The Laryngoscope C 2017 The American Laryngological, V
Rhinological and Otological Society, Inc.
Endoscopic Anterior–Posterior Cricoid Split for Pediatric Bilateral Vocal Fold Paralysis Michael J. Rutter, MD; Catherine K. Hart, MD; Alessandro de Alarcon, MD, MPH; Sam Daniel, MDCM, FRCSC, MSc; Sanjay R. Parikh, MD; Karthik Balakrishnan, MD, MPH; Derek Lam, MD, MPH; Kaalan Johnson, MD; Douglas R. Sidell, MD Objectives/Hypothesis: Children with bilateral true vocal fold immobility (BTVFI) may present with significant airway distress necessitating tracheostomy. The objective of this study was to review our preliminary experience with the anterior– posterior cricoid split (APCS), an endoscopic intervention used as an alternative to tracheostomy in children with BTVFI. Study Design: Multicenter review. Methods: A review of patients undergoing endoscopic APCS for BTVFI at four institutions was performed. Patients were evaluated for the ability to ventilate without the requirement for tracheostomy or reintubation. Additional data extracted included the duration of intubation following APCS, the requirement for additional procedures, and demographics. Surgical success was defined as the ability to avoid tracheostomy and to cap or decannulate without respiratory symptoms if a tracheostomy was present prior to APCS. Results: Nineteen APCS procedures were performed between October 2010 and June 2016. There were 12 male patients, the mean age at APCS was 4.7 months. BTVFI was primarily idiopathic (58%) and associated with other comorbidities (74%). All patients were candidates for tracheostomy prior to APCS. Fourteen patients (74%) were considered surgical successes. Of the unsuccessful patients, three (66%) required tracheostomy following APCS, and one was treated with a posterior cartilage graft. There was one nonsurgical mortality greater than 2 months after APCS and thought to be unrelated to the airway. Conclusions: Endoscopic APCS appears to be a safe and effective intervention for pediatric BTVFI. Under the correct circumstances, this can be performed as a single procedure, obviating tracheostomy. Further study is warranted. Key Words: Pediatrics, airway (nonsleep), laryngology, vocal fold paralysis, endoscopic. Level of Evidence: 4 Laryngoscope, 00:000–000, 2017
INTRODUCTION Bilateral true vocal fold immobility (BTVFI) is a recognized source of stridor and respiratory distress in the
From the Division of Pediatric Otolaryngology–Head and Neck Surgery (M.J.R., C.K.H., A.D.), Cincinnati Children’s Hospital Medical Center, Cincinnati, Ohio, U.S.A.; Department of Otolaryngology–Head and Neck Surgery (M.J.R., C.K.H., A.D.), University of Cincinnati College of Medicine, Cincinnati, Ohio, U.S.A.; Aerodigestive and Esophageal Center (M.J.R., C.K.H., A.D.), Cincinnati Children’s Hospital Medical Center, Cincinnati, Ohio, U.S.A.; Department of Otolaryngology–Head and Neck Surgery (S.D.), Montreal Children’s Hospital, McGill University Health Centre, Montreal, Quebec, Canada; Department of Otolaryngology–Head and Neck Surgery (S.R.P., K.J.), University of Washington School of Medicine and Seattle Children’s Hospital, Seattle, Washington, U.S.A.; Department of Otorhinolaryngology (K.B.), Mayo Clinic College of Medicine, Rochester, Minnesota, U.S.A.; Division of Pediatric Otolaryngology (D.L.), Oregon Health & Science University Doernbecher Children’s Hospital, Oregon Health and Science University, Portland, Oregon, U.S.A.; Department of Otolaryngology–Head and Neck Surgery, (D.R.S.)Division of Pediatric Otolaryngology, Lucile Packard Children’s Hospital Stanford, Stanford University School of Medicine, Stanford, California, U.S.A. Editor’s Note: This Manuscript was accepted for publication January 27, 2017. The authors have no funding, financial relationships, or conflicts of interest to disclose. Send correspondence to Michael J. Rutter, MD, Division of Pediatric Otolaryngology–Head and Neck Surgery, Cincinnati Children’s Hospital Medical Center, 3333 Burnet Avenue, MLC 2018, Cincinnati, OH 45229-3039. E-mail: [email protected] DOI: 10.1002/lary.26547
Laryngoscope 00: Month 2017
neonatal period. Although congenital vocal fold immobility is thought to affect fewer than one child per million births annually, it remains an important consideration for any patient with respiratory distress during infancy.1–3 More than 50% of children with BTVFI will have spontaneous resolution in the first 12 months of life.4,5 Because of this potential for spontaneous return of vocal fold function, any procedure to establish a stable airway should ideally have minimal impact on the ability to phonate and swallow.1,2,6,7 Historically, tracheostomy served this function and was performed on the majority of patients with BTVFI.3,8,9 Although tracheostomy remains the standard for establishing a safe airway in the setting of glottic obstruction, it is associated with a significant long-term care burden, cost, and mortality risks. A recent review of in-hospital mortality after tracheostomy was 14% in a large national series of 885 infants.10 Several alternative procedures have been developed to manage BTVFI. Despite their success at improving airway patency, all have the ability to produce permanent changes at the level of the larynx that increase the risk of aspiration and/or dysphonia postoperatively. As a result, these procedures are not universally indicated if the potential for a return of vocal fold function exists.5,11,12 To avoid tracheostomy while preserving the gross anatomic structure of the glottis, several institutions Rutter et al.: Endoscopic APCS for Vocal Fold Paralysis
1
Operative Technique
Fig. 1. Posterior cricoid incision with laryngeal spreaders in place.
have begun performing endoscopic anterior–posterior cricoid split (APCS) in infants with BTVFI. The purpose of this report was to describe the preliminary experience with endoscopic APCS at four academic children’s hospitals. Operative techniques, pearls and pitfalls, and select patient details are discussed.
MATERIALS AND METHODS This investigation was approved by the institutional review board at all involved institutions. A retrospective analysis of all patients 2 years of age or younger with bilateral vocal fold immobility undergoing APCS between December 2009 and June 2016 were reviewed. At the time of writing, APCS was performed only by the authors of this article at their respective institutions. Patients who required chronic mechanical ventilation were not candidates for this procedure and were therefore not included in this study.
Patient Demographics, Medical and Surgical History Standard demographic data were obtained from the patient record, including age at the time of presentation, age at the time of APCS, and gender. Additional patient-related data extracted include the symptoms on presentation, the etiology of vocal fold immobility, and patient comorbidities. Surgical data extracted included the airway size before and after performing APCS, the APCS technique used (cold vs. laser), the duration of intubation following APCS, the presence or absence of concurrent supraglottoplasty, the requirement for additional endoscopic procedures, and the requirement for tracheostomy following initial postsurgical extubation. The decision to perform additional procedures was determined on an individual basis, influenced primarily by the presence of increased respiratory effort. All patients who received additional endoscopic interventions were included in the analysis regardless of the ultimate surgical outcome. Operative success was defined as having avoided tracheostomy, and in the subset of patients who received tracheostomy prior to the procedure, as the ability to decannulate or cap the tracheostomy without respiratory symptoms.
Laryngoscope 00: Month 2017
2
The procedure is performed under general anesthesia with the patient spontaneously ventilating. Laryngoscopy is performed to allow for palpation of the arytenoid cartilages and to evaluate for concomitant airway abnormalities. An operating laryngoscope is then used to suspend the larynx. For most patients, a small or medium pediatric Lindholm laryngoscope can be accommodated and is the preferred laryngoscope for this purpose. An operating microscope is then brought into the field. The airway is sized using the Myer-Cotton grading scale13 while observing for a leak around the endotracheal tube at 20 cm H2O. A supraglottoplasty is performed if necessary to provide adequate exposure to the subglottis for APCS. Inverted infant laryngeal spreaders are placed, and the anterior and posterior cricoid plates are injected using an orotracheal injection needle with 1% lidocaine with 1:100,000 epinephrine. The posterior cricoid is incised first using a laryngeal knife of CO2 laser, and carried through the cartilage to, but not through, the posterior perichondrium (Fig. 1). The incision can be palpated using a small microlaryngeal right-angle hook to ensure complete division of the cricoid while avoiding esophageal injury. The anterior cricoid is then incised. Visualization is achieved by reversing the orientation of the laryngeal spreaders. External pressure is placed over the anterior cricoid with the nondominant hand, bringing the planned incision site into view (Fig. 2). A mucosal incision is made with a Blitzer knife or sharp sickle knife in the midline of the anterior cricoid mucosa while exercising caution so as to avoid injury to the anterior commissure (Fig. 3). The anterior incision relieves the natural spring of the cricoid ring that will otherwise have the tendency to drive the edges of the posterior cricoid incision together. After completing the anterior and posterior split, a noncompliant balloon is placed traversing the glottis and cricoid plate and expanded once to burst pressure. Balloon size is chosen based on the outer diameter of an endotracheal tube that is a half-size larger than the ageappropriate endotracheal tube for the child. The patient is then nasotracheally intubated with an endotracheal tube that is a half-size larger than the age-appropriate tube for a period up to 2 weeks. For the purposes of this study, the duration of intubation was determined by the individual surgeon. If a tracheostomy tube is in place, an option exists to place an appropriately sized endoscopic suprastomal laryngeal stent, or to temporarily intubate the patient. Microlaryngoscopy with bronchoscopy is repeated approximately 1 day prior to extubation. The APCS site is evaluated by performing a leak test at 20 cm H2O, and the endotracheal tube is downsized to an age-appropriate endotracheal tube size. Extubation is then ideally performed 24 to 48 hours later in the intensive care unit (ICU). Periextubation,
Fig. 2. Anterior cricoid pressure with cricoid plate visualized distal to anterior commissure.
Rutter et al.: Endoscopic APCS for Vocal Fold Paralysis
comorbidities. Excluding Chiari malformation, 11 patients (58%) had at least one additional comorbidity (Table I).
Surgical Data and Patient Outcomes
Fig. 3. Anterior incision, extending through 75% of the cricoid ring. dexamethasone (0.5 mg/kg) is occasionally administered. Although an air leak is often present at the time of extubation in the ICU, this was not a standard requirement for extubation. Following extubation, additional procedures, such as balloon dilation, were performed on an as-needed basis, dictated primarily by the presence of persistent respiratory distress.
Statistical Analysis Patient characteristics and surgical data were compared between those children undergoing successful and unsuccessful APCS procedures based on the study definition of success. Binary categorical variables and continuous variables were evaluated using standard v2 and t tests, respectively. A P value of 2 months following APCS. Although the cause of death in this patient appeared to have been unrelated to the surgical procedure performed, one might argue that tracheostomy would be a more appropriate intervention in critically ill patients. Nevertheless, two patients in this study had multiple comorbidities, and their parents ultimately chose to pursue APCS rather than tracheostomy, both with successful outcomes. Though anecdotal, this result suggests that APCS is safe to consider even in patients with multiple comorbidities.
Limitations This multi-institutional study provides important preliminary data on the outcomes of endoscopic APCS, a procedure designed to avoid tracheostomy or permanent destructive procedures in a subset of patients with BTVFI. Despite the potential benefits, several study limitations merit mention. Foremost, this report represents a case series, and the number of patients is relatively small. As a result, all analyses should be considered exploratory. Likewise, several statistical tests were performed across multiple variables, and the concerns of multiple hypothesis testing should be considered. When controlling the error rate, the return of vocal fold function no longer reaches statistical significance when comparing successful versus unsuccessful APCS procedures. We do acknowledge that the loss of statistical significance when using this correction may be due to the reduction in statistical power. Finally, an ideal preoperative and postoperative comparison for airway obstruction
Laryngoscope 00: Month 2017
would include polysomnography. Unfortunately, complete inpatient sleep studies are not available at all centers, and several patients were intubated prior to APCS.
CONCLUSION Based on our preliminary experience, APCS appears to be a promising alternative to tracheostomy in select patients with BTVFI. Further study is ongoing to better define patient outcomes and appropriate patient selection. Thus far, we feel that APCS may be considered in any patient with BTVFI who is free of other obstructive airway pathology. Over time, controllable surgical and patient-related characteristics may prove influential on the success of this intervention.
BIBLIOGRAPHY 1. Miyamoto RC, Parikh SR, Gellad W, Licameli GR. Bilateral congenital vocal cord paralysis: a 16-year institutional review. Otolaryngol Head Neck Surg 2005;133:241–245. 2. Nisa L, Holtz F, Sandu K. Paralyzed neonatal larynx in adduction. Case series, systematic review and analysis. Int J Ped Otorhinolaryngol 2013; 77:13–18. 3. Holinger LD, Holinger PC, Holinger PH. Etiology of bilateral abductor vocal cord paralysis: a review of 389 cases. Ann Otol Rhinol Laryngol 1976;85:428–436. 4. Berkowitz RG. Congenital bilateral adductor vocal cord paralysis. Ann Otol Rhinol Laryngol 2003;112:764–767. 5. Aubry K, Leboulanger N, Harris R, et al. Laser arytenoidectomy in the management of bilateral vocal cord paralysis in children. Int J Pediatr Otorhinolaryngol 2010;74:451–455. 6. Zbar RI, Smith RJ. Vocal fold paralysis in infants twelve months of age and younger. Otolaryngol Head Neck Surg 1996;114:18–21. 7. Lewis AF, Carron JD, Vedanarayanan V. Congenital bilateral vocal fold paralysis and Charcot-Marie-Tooth disease. Ann Otol Rhinol Laryngol 2010;119:47–49. 8. Gentile RD, Miller RH, Woodson GE. Vocal cord paralysis in children 1 year of age and younger. Ann Otol Rhinol Laryngol 1986:95:622–625. 9. Emery PJ, Fearon B. Vocal cord paralysis in pediatric practice: a review of 71 cases. Int J Ped Otorhinolaryngol 1984:8:147–154. 10. Lee JH, Smith PB, Quek MB, Laughon MM, Clark RH, Hornik CP. Risk factors and in-hospital outcomes following tracheostomy in infants. J Pediatr 2016;173:39–44. 11. Gerber ME, Modi VK, Ward R Gower VM, Thomsen J. Endoscopic posterior cricoid split and costal cartilage graft placement in children. Otolaryngol Head Neck Surg 2012;148:494–502. 12. Zawadzka-Glos L, Frackiewicz M, Mieczyslaw C. Endoscopic laterofixation in bilateral vocal cord paralysis in children. Int J Ped Otorhinolaryngol 2010;74:601–603. 13. Myer CM III, O’Connor DM, Cotton RT. Proposed grading system for subglottic stenosis based on endotracheal tube sizes. Ann Otol Rhinol Laryngol 1994;103:319–323. 14. Cohen SR, Geller KA, Birns JW, et al. Laryngeal paralysis in children: a long-term retrospective study. Ann Otol Rhinol Laryngol 1982;91: 417–424.
Rutter et al.: Endoscopic APCS for Vocal Fold Paralysis
7
Rhinological and Otological Society, Inc.
Endoscopic Anterior–Posterior Cricoid Split for Pediatric Bilateral Vocal Fold Paralysis Michael J. Rutter, MD; Catherine K. Hart, MD; Alessandro de Alarcon, MD, MPH; Sam Daniel, MDCM, FRCSC, MSc; Sanjay R. Parikh, MD; Karthik Balakrishnan, MD, MPH; Derek Lam, MD, MPH; Kaalan Johnson, MD; Douglas R. Sidell, MD Objectives/Hypothesis: Children with bilateral true vocal fold immobility (BTVFI) may present with significant airway distress necessitating tracheostomy. The objective of this study was to review our preliminary experience with the anterior– posterior cricoid split (APCS), an endoscopic intervention used as an alternative to tracheostomy in children with BTVFI. Study Design: Multicenter review. Methods: A review of patients undergoing endoscopic APCS for BTVFI at four institutions was performed. Patients were evaluated for the ability to ventilate without the requirement for tracheostomy or reintubation. Additional data extracted included the duration of intubation following APCS, the requirement for additional procedures, and demographics. Surgical success was defined as the ability to avoid tracheostomy and to cap or decannulate without respiratory symptoms if a tracheostomy was present prior to APCS. Results: Nineteen APCS procedures were performed between October 2010 and June 2016. There were 12 male patients, the mean age at APCS was 4.7 months. BTVFI was primarily idiopathic (58%) and associated with other comorbidities (74%). All patients were candidates for tracheostomy prior to APCS. Fourteen patients (74%) were considered surgical successes. Of the unsuccessful patients, three (66%) required tracheostomy following APCS, and one was treated with a posterior cartilage graft. There was one nonsurgical mortality greater than 2 months after APCS and thought to be unrelated to the airway. Conclusions: Endoscopic APCS appears to be a safe and effective intervention for pediatric BTVFI. Under the correct circumstances, this can be performed as a single procedure, obviating tracheostomy. Further study is warranted. Key Words: Pediatrics, airway (nonsleep), laryngology, vocal fold paralysis, endoscopic. Level of Evidence: 4 Laryngoscope, 00:000–000, 2017
INTRODUCTION Bilateral true vocal fold immobility (BTVFI) is a recognized source of stridor and respiratory distress in the
From the Division of Pediatric Otolaryngology–Head and Neck Surgery (M.J.R., C.K.H., A.D.), Cincinnati Children’s Hospital Medical Center, Cincinnati, Ohio, U.S.A.; Department of Otolaryngology–Head and Neck Surgery (M.J.R., C.K.H., A.D.), University of Cincinnati College of Medicine, Cincinnati, Ohio, U.S.A.; Aerodigestive and Esophageal Center (M.J.R., C.K.H., A.D.), Cincinnati Children’s Hospital Medical Center, Cincinnati, Ohio, U.S.A.; Department of Otolaryngology–Head and Neck Surgery (S.D.), Montreal Children’s Hospital, McGill University Health Centre, Montreal, Quebec, Canada; Department of Otolaryngology–Head and Neck Surgery (S.R.P., K.J.), University of Washington School of Medicine and Seattle Children’s Hospital, Seattle, Washington, U.S.A.; Department of Otorhinolaryngology (K.B.), Mayo Clinic College of Medicine, Rochester, Minnesota, U.S.A.; Division of Pediatric Otolaryngology (D.L.), Oregon Health & Science University Doernbecher Children’s Hospital, Oregon Health and Science University, Portland, Oregon, U.S.A.; Department of Otolaryngology–Head and Neck Surgery, (D.R.S.)Division of Pediatric Otolaryngology, Lucile Packard Children’s Hospital Stanford, Stanford University School of Medicine, Stanford, California, U.S.A. Editor’s Note: This Manuscript was accepted for publication January 27, 2017. The authors have no funding, financial relationships, or conflicts of interest to disclose. Send correspondence to Michael J. Rutter, MD, Division of Pediatric Otolaryngology–Head and Neck Surgery, Cincinnati Children’s Hospital Medical Center, 3333 Burnet Avenue, MLC 2018, Cincinnati, OH 45229-3039. E-mail: [email protected] DOI: 10.1002/lary.26547
Laryngoscope 00: Month 2017
neonatal period. Although congenital vocal fold immobility is thought to affect fewer than one child per million births annually, it remains an important consideration for any patient with respiratory distress during infancy.1–3 More than 50% of children with BTVFI will have spontaneous resolution in the first 12 months of life.4,5 Because of this potential for spontaneous return of vocal fold function, any procedure to establish a stable airway should ideally have minimal impact on the ability to phonate and swallow.1,2,6,7 Historically, tracheostomy served this function and was performed on the majority of patients with BTVFI.3,8,9 Although tracheostomy remains the standard for establishing a safe airway in the setting of glottic obstruction, it is associated with a significant long-term care burden, cost, and mortality risks. A recent review of in-hospital mortality after tracheostomy was 14% in a large national series of 885 infants.10 Several alternative procedures have been developed to manage BTVFI. Despite their success at improving airway patency, all have the ability to produce permanent changes at the level of the larynx that increase the risk of aspiration and/or dysphonia postoperatively. As a result, these procedures are not universally indicated if the potential for a return of vocal fold function exists.5,11,12 To avoid tracheostomy while preserving the gross anatomic structure of the glottis, several institutions Rutter et al.: Endoscopic APCS for Vocal Fold Paralysis
1
Operative Technique
Fig. 1. Posterior cricoid incision with laryngeal spreaders in place.
have begun performing endoscopic anterior–posterior cricoid split (APCS) in infants with BTVFI. The purpose of this report was to describe the preliminary experience with endoscopic APCS at four academic children’s hospitals. Operative techniques, pearls and pitfalls, and select patient details are discussed.
MATERIALS AND METHODS This investigation was approved by the institutional review board at all involved institutions. A retrospective analysis of all patients 2 years of age or younger with bilateral vocal fold immobility undergoing APCS between December 2009 and June 2016 were reviewed. At the time of writing, APCS was performed only by the authors of this article at their respective institutions. Patients who required chronic mechanical ventilation were not candidates for this procedure and were therefore not included in this study.
Patient Demographics, Medical and Surgical History Standard demographic data were obtained from the patient record, including age at the time of presentation, age at the time of APCS, and gender. Additional patient-related data extracted include the symptoms on presentation, the etiology of vocal fold immobility, and patient comorbidities. Surgical data extracted included the airway size before and after performing APCS, the APCS technique used (cold vs. laser), the duration of intubation following APCS, the presence or absence of concurrent supraglottoplasty, the requirement for additional endoscopic procedures, and the requirement for tracheostomy following initial postsurgical extubation. The decision to perform additional procedures was determined on an individual basis, influenced primarily by the presence of increased respiratory effort. All patients who received additional endoscopic interventions were included in the analysis regardless of the ultimate surgical outcome. Operative success was defined as having avoided tracheostomy, and in the subset of patients who received tracheostomy prior to the procedure, as the ability to decannulate or cap the tracheostomy without respiratory symptoms.
Laryngoscope 00: Month 2017
2
The procedure is performed under general anesthesia with the patient spontaneously ventilating. Laryngoscopy is performed to allow for palpation of the arytenoid cartilages and to evaluate for concomitant airway abnormalities. An operating laryngoscope is then used to suspend the larynx. For most patients, a small or medium pediatric Lindholm laryngoscope can be accommodated and is the preferred laryngoscope for this purpose. An operating microscope is then brought into the field. The airway is sized using the Myer-Cotton grading scale13 while observing for a leak around the endotracheal tube at 20 cm H2O. A supraglottoplasty is performed if necessary to provide adequate exposure to the subglottis for APCS. Inverted infant laryngeal spreaders are placed, and the anterior and posterior cricoid plates are injected using an orotracheal injection needle with 1% lidocaine with 1:100,000 epinephrine. The posterior cricoid is incised first using a laryngeal knife of CO2 laser, and carried through the cartilage to, but not through, the posterior perichondrium (Fig. 1). The incision can be palpated using a small microlaryngeal right-angle hook to ensure complete division of the cricoid while avoiding esophageal injury. The anterior cricoid is then incised. Visualization is achieved by reversing the orientation of the laryngeal spreaders. External pressure is placed over the anterior cricoid with the nondominant hand, bringing the planned incision site into view (Fig. 2). A mucosal incision is made with a Blitzer knife or sharp sickle knife in the midline of the anterior cricoid mucosa while exercising caution so as to avoid injury to the anterior commissure (Fig. 3). The anterior incision relieves the natural spring of the cricoid ring that will otherwise have the tendency to drive the edges of the posterior cricoid incision together. After completing the anterior and posterior split, a noncompliant balloon is placed traversing the glottis and cricoid plate and expanded once to burst pressure. Balloon size is chosen based on the outer diameter of an endotracheal tube that is a half-size larger than the ageappropriate endotracheal tube for the child. The patient is then nasotracheally intubated with an endotracheal tube that is a half-size larger than the age-appropriate tube for a period up to 2 weeks. For the purposes of this study, the duration of intubation was determined by the individual surgeon. If a tracheostomy tube is in place, an option exists to place an appropriately sized endoscopic suprastomal laryngeal stent, or to temporarily intubate the patient. Microlaryngoscopy with bronchoscopy is repeated approximately 1 day prior to extubation. The APCS site is evaluated by performing a leak test at 20 cm H2O, and the endotracheal tube is downsized to an age-appropriate endotracheal tube size. Extubation is then ideally performed 24 to 48 hours later in the intensive care unit (ICU). Periextubation,
Fig. 2. Anterior cricoid pressure with cricoid plate visualized distal to anterior commissure.
Rutter et al.: Endoscopic APCS for Vocal Fold Paralysis
comorbidities. Excluding Chiari malformation, 11 patients (58%) had at least one additional comorbidity (Table I).
Surgical Data and Patient Outcomes
Fig. 3. Anterior incision, extending through 75% of the cricoid ring. dexamethasone (0.5 mg/kg) is occasionally administered. Although an air leak is often present at the time of extubation in the ICU, this was not a standard requirement for extubation. Following extubation, additional procedures, such as balloon dilation, were performed on an as-needed basis, dictated primarily by the presence of persistent respiratory distress.
Statistical Analysis Patient characteristics and surgical data were compared between those children undergoing successful and unsuccessful APCS procedures based on the study definition of success. Binary categorical variables and continuous variables were evaluated using standard v2 and t tests, respectively. A P value of 2 months following APCS. Although the cause of death in this patient appeared to have been unrelated to the surgical procedure performed, one might argue that tracheostomy would be a more appropriate intervention in critically ill patients. Nevertheless, two patients in this study had multiple comorbidities, and their parents ultimately chose to pursue APCS rather than tracheostomy, both with successful outcomes. Though anecdotal, this result suggests that APCS is safe to consider even in patients with multiple comorbidities.
Limitations This multi-institutional study provides important preliminary data on the outcomes of endoscopic APCS, a procedure designed to avoid tracheostomy or permanent destructive procedures in a subset of patients with BTVFI. Despite the potential benefits, several study limitations merit mention. Foremost, this report represents a case series, and the number of patients is relatively small. As a result, all analyses should be considered exploratory. Likewise, several statistical tests were performed across multiple variables, and the concerns of multiple hypothesis testing should be considered. When controlling the error rate, the return of vocal fold function no longer reaches statistical significance when comparing successful versus unsuccessful APCS procedures. We do acknowledge that the loss of statistical significance when using this correction may be due to the reduction in statistical power. Finally, an ideal preoperative and postoperative comparison for airway obstruction
Laryngoscope 00: Month 2017
would include polysomnography. Unfortunately, complete inpatient sleep studies are not available at all centers, and several patients were intubated prior to APCS.
CONCLUSION Based on our preliminary experience, APCS appears to be a promising alternative to tracheostomy in select patients with BTVFI. Further study is ongoing to better define patient outcomes and appropriate patient selection. Thus far, we feel that APCS may be considered in any patient with BTVFI who is free of other obstructive airway pathology. Over time, controllable surgical and patient-related characteristics may prove influential on the success of this intervention.
BIBLIOGRAPHY 1. Miyamoto RC, Parikh SR, Gellad W, Licameli GR. Bilateral congenital vocal cord paralysis: a 16-year institutional review. Otolaryngol Head Neck Surg 2005;133:241–245. 2. Nisa L, Holtz F, Sandu K. Paralyzed neonatal larynx in adduction. Case series, systematic review and analysis. Int J Ped Otorhinolaryngol 2013; 77:13–18. 3. Holinger LD, Holinger PC, Holinger PH. Etiology of bilateral abductor vocal cord paralysis: a review of 389 cases. Ann Otol Rhinol Laryngol 1976;85:428–436. 4. Berkowitz RG. Congenital bilateral adductor vocal cord paralysis. Ann Otol Rhinol Laryngol 2003;112:764–767. 5. Aubry K, Leboulanger N, Harris R, et al. Laser arytenoidectomy in the management of bilateral vocal cord paralysis in children. Int J Pediatr Otorhinolaryngol 2010;74:451–455. 6. Zbar RI, Smith RJ. Vocal fold paralysis in infants twelve months of age and younger. Otolaryngol Head Neck Surg 1996;114:18–21. 7. Lewis AF, Carron JD, Vedanarayanan V. Congenital bilateral vocal fold paralysis and Charcot-Marie-Tooth disease. Ann Otol Rhinol Laryngol 2010;119:47–49. 8. Gentile RD, Miller RH, Woodson GE. Vocal cord paralysis in children 1 year of age and younger. Ann Otol Rhinol Laryngol 1986:95:622–625. 9. Emery PJ, Fearon B. Vocal cord paralysis in pediatric practice: a review of 71 cases. Int J Ped Otorhinolaryngol 1984:8:147–154. 10. Lee JH, Smith PB, Quek MB, Laughon MM, Clark RH, Hornik CP. Risk factors and in-hospital outcomes following tracheostomy in infants. J Pediatr 2016;173:39–44. 11. Gerber ME, Modi VK, Ward R Gower VM, Thomsen J. Endoscopic posterior cricoid split and costal cartilage graft placement in children. Otolaryngol Head Neck Surg 2012;148:494–502. 12. Zawadzka-Glos L, Frackiewicz M, Mieczyslaw C. Endoscopic laterofixation in bilateral vocal cord paralysis in children. Int J Ped Otorhinolaryngol 2010;74:601–603. 13. Myer CM III, O’Connor DM, Cotton RT. Proposed grading system for subglottic stenosis based on endotracheal tube sizes. Ann Otol Rhinol Laryngol 1994;103:319–323. 14. Cohen SR, Geller KA, Birns JW, et al. Laryngeal paralysis in children: a long-term retrospective study. Ann Otol Rhinol Laryngol 1982;91: 417–424.
Rutter et al.: Endoscopic APCS for Vocal Fold Paralysis
7
