomalacia and sensorineural hearing loss, with the latter phenotype also being a feature of variants in the partially overlapping 1 locus, yet none of these had reported corneal phenotypes. Together, these data define a novel microdeletion-associated syndromic progressive corneal opacification and suggest that a combination of genes within the microdeletion may contribute to ECM dysregulation leading to pathogenesis.
Topics: Male; Female; Humans; Laryngomalacia; Hearing Loss, Sensorineural; Syndrome; Siblings; Microarray Analysis; Protein Serine-Threonine Kinases; Tumor Suppressor Proteins
PubMed: 37239394
DOI: 10.3390/genes14051034 -
Italian Journal of Pediatrics Mar 2020Chronic cough is a common symptom in children and protracted bacterial bronchitis (PBB) is one of the causes of chronic cough. However, the understanding of this disease...
BACKGROUND
Chronic cough is a common symptom in children and protracted bacterial bronchitis (PBB) is one of the causes of chronic cough. However, the understanding of this disease remains limited. The present study aims to update PBB in children.
METHODS
The clinical data of children with PBB from 2014 to 2018 were retrospectively analyzed, and PBB clinical features of published studies were summarized. Electronic databases were searched in May 2019. Clinical studies were included in the present study. Reviews were undertaken in duplicate.
RESULTS
Totally 712 cases were analyzed in this study, including 52 cases in our center and 660 cases from 14 studies. In the 52 cases, 88.5% of patients with PBB were less than 6 years old and all of them complained of wet cough. Three cases were confirmed with laryngomalacia, and microbiologically-based-PBB were identified in 13 cases (9 Streptococcus pneumonia, 3 Staphylococcus aureus, and 1 Pseudomonas aeruginosa). Twenty cases were completely remitted after treatment. In the 14 studies, the patients with PBB were typically younger than 3 years old, accompanying wheezing and airway malacia. Co-infection was common in most western cases, Streptococcus pneumonia, Haemophilus influenza and Moraxella catarrhalis were the top three pathogens. Symptoms were improved in most patients, whereas some cases with comorbidities required prolonged antibiotics treatment.
CONCLUSIONS
PBB is common in male infants with chronic wet cough and accompanied by wheezing and airway deformities. Most cases are clinically diagnosed PBB in China and microbiologically-based-PBB is common in western countries. Co-infection could be found, Streptococcus pneumoniae and Haemophilus influenza were the most frequent etiology in China and western countries, respectively. Patients with comorbidities may need extended antibiotics treatment for more than 2 weeks.
Topics: Adolescent; Anti-Bacterial Agents; Bacterial Infections; Bronchitis; Child; Child, Preschool; Chronic Disease; Cough; Female; Humans; Infant; Infant, Newborn; Male; Retrospective Studies
PubMed: 32228653
DOI: 10.1186/s13052-020-0802-z -
Medicine Jan 2022To review our experience with endoscopic coblation-assisted and partial arytenoidectomy (ECPA) in treating idiopathic bilateral vocal cord paralysis (BVCP).A...
To review our experience with endoscopic coblation-assisted and partial arytenoidectomy (ECPA) in treating idiopathic bilateral vocal cord paralysis (BVCP).A retrospective analysis of thirty-three infants (19 boys and 14 girls, aged 1-10 months) with idiopathic BVCP undergoing ECPA was performed. The therapeutic process and outcomes (surgical success, swallowing function, and voice) were reviewed. The follow-up period was >33 months.Among the thirty-three infants with idiopathic BVCP, surgery was successful in twenty-nine cases but failed in four cases. Twenty-one, nine, and three patients underwent right, left, and bilateral ECPA, with surgical success rates of 90.5%, 100.0%, and 33.3%, respectively. In addition, four and six cases were combined with subglottic stenosis (SGS) and laryngomalacia, respectively. The surgical success rates of BVCP alone and BVCP+ other airway abnormalities were 95.6% and 70.0%, respectively. During the follow-up, five infants had slight difficulty swallowing, 12 infants had partial or complete recovery movement of at least one vocal cord with satisfactory voice outcome, and five infants had early granuloma formation, which disappeared spontaneously.ECPA appears to be a promising alternative to tracheostomy and initial management in infants with idiopathic BVCP who are free of other airway abnormalities.
Topics: Endoscopy; Female; Humans; Infant; Laryngeal Muscles; Laryngoplasty; Laryngoscopy; Male; Retrospective Studies; Vocal Cord Paralysis
PubMed: 35089194
DOI: 10.1097/MD.0000000000028593 -
Frontiers in Pediatrics 2023The aim of this study was to describe a novel surgical technique of endoscopic percutaneous repair in pediatric patients with type 1, type 2 and type 3 laryngeal cleft...
OBJECTIVE
The aim of this study was to describe a novel surgical technique of endoscopic percutaneous repair in pediatric patients with type 1, type 2 and type 3 laryngeal cleft (LC).
METHODS
A retrospective study involving 12 patients with LC was performed at a tertiary pediatric hospital between February 2021 and June 2022. Endoscopic percutaneous repair was performed in all the patients. Information such as demographics, comorbidities, history of tracheostomy and the open approach for the repair, type of cleft and complications were analyzed.
RESULTS
Twelve patients were diagnosed with LC. The median age of the patients at the time of surgery was 8.50 months (interquartile range, 49.50 months). Seven patients had tracheomalacia, four patients had subglottic stenosis, three patients had laryngomalacia. No surgical complications occurred in the 10 patients who underwent the primary procedure. For two patients who underwent a secondary procedure, endoscopic percutaneous repair failed again to heal the cleft. During the follow-up period after surgery, none of the patients had stridor, recurrent pneumonia, feeding difficulties, or dyspnea. Follow-up modified barium swallow postoperatively demonstrated no aspiration in 10 patients. Only the 2 patients with a secondary procedure had intermittent cough while taking large gulps of water. The cure rate of endoscopic percutaneous repairer was 83.3% (95% confidence interval: 73.9%-92.8%).
CONCLUSION
Endoscopic percutaneous repair should be considered as an alternative to the open transcervical approach and the traditional endoscopic approach for type 1, type 2 and type 3 LC.
PubMed: 36911017
DOI: 10.3389/fped.2023.1113894 -
Journal of Clinical Sleep Medicine :... Apr 2021Obstructive sleep apnea (OSA) and central sleep apnea (CSA) are common in infants with laryngomalacia. The purpose of this study was to evaluate developmental changes in...
STUDY OBJECTIVES
Obstructive sleep apnea (OSA) and central sleep apnea (CSA) are common in infants with laryngomalacia. The purpose of this study was to evaluate developmental changes in sleep-related breathing disorders over time in infants with laryngomalacia and understand the effect of supraglottoplasty (SGP) and nonsurgical treatment.
METHODS
This is a retrospective review of infants with laryngomalacia who had at least 2 diagnostic polysomnography studies performed from January 2000 and May 2015. We included infants who had either OSA or CSA. Comparison of sleep and respiratory parameters by age group (0-6, 6-12, and >12 months old) was performed in both SGP and non-SGP groups using a mixed-effect regression model. A log-normal mixed model was used to explore the changes in sleep and respiratory parameters with age. The time to resolution of CSA and OSA was analyzed using nonparametric survival analysis.
RESULTS
A total of 102 infants were included; 57 had only OSA and 45 had both CSA and OSA. There were significant decreases in apnea-hypopnea index, obstructive index, central apnea index, and arousal index with increasing age in both SGP and non-SGP groups. The mean age at resolution of CSA (central apnea index < 5) was 7.60 months old for SGP and 12.57 months old for non-SGP (P < .05). There were no significant differences in the mean age at resolution of OSA (obstructive index < 1; 35.18 [SGP] vs 41.55 months [non-SGP]; P = .60) between SGP and non-SGP groups. Infants with neurologic disease, congenital anomalies, or genetic syndromes required significantly more time to resolve OSA (28.12 [normal] vs 53.13 [neurological] vs 59.53 months [congenital anomalies and genetic]; P < .01).
CONCLUSIONS
Both OSA and CSA improve in infants with laryngomalacia with increasing age regardless of SGP. The mechanism underlying these changes may involve airway growth and maturation of respiratory control. Time to resolution of OSA is affected by the presence of neurologic diseases, congenital anomalies, and genetic syndromes. Further studies are needed to confirm these findings and to evaluate long-term outcomes in this population.
Topics: Humans; Infant; Infant, Newborn; Laryngomalacia; Polysomnography; Retrospective Studies; Sleep; Sleep Apnea, Obstructive
PubMed: 33295276
DOI: 10.5664/jcsm.9046