Endoscopic management of bilateral vocal fold paralysis in newborns and infants

doi:10.1016/j.ijporl.2017.03.021

International Journal of Pediatric Otorhinolaryngology

Volume 97, June 2017, Pages 13-17

https://doi.org/10.1016/j.ijporl.2017.03.021 Get rights and content

Abstract

Introduction

Bilateral vocal cord paralysis in adducted position (BVCPAd) is a severe cause of airway obstruction and usually debuts with stridor and airway distress necessitating immediate intervention. Tracheostomy has long been the gold standard for treating this condition, but has significant associated morbidity and mortality in pediatric patients. New conservative procedures have emerged to treat this condition thus avoiding tracheostomy, like endoscopic anterior and posterior cricoid split (EAPCS). The objective of this paper was to review our experience with EAPCS in newborns and infants.

Methods

Prospective study involving patients undergoing endoscopic EAPCS for symptomatic BVCPAd. The primary outcomes were tracheostomy avoidance and resolution of airway symptoms.

Results

Three patients underwent EAPCS between January 2016 and December 2016. All patients stayed at least 7 days in the Intensive Care Unit (ICU) intubated. All patients presented complete resolution of their symptoms due to airway obstruction, without the need for tracheostomy.

Conclusion

EAPCS is a novel and effective alternative to treat BVCPAd in patients under 1 year old. Our study is an initial experience; more cases are required to identify the real impact and benefits of this technique and to determine the proper selection of patients.

Introduction

Stridor in the neonatal period may be a manifestation of multiple congenital anomalies of the respiratory tract. Due to the size and the shape of the airway of the newborn, stridor represents a warning sign that could be secondary to a potentially dangerous anomaly. Independent of its etiology, the approach is to ensure a stable and secure airway, and then develop a diagnostic approach [1].

Unilateral or bilateral vocal cord paralysis (VCP) is a known cause of stridor in the neonate [2], corresponding to the second most common cause of congenital anomaly of the larynx after laryngomalacia, being more frequent than congenital subglottic stenosis [3]. Reports in the literature refer to both unilateral and bilateral vocal cord paralysis [4], [5], [6], [7], [8], [9], the latter being a rare condition, with an estimated incidence of 0.75 cases per 1 million births per year [10].

The term VCP is one of the most common causes of vocal cord immobility, which covers a wide range of clinical conditions and includes causes such as ankylosed cricoarytenoid joints and posterior glottis stenosis. The most important considerations in the diagnostic approach of pediatric patients with VCP is to determine if a VCP is congenital or acquired and if it involves one or both vocal folds. More than 50% of pediatric patients present a spontaneous recovery of their VCP in the first 12 months of life [4], [5], [7].

Bilateral true vocal fold immobility (BTVFI) in adducted position represents a subgroup that has the additional challenge of upper airway obstruction during inspiration. It has been described as idiopathic or secondary to neurological disorders such as the Arnold-Chiari malformation, hydrocephalus, myelomeningocele, cerebral palsy, hypoxia and hemorrhage [10].

The primary goal in the management of VCP during infancy is establishing an adequate airway while maintaining an acceptable voice and safe swallowing function. Tracheostomy has long been the gold standard to achieve this goal. This procedure allows to preserve an adequate respiratory function and laryngeal architecture, but has significant morbidity and even mortality in the pediatric group [11]. Since there is potential for spontaneous recovery of vocal cord mobility, in the last 20 years, the management of this condition has evolved to a more conservative approach, developing different management strategies and surgical alternatives seeking to increase the glottis area thus avoiding a tracheostomy. Unfortunately, these procedures may result in irreversible sequela at the level of the larynx and an increased risk of postoperative aspiration and/or dysphonia [4], [5], [6], [7], [8], [9], [12], [13].

We present 3 cases of bilateral vocal cord paralysis with onset in the neonatal period with significant compromise of respiratory function, in whom, to avoid a tracheostomy and its associated morbidity, we perform a novel surgical technique that aims to achieve relief of airway obstruction without compromising swallowing function and voice. This technique which was recently described by Rutter [14] consists of an endoscopic anterior and posterior cricoid split (EAPCS) associated with balloon dilation and endotracheal tube (ETT) stenting postoperatively. The aim of this paper is to report our initial experience with this surgical technique, and to develop a discussion of its most important aspects and findings.

Section snippets

Material and method

Prospective study in which patients younger than 1-year-old with bilateral vocal cord paralysis in adducted position with compromised respiratory function at the Hospital Guillermo Grant Benavente of Concepcion, Chile during the year 2016 were enrolled to be treated by EAPCS. Parental consent was obtained prior to the procedure. The demographic data included in this study were gender, age of onset, age at time of surgery, symptoms, etiology and comorbidities. The surgical information gathered

Case 1

A 2 months old male patient without comorbidities, but with family history of an older brother with congenital BTVFI, presented with biphasic stridor since birth with progressive supraclavicular and intercostal retraction evolving to severe respiratory distress at 2 months of age. Flexible scope exam revealed a bilateral vocal cord paralysis in adducted position. A magnetic resonance imaging of the brain (MRI) and echocardiography did not reveal any other pathological findings.

In the operating

Discussion

Congenital vocal cord paralysis is bilateral in over 50% of reported cases [4], [8], [11], [16]. Its most common symptoms are stridor, dyspnea and swallowing disorder. Despite being a rare pathology, bilateral vocal cord paralysis in adducted position (BVCPAd) should be considered in the differential diagnosis of any newborn with stridor. The diagnosis is made by direct observation with an endoscopic laryngeal examination, which allows to characterize the dynamic aspect of motor function and

Conclusion

In patients with bilateral adducted vocal cord paralysis younger than one-year-old, EAPCS in properly selected patients is a surgical alternative to tracheostomy, with lower perioperative morbidity and a positive impact on quality of life while preserving the laryngeal architecture. Our study is an initial experience; more cases are required to identify the real impact and the benefits of this technique and to determine the proper selection of patients.

Conflicts of interest

None.

Acknowledgments

This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

References (23)

A.L. de Jong et al.
Vocal cord paralysis in infants and children
Otolaryngol. Clin. North Am.
(2000)
I. de Gaudemar et al.
Outcome of laryngeal paralysis in neonates: a long term retrospective study of 113 cases
Int. J. Pediatr. Otorhinolaryngol.
(1996)
P.J. Emery et al.
Vocal cord palsy in pediatric practice: a review of 71 cases
Int. J. Pediatr. Otorhinolaryngol.
(1984)
R.I. Zbar et al.
Vocal fold paralysis in infants twelve months of age and younger
Otolaryngol. Head. Neck Surg.
(1996)
E.Y. Chen et al.
Bilateral vocal cord paralysis in children
Otolaryngol. Clin. North. Am.
(2008)
L. Nisa et al.
Paralyzed neonatal larynx in adduction. Case series, systematic review and analysis
Int. J. Pediatr. Otorhinolaryngol.
(2013)
M. Daniel et al.
Neonatal stridor
Int. J. Pediatr.
(2012)
S.M. Ahmad et al.
Congenital anomalies of the larynx
Otolaryngol. Clin. North. Am.
(2007)
S.R. Cohen et al.
Laryngeal paralysis in children: a long-term retrospective study
Ann. Otol. Rhinol. Laryngol.
(1982)
H. Daya et al.
Pediatric vocal fold paralysis: a long-term retrospective study
Arch. Otolaryngol. Head. Neck Surg.
(2000)

R.D. Gentile et al.

Vocal cord paralysis in children 1 year of age and younger

Ann. Otol. Rhinol. Laryngol.

(1986)

Cited by (16)

Outcomes of endoscopic percutaneous suture lateralization for neonatal and infantal bilateral vocal ford paralysis
2023, Brazilian Journal of Otorhinolaryngology
Bilateral Vocal Fold Paralysis (BVFP) is a rare but significant resource of respiratory distress in neonates and infants. The objective of this study was to evaluate the efficacy and functional outcomes of Endoscopic Percutaneous Suture Lateralization (EPSL) for the treatment of BVFP in neonates and infants.
A case series study of nine patients undergoing EPSL for BVFP between January 2019 and June 2021 was conducted. All patients were candidates for tracheostomy prior to EPSL. Demographic features including gender, age at diagnosis and surgery, main symptoms, airway comorbidities, airway support, and etiology were collected preoperatively. Patients were evaluated for breathing, swallowing and phonation postoperatively. Surgical success was defined as the ability to avoid tracheostomy. Functional Endoscopic Evaluation of Swallowing (FEES) was conducted to identify aspiration. Voice evaluation was based on clinical observation.
Nine patients underwent ten EPSL procedures (one in the left vocal fold, and nine in the right vocal fold). Eight patients (8/9) were able to successfully avoid tracheostomy and feed orally without aspiration after the procedure. One patient experienced clinical improvement in respiratory support requirements and underwent laparoscopic nissen and gastrostomy tube placement. At the last follow-up, two patients regained normal voice, two patients had mild dysphonia, and five patients had moderate dysphonia. Five patients showed partial return of the contralateral vocal fold function.
EPSL is an effective and safe treatment for neonatal and infantal BVFP, which enables patients free from tracheostomy without significant impact on swallowing function or phonation.
Level 4.
Clinical and Financial Outcomes Associated With Vocal Fold Paralysis in Congenital Cardiac Surgery
2022, Journal of Cardiothoracic and Vascular Anesthesia
Citation Excerpt :
Postoperative tracheostomy and feeding tube use are increased in this patient subset and may be used to mitigate aspiration of oral contents into the respiratory tract.29 Although endoscopic anterior and posterior cricoid split has emerged as a possible alternative to tracheostomy, further investigation is necessary to verify its clinical use.29,30 Consistent with the authors’ findings, VFP previously has been associated with increased odds of aspiration pneumonia and postoperative tracheostomy in adult cardiac and thoracic surgical patients.25,31
Vocal fold paralysis (VFP) has proven to increase resource use in several surgical fields. However, its burden in congenital cardiac surgery, a specialty known to be associated with high resource use, has not yet been examined. The authors aimed to assess the impact of VFP on costs, lengths of stay, and readmissions following congenital cardiac surgery.
A retrospective analysis of administrative data.
The 2010-2017 National Readmissions Database.
All pediatric patients undergoing congenital cardiac surgery.
None.
Vocal fold paralysis was defined using International Classification of Diseases, Ninth and Tenth Revisions, diagnosis codes. The primary outcome of interest was 30-day nonelective readmissions and 90-day readmissions; costs, length of stay, and discharge status also were considered. Of an estimated 124,486 patients meeting study criteria, 2,868 (2.3%) were identified with VFP. Incidence of VFP increased during the study period (0.7% in 2010 to 3.2% in 2017, nptrend < 0.001). Rates of nonhome discharge (30.0% v 16.4%, p < 0.001), 30-day readmission (23.9% v 12.4%, p < 0.001), and 90-day readmission (8.3% v 4.4%, p = 0.03) were increased in the VFP cohort, as were lengths of stay (42.1 v 27.0 days, p < 0.001) and costs ($196,000 v $128,000, p < 0.001). After adjustment for patient and hospital factors, VFP was independently associated with greater odds of nonhome discharge (adjusted odds ratios [AOR], 1.66, 95% CI, 1.14-2.40), 30-day readmission (AOR, 1.58, 95% CI, 1.03-2.42), 90-day readmission (AOR, 2.07, 95% CI, 1.22-3.52), longer lengths of stay (+ 6.1 days, 95% CI, 1.3-10.8), and higher hospitalization costs (+$22,000, 95% CI, 3,000-39,000).
Readmission rates after congenital cardiac surgery are significantly greater among those with VFP, as are costs, lengths of stay, and nonhome discharges. Therefore, further efforts are necessary to increase awareness and reduce the incidence of VFP in this vulnerable population to minimize the financial burden of congenital cardiac surgery on the US medical system.
Endoscopic anterior-posterior cricoid split to avoid tracheostomy in infants with bilateral vocal fold paralysis
2020, International Journal of Pediatric Otorhinolaryngology
Citation Excerpt :
Endoscopic anterior-posterior cricoid split (APCS) with balloon dilation was recently described as an alternative to tracheostomy in select infants with BVFP [23]. Preliminary reports have suggested this procedure is safe and effective, while also minimizing permanent structural changes to the larynx that might lead to aspiration and dysphonia [23,24]. In this study, we report our institution's early experience using APCS in infants with BVFP, describing our surgical technique and attempting to describe factors that may predispose to success or failure in our patient population.
Infants with bilateral vocal fold paralysis (BVFP) can present with stridor and respiratory distress necessitating tracheostomy. The endoscopic anterior-posterior cricoid split (APCS) with balloon dilation procedure has been described as an alternative to tracheostomy in these patients. Here, we report our institution's preliminary experience with APCS and evaluate patient factors that may predispose to the success or failure of this procedure in infants with BVFP.
Electronic charts of patients who underwent APCS with balloon dilation at a single institution were reviewed for the following variables: patient demographics, comorbidities, etiology of vocal fold paralysis, symptoms at presentation, need for respiratory support, intra-operative findings, duration of intubation, perioperative medical treatments, subsequent airway management, and findings of follow-up evaluations. APCS was considered successful if the patient did not undergo tracheostomy.
Six patients underwent APCS with balloon dilation between August 2014 and October 2019. Four patients (66.7%) were male, and 5 of 6 (83.3%) were born full term. The etiology of vocal fold paralysis was idiopathic in four patients (66.7%) and associated with a neuromuscular disorder and hydrocephalus in the remaining two patients. Mean age at the time of the procedure was 10.3 weeks. Three infants (50%) avoided tracheostomy and had marked alleviation of airway symptoms. Three patients who required tracheostomy had more severe respiratory symptoms pre-operatively, requiring either intubation or positive pressure support. Among all patients, there were no mortalities in our series.
APCS is safe and may be effective at the elimination of airway symptoms in select infants with BVFP, avoiding the need for tracheostomy, however more investigation is needed to establish its precise role in this patient population.
Innovations in Airway Surgery
2019, Otolaryngologic Clinics of North America
Citation Excerpt :
Their approach involved division of the cricoid ring, allowing for expansion, followed by intubation with a larger endotracheal tube for a period of time as a stent. More recently, this general technique has been applied endoscopically by several groups, for management of both subglottic stenosis (SGS) and bilateral vocal fold immobility (BVFI).38–42 The procedure is performed under general anesthesia with the patient spontaneously ventilating.
Airway evaluation in children with single ventricle cardiac physiology
2018, International Journal of Pediatric Otorhinolaryngology
Citation Excerpt :
Instead, children will often present with dysphonia and dysphagia from unilateral VCP [16]. Bilateral vocal cord paralysis can, at times, be managed conservatively [17–19]. This study may also be underpowered to detect bilateral vocal cord paralysis as a risk factor for tracheostomy placement as a recent large, retrospective database study suggest that there may be an association [20].
Children with single ventricle cardiac physiology (SVC) often require airway procedures as an adjunct to their care. Descriptive analysis with a focus on outcomes of airway procedures in SVC patients have not been fully described in the literature.
Retrospective, single-center cohort review of 270 patients born between Aug-2007 and Jan-2017. Patients were identified by cardiac database for single ventricle pathophysiology. A subset of these patients were identified to have been evaluated by otolaryngology with airway evaluations and/or interventions.
88/270 patients (32.6%) required investigation or intervention for airway pathology. The most frequent procedure was flexible fiberoptic laryngoscopy (58/88 patients); it was the only procedure performed in 40 patients. Seventeen patients required tracheostomies with an associated increased length of stay (p < 0.001). Patients with cardiac procedures involving dissection around the aortic arch were considered higher airway risk due to the threat of recurrent laryngeal nerve injury, and were more likely to have vocal cord paralysis (58%) compared to patients with lower risk procedures (21%; p < 0.001). However, on multivariate logistic regression, vocal cord paralysis did not statistically impact the odds for tracheostomy placement, although the presence of subglottic stenosis increased the odds ratio of tracheostomy by 14.7 (p = 0.02).
Children with SVC often require airway evaluation and intervention. Patients with high risk cardiac procedures had a higher risk of recurrent laryngeal nerve injury but the presence of subglottic stenosis was the best predictor for a tracheostomy. This study represents one of the largest series of SVC children evaluated for airway pathology.
Endoscopic percutaneous suture lateralization for neonatal bilateral vocal fold immobility
2018, International Journal of Pediatric Otorhinolaryngology
Citation Excerpt :
The primary aim of treatment is to provide an adequate airway for ventilation while minimizing adverse effects such as aspiration and dysphonia. Current management options vary widely on a spectrum that includes non-invasive positive pressure ventilation (NIPPV), endoscopic surgery such as cricoid split [2–4], cordotomy [5], and tracheotomy. In some series, up to 90% of patients with BVFI underwent tracheostomy [6,7].
Bilateral vocal-fold immobility (BFVI) is a rare but significant cause of severe respiratory distress in neonates. The primary aim of treatment is to provide an adequate airway while minimizing adverse effects such as aspiration and dysphonia. Our objective here is to describe the outcomes of a series of neonates undergoing percutaneous endoscopic suture lateralization for BVFI using a novel technique.
In this retrospective case series, we present 6 neonates (mean age: 18 days) with BVFI from three tertiary academic medical centers. The etiologies included 4 idiopathic, 1 unspecified neurodegenerative disorder, and 1 acquired from cardiac surgery. All had stridor and respiratory distress with hypoxemia requiring respiratory support at diagnosis. Endoscopic vocal-fold lateralization was performed under spontaneous-breathing suspension laryngoscopy using a novel technique of percutaneous needle-directed placement of 4–0 prolene suture without use of specialized equipment.
All patients had clinical improvement in stridor and respiratory support requirements and avoided tracheostomy. One patient had persistent aspiration after lateralization that resolved after suture removal. One patient required bilateral lateralization procedures. One patient expired of epilepsy due to neurodegenerative disease unrelated to airway pathology. At last follow-up (mean 12.6 months), 5/5 remaining patients were on room air without tracheostomy and feeding orally without aspiration; 4/5 had partial or complete return of vocal-fold function.
Endoscopic percutaneous suture lateralization may be a safe and effective non-destructive primary treatment modality for neonatal BVFI. All neonates undergoing this procedure avoided tracheotomy.

View all citing articles on Scopus

View full text

Endoscopic management of bilateral vocal fold paralysis in newborns and infants

Abstract

Introduction

Methods

Results

Conclusion

Introduction

Section snippets

Material and method

Case 1

Discussion

Conclusion

Conflicts of interest

Acknowledgments

Otolaryngol. Clin. North Am.

Int. J. Pediatr. Otorhinolaryngol.

Int. J. Pediatr. Otorhinolaryngol.

Otolaryngol. Head. Neck Surg.

Otolaryngol. Clin. North. Am.

Int. J. Pediatr. Otorhinolaryngol.

Neonatal stridor

Int. J. Pediatr.

Congenital anomalies of the larynx

Otolaryngol. Clin. North. Am.

Laryngeal paralysis in children: a long-term retrospective study

Ann. Otol. Rhinol. Laryngol.

Pediatric vocal fold paralysis: a long-term retrospective study

Arch. Otolaryngol. Head. Neck Surg.

Vocal cord paralysis in children 1 year of age and younger

Ann. Otol. Rhinol. Laryngol.