Cricotracheal resection for pediatric subglottic stenosis.

Until recently, cricotracheal resection (CTR) has not been commonly accepted as a treatment modality for severe subglottic stenosis in the pediatric age group. The reasons have included the risk of a possible dehiscence at the site of the anastomosis, the likelihood of injury to the recurrent laryngeal nerves, and the interference with normal growth of the larynx. Thirty-eight infants and children with a severe subglottic stenosis underwent a partial cricoid resection with primary thyrotracheal anastomosis. Thirty-three patients were tracheotomy-dependent at the time of surgery and 34 were referred cases; 27 were classified as grade III, and 10 as grade IV stenoses according to new Cotton's classification. Nineteen patients were younger than 3 years of age at the time of surgery. The tracheotomy was resected during the surgical procedure in 21 cases. Decannulation was achieved in 36/38 cases after an open procedure. There is one complete restenosis and one good result awaiting decannulation after further surgery for a Pierre Robin syndrome. The authors experienced no lesion of the recurrent laryngeal nerves and no fatality. Thirty-one patients show no exertional dyspnea, three a slight stridor while exercising, and two patients are not decannulated. The postoperative follow-up in longer than 10 years in eight cases. All patients show a normal growth of the larynx and trachea. Compared to laryngotracheoplasties, CTR gives better results for severe subglottic stenosis. This operation should become the treatment of choice for severe (grade III and IV) subglottic stenosis in infants and children.

Laryngotracheal reconstruction for pediatric glotto-subglottic stenosis.

BACKGROUND: The management of pediatric laryngotracheal stenosis (LTS) can be challenging, and laryngotracheal reconstruction (LTR) with cartilage interposition grafting remains the mainstay of surgical treatment for pediatric LTS in most experienced centers. The purpose of this study was to report the results of this procedure in a center where primary cricotracheal resection is frequently performed. METHODS: A retrospective chart review was performed on 45 patients who underwent LTR in our hospital between October 1997 and July 2012. Demographic characteristics and information on the preoperative status, stenosis, and operation were collected. Primary outcomes were measured as overall (ODR) and operation-specific (OSDR) decannulation rates and secondary outcomes as morbidity, mortality, and postoperative functional results. RESULTS: ODR and OSDR were 86.7% (39/45) and 66.7% (30/45), respectively. Re-stenosis was observed in 11/45 (24%) patients, all of whom were endoscopically or surgically treated. Revision surgery was performed in 10 patients, 6 for re-stenosis and 2 for peristomial tracheomalacia. Two children died of mucous obstruction of tracheostomy tube at 3 and 6 months postoperatively (4.4%). Respiratory, voice, and swallowing functions were excellent or good in 86, 75, and 84% of patients, respectively. CONCLUSIONS: LTR for pediatric LTS has high decannulation rates with acceptable morbidity and mortality in selected patients. Most LTR procedures were double-stage for lower grade subglottic stenoses associated with glottic involvement that required stenting. Careful preoperative evaluation and adequate surgical indications are extremely important to achieve high decannulation rates.

Proposal of a new classification for optimising outcome assessment following partial cricotracheal resections in severe pediatric subglottic stenosis.

OBJECTIVE: Creation of a patent subglottic airway after partial cricotracheal resection (PCTR) may not always result in successful decannulation due to associated parameters such as co-morbidity and/or glottic involvement. We classified patients after incorporating these additional parameters into the original Myer-Cotton classification to assess whether this could better predict the outcome measures after PCTR. METHODS: One hundred children with Myer-Cotton grade III or IV subglottic stenosis who underwent PCTR between 1978 and 2008 were identified from a prospectively collected database. The patients were classified into four groups based on the association of co-morbidity and/or glottic involvement. Delay in decannulation, revision open surgery and rates of decannulation were the outcome measures compared between the groups. RESULTS: There were 68 children with Myer-Cotton grade III and 32 children with grade IV stenosis. Based on the new classification, there were 36 children with isolated SGS, 31 with associated co-morbidity, 19 with associated glottic involvement and 14 children with both co-morbidity and glottic involvement. A trend towards less optimal results was noticed with the association of co-morbidity and/or glottic involvement. Statistical significance was reached for maximum decannulation failure in the group with both co-morbidity and glottic involvement. Delayed decannulation significantly correlated in the group with associated glottic involvement. CONCLUSION: This new classification is relatively simple and aimed at providing more accurate and uniform prognostic information to both patients and surgeons when dealing with the whole spectrum of severe SGS.

Long-term voice outcome following partial cricotracheal resection in children for severe subglottic stenosis.

OBJECTIVE: To correlate the postoperative voice outcome to preoperative glottic involvement, following partial cricotracheal resection (PCTR) in children. The glottic involvement was analysed based on the extent of subglottic stenosis (SGS) in the endoscopic image and functional dynamic assessment using flexible endoscopy. METHODS: We conducted an interobserver study in which two ENT surgeons, blinded to one another's interpretation, independently rated the extent of SGS based on the endoscopic image along with the dynamic functional airway assessment, of 108 children who underwent PCTR for grade III or IV stenosis. Based on the observation, the glottic involvement was rated into 4 categories: Evaluation of the voice was based on a parent/patient proxy questionnaire sent in 2008 to assess the current functional status of the patient's voice. RESULTS: Among the 77 patients available for long-term outcome with a minimum 1-year follow-up, 31 patients had isolated SGS free from vocal cords (group A) and 30 had SGS reaching the under surface of vocal cords with partial or no impairment of abduction of vocal cords (group B). Twelve patients belonged to group C with posterior glottic stenosis and/or vocal cord fusion (without cricoarytenoid ankylosis) and 4 patients had transglottic stenosis and or/bilateral cricoarytenoid ankylosis (group D). The long-term voice outcome following PCTR as perceived by the parent or patient was normal in 18% (14 of 77 patients) and the remaining 63 patients demonstrated mild to severe dysphonia. Patients belonging to group A and B exhibited either normal voice or mild dysphonia. Patients in group C demonstrated dysphonia, which was moderate in severity in the majority (83%). All patients in group D with transglottic stenosis and/or CAA showed severe dysphonia. CONCLUSION: Children with associated glottic involvement are at high risk for poor voice outcome following PCTR. The severity of dysphonia was found to be proportional to the preoperative glottic involvement. Preoperative rating of the extent of glottic involvement based on endoscopic image and dynamic assessment was found to be useful in prognosticating the voice outcome.

Prolonged remission of juvenile-onset respiratory papillomatosis: a post-expositional role of the tetravalent anti-HPV vaccine?

Juvenile-onset recurrent respiratory papillomatosis (JORRP) is an HPV-related neoplasm affecting primarily the larynx. JORRP often requires repeated surgical debridement, which yield variable but generally moderate remission periods. We report the case of a 6-year-old boy with severe course JORRP since the age of 2, requiring tracheostomy, that underwent prolonged remission and was decannulated some months after administration of the HPV vaccine. The post-exposure use for the anti-HPV vaccine in JORRP is a topic of capital interest but still poorly characterized. Some published cases suggest a potential post-exposure role of the vaccine in JORRP, but prospective multicentric trials are still needed.

Paralyzed neonatal larynx in adduction. Case series, systematic review and analysis.

OBJECTIVE: Bilateral vocal cord abductor paralysis (BVCAbP) is considered a rare cause of stridor in the newborn. The goal of this work is to present a case series and to review systematically the literature on bilateral vocal cord abductor paralysis in the newborn to better characterize the current knowledge on this entity. METHODS: We performed a systematic literature review with Medline (1950-2011). The authors screened all cases of BVCAbP reported and selected those affecting newborns. RESULTS: Out of the 129 articles screened, 16 were included. A total of 69 cases could be retrieved and analyzed. Associated co-morbidities were found in 54% of the patients, most notably malformative conditions (intracranial or other), or a positive perinatal history (trauma/asphyxia, prematurity). Tracheostomy placement was required in 59% of children, and of these 44% were successfully decannulated. In terms of functional outcome full recovery or improvement were seen in 61% of patients. Major underlying co-morbidities affected negatively the functional outcome (p=.004), but not the need for tracheostomy (p=.604) or the decannulation success rate (p=.063). CONCLUSION: BVCAbP in the newborn is a serious cause of airway obstruction. It can be seen either in a context of multisystem anomalies or as an isolated finding. Newborns with major co-morbidities affecting their normal development are more likely to have poor functional outcomes and to remain tracheostomy-dependant.

Clinically silent subdural hemorrhage causes bilateral vocal fold paralysis in newborn infant.

Bilateral congenital vocal fold paralysis (BVFP) may result from multiple etiologies or remain idiopathic when no real cause can be identified. If obstructive dyspnea is significant and requires urgent stabilization of the airway, then intubation is performed first and an MRI of the brain is conducted to rule out an Arnold-Chiari malformation that can benefit from a shunt procedure and thus alleviate the need for a tracheostomy. Clinically silent subdural hemorrhage without any birth trauma represents another cause of neonatal BVFP that resolves spontaneously within a month. It is of clinical relevance to recognize this potential cause of BVFP as its short duration may alleviate the need for a tracheostomy. In this article, we present such a case and review the literature to draw the otolaryngologist's attention to this possible etiology.

Partial cricotracheal resection for congenital subglottic stenosis in children: the effect of concomitant anomalies.

OBJECTIVE: To review the surgical outcomes of partial cricotracheal resection in children with severe congenital subglottic stenosis and define the effect of concomitant anomalies or syndromes affecting outcome. METHODS: Forty-one children with subglottic stenosis of congenital and mixed (acquired on congenital) etiologies who underwent partial cricotracheal resection were identified from a prospectively collected database. Children with congenital subglottic stenosis and concomitant anomalies/syndromes were compared to children with congenital subglottic stenosis with no syndromes or concomitant anomalies. Operation-specific decannulation rates and complication rates were the primary outcome measures. We performed a two-sample test of proportion using the STATA-10 software for categorical variables to detect differences in proportions. Significance was set at p value<0.05. RESULTS: Twenty-seven (66%) of 41 children had concomitant anomalies/syndromes and 14 (34%) had congenital subglottic stenosis without concomitant anomalies/syndromes. Four patients needed revision surgery in the concomitant anomaly group and two patients needed revision surgery in the non concomitant anomaly group before achieving decannulation. The operation-specific decannulation rate in the concomitant anomaly group was 85% and 86% in the non anomaly group. When compared to children without concomitant anomaly, children with concomitant anomalies were more likely to have delayed decannulation following partial cricotracheal resection. However, this difference was not found to be statistically significant. The complication and operation-specific decannulation rates after partial cricotracheal resection were comparable to children without concomitant anomalies. Mortality rate was 11% (three of 27 patients) in the group with associated congenital anomalies or syndromes. Two patients succumbed to the primary pathology and one patient died due to tracheostomy-tube obstruction. There was no post-operative death in the non anomaly group. CONCLUSION: Partial cricotracheal resection can be done safely and effectively in children with concomitant anomalies/syndromes to achieve decannulation. The post-operative course may be prolonged but the decannulation and the complication rates are comparable to those children with congenital subglottic stenosis without concomitant anomalies.

Endoscopic repair of laryngotracheoesophageal clefts: experience in 17 cases.

OBJECTIVE: To review the presentation and evaluation of laryngotracheoesophageal clefts as well as their treatment modalities, especially endoscopic closure. STUDY DESIGN: retrospective case series. METHODS: All patients treated for laryngotracheoesophageal clefts in our clinic during the last 15 years were included. Analysis of preoperative data, surgical success and functional outcome was performed. RESULTS: A total of 18 patients were included in our study. Cleft distribution was: type I (n=1), type II (n=3), type IIIa (n=5), type IIIb (n=8) and type IVa (n=1). All clefts were closed endoscopically by CO2 laser repair except for two patients who benfited from open surgery (one type I, one type IIIb). 7 of our 18 patients (39%) experienced a complication necessitating reoperation. Surgical treatment of LTEC allowed cessation of feeding tube assistance and artificial ventilation in 47% and 42% of patients respectively. CONCLUSION: Surgical treatement of laryngotracheoesophageal clefts remains a complex procedure with a high rate of morbidity for high grade clefts. Post-surgical difficulties in feeding and breathing are associated with concomitant congenital anomalies. Endoscopic repair is a successful technique for treating up to grade IIIa laryngeal clefts. Further investigation is needed to assess the best approach for treating longer clefts.

Buccal mucosa graft for laryngotracheal reconstruction in severe laryngeal stenosis.

OBJECTIVE: An operative technique is described as a salvage treatment for severe subglottic and supraglottic laryngeal stenosis. In addition to expansion of the laryngeal framework with an anterior cartilage graft, as used in a classical laryngotracheal reconstruction, the scar tissue obliterating the airway lumen is excised and a mucosal graft is placed to reconstruct the inner lining of the airway. The graft is harvested from buccal mucosa. METHODS: The operative technique is outlined. Three cases, 2 paediatric and one adult, with complete or near complete laryngeal stenosis are presented where this operative technique was employed. In all patients several surgeries had been performed previously which were unsuccessful. RESULTS: In all 3 patients a patent airway was achieved with decannulation of the tracheostomy in the 2 paediatric patients. CONCLUSIONS: In patients with severe subglottic or supraglottic airway stenosis where other surgeries have failed, excision of endoluminal scar tissue and placement of a buccal mucosal graft, in addition to conventional laryngotracheal reconstruction, is a promising technique. In revision cases of subglottic stenosis cricotracheal resection might not be an option because of scarring from previous surgeries. This operation is an alternative, which allows an increase in the airway lumen by excising the scar tissue then re-lining the exposed internal lumen. The buccal mucosa reduces granulation formation and re-stenosis.

Congenital lobar emphysema presenting as an airway foreign body.

We report here the case of a 15 months old girl presenting with clinical signs and radiological exams highly suggestive of a foreign body (FB) aspiration. Diagnostic endoscopy revealed an overlooked bronchial malformation responsible for a congenital lobar emphysema (CLE). CLE presenting after one year of age is rare and can easily be misdiagnosed. Therefore, the possibility of an overlooked CLE needs to be known and evoked as an alternative diagnosis when dealing with children presenting with suspected FB aspirations. We report on this unexpected, yet misleading presentation of CLE and review the available literature on the subject.