543100
research-article2014
AORXXX10.1177/0003489414543100Annals of Otology, Rhinology & LaryngologySidell et al
Article
Surgical Management of Posterior Glottic Diastasis in Children
Annals of Otology, Rhinology & Laryngology 2015, Vol. 124(1) 72–78 © The Author(s) 2014 Reprints and permissions: sagepub.com/journalsPermissions.nav DOI: 10.1177/0003489414543100 aor.sagepub.com
Douglas R. Sidell, MD1, Stephanie Zacharias, PhD, CCC-SLP2,3,4, Karthik Balakrishnan1, Michael J. Rutter, MBChB, FRACS1,5,6, and Alessandro de Alarcón, MD, MPH1,3,4,5,6
Abstract Introduction: The purpose of this study was to report our clinical experience in the surgical management of patients with posterior glottic diastasis (PGD) secondary to prolonged intubation and/or laryngotracheoplasty (LTP) during childhood. Methods: We reviewed the charts of patients with a history of prolonged intubation and/or LTP who had undergone surgical correction for PGD at our institution between 2010 and 2014. We documented demographic data and pertinent information regarding medical and surgical histories. The Pediatric Voice Handicap Index (pVHI) and/or the Consensus Auditory-Perceptual Evaluation of Voice (CAPE-V) were used to assess patients both before and after undergoing treatment for voice disorders. Results: Six patients met our inclusion criteria. With 1 exception, all patients with complete voice data demonstrated improvements in perceptual, patient-reported, and acoustic voice measures. There were no perioperative complications. Conclusion: Our case series demonstrates that operative intervention can lead to improved voice in carefully selected patients with PGD secondary to prolonged intubation and/or LTP during childhood. Patients exhibited postoperative improvement in loudness and vocal endurance; however, they also exhibited a degree of compromise in voice quality. Keywords laryngotracheoplasty, posterior glottic diastasis, dysphonia, pediatric, voice disorders
Introduction A number of studies over the past several decades have shown that despite the lifesaving benefits of both laryngotracheoplasty (LTP) and prolonged intubation, many children who undergo either of these interventions are left with dysphonia.1-4 Although voice issues have long been an accepted iatrogenic consequence of these interventions, clinicians as well as caregivers have become increasingly sensitive to the long-term psychosocial effect of dysphonia.1 Of necessity, the initial priority for patients with airway obstruction during infancy or early childhood is to establish a patent airway. However, the ability to phonate adequately becomes increasingly important with age, particularly during the teenage years. During adolescence and early adulthood, voice becomes an integral component of social interaction, with abnormalities often affecting career decisions. Published reports from our own clinical practice and from other institutions indicate that children who have undergone LTP early in life or have incurred laryngeal injury resulting from prolonged intubation are now presenting during adolescence with the primary complaint of dysphonia.1-6
Advancements in the aerodynamic and acoustic assessment of the pediatric voice have led to the identification and characterization of specific voice abnormalities5-8 and a more in-depth understanding of iatrogenic dysphonia. Zeitels et al9 described persistent posterior glottic incompetence in adults with a history of laryngeal injury resulting 1
Division of Pediatric Otolaryngology–Head and Neck Surgery, Cincinnati Children’s Hospital Medical Center, Cincinnati, Ohio, USA 2 Division of Speech-Language Pathology, Cincinnati Children’s Hospital Medical Center, Cincinnati, Ohio, USA 3 Communication Sciences Research Center, Cincinnati Children’s Hospital Medical Center, Cincinnati, Ohio, USA 4 Centers for Pediatric Voice Disorders, Cincinnati Children’s Hospital Medical Center, Cincinnati, Ohio, USA 5 Aerodigestive and Esophageal Center, Cincinnati Children’s Hospital Medical Center, Cincinnati, Ohio, USA 6 Department of Otolaryngology–Head and Neck Surgery, University of Cincinnati College of Medicine, Cincinnati, Ohio, USA Corresponding Author: Alessandro de Alarcón, MD, MPH, Division of Pediatric Otolaryngology– Head and Neck Surgery, Cincinnati Children’s Hospital Medical Center, 3333 Burnet Avenue, MLC 2018, Cincinnati, OH 45229-3039, USA. Email: [email protected]
Downloaded from aor.sagepub.com at UNIV OF GEORGIA LIBRARIES on May 28, 2015
73
Sidell et al from intubation. These authors referred to this phenomenon as posterior glottic diastasis (PGD) and suggested its relevance in patients with a history of airway expansion procedures. Posterior glottic diastasis would be especially relevant in pediatric airway patients, who frequently undergo prolonged intubation and/or posterior grafts for airway expansion. To surgically correct PGD, Zeitels and his colleagues developed 2 operative procedures: the open partial posterior cricoid resection and an endoscopic advancement-rotation flap with interarytenoid interposition. The purpose of our study was to report our clinical experience applying these operative procedures to the management of patients with PGD secondary to prolonged intubation and/or LTP during childhood. To date, these operations have not been evaluated in this population.
Materials and Methods We reviewed the medical records of patients who had a history of LTP and/or prolonged intubation during childhood and who had undergone surgical correction for PGD between January 2010 and January 2014 at our institution. We obtained demographic data, including race, sex, and age at the time of surgery, and pertinent information regarding medical and surgical histories. All patients underwent a comprehensive voice evaluation preoperatively. They were asked to return for an additional evaluation postoperatively and a third evaluation following a course of postoperative voice therapy.
Comprehensive Voice Evaluation The Pediatric Voice Handicap Index (pVHI) and/or the Consensus Auditory-Perceptual Evaluation of Voice (CAPE-V) were used to assess patients both before and after undergoing treatment for voice disorders. These tests were administered using previously described protocols for a full voice evaluation.10 Acoustic data were obtained in a sound treated booth using the Computerized Speech Lab with Real Time Pitch software (model 4500; KayPENTAX, Lincoln Park, New Jersey, USA). A Shure SM48 unidirectional dynamic microphone was used with a constant microphone-to-mouth distance of 5 cm maintained in an off-axis position. A Quest 201 sound level meter positioned 15 cm from the patient’s mouth was used to collect intensity data. Phonatory tasks were performed 3 times and averaged to obtain maximum phonation times (MPTs) and intensity data. The vowel “ah,” sustained at a comfortable pitch, was collected and inspected visually to determine signal type using the classification system described by Titze.11 Each patient also underwent a transoral and/or transnasal digital stroboscopic examination. The decision to perform a
transnasal flexible endoscopy was based on the consensus of the voice team when considering the technical needs of the examination, the ability of the patient to cooperate, and the specific nasal anatomy of each patient. Flexible endoscopy was performed after application of oxymetazoline hydrochloride and tetracaine hydrochloride to the nasal passages. This study was approved by the institutional review board at Cincinnati Children’s Hospital Medical Center.
Diagnosis and Corrective Surgical Procedures Posterior glottic diastasis was suspected in patients with a history of airway expansion or prolonged intubation who presented with breathy dysphonia. Rigid endoscopy was performed to confirm the presence of a broad posterior cricoid plate and interarytenoid space contributing to a persistent posterior keyhole aperture. After confirming a diagnosis of PGD, patients in this study were managed using 1 of the 2 procedures described below. The decision to perform an arytenoid/aryepiglottic flap or posterior cricoid reduction was based on vocal fold mobility and the maximum crosssectional area of the larynx on stroboscopy. Patients with a narrow glottic aperture (vocal fold immobility) were managed with an arytenoid/aryepiglottic flap.
Endoscopic Posterior Cricoid Reduction A single dose of perioperative antibiotics was given. Laryngoscopy and bronchoscopy were performed and the airway was sized to confirm adequacy of the airway lumen prior to reduction. With the patient ventilating spontaneously under general anesthesia, a Zeitels laryngoscope was inserted into the laryngeal vestibule, splaying the false vocal folds and affording a view of the posterior cricoid plate (Figure 1). The laryngoscope was suspended and an operating microscope was brought into the field. The posterior cricoid plate was injected with 1% lidocaine with 1:100 000 parts epinephrine. A Blitzer knife and/or D-knife were used to excise an appropriate width of the posterior cricoid plate. In selected patients, a C02 laser was used to excise the posterior cricoid strip. The cricoid was then reapproximated using a No. 4-0 polydioxanone suture in an interrupted fashion (Figure 2). Complete closure was generally performed with 2 sutures. A vocal fold spreader was often necessary to visualize the proximal aspect of the posterior cricoid plate. The airway was again sized following reduction. The patient was then allowed to awaken. Patients were observed in the specialty airway unit or pediatric intensive care unit (PICU) postoperatively. A proton pump inhibitor was given for 1 month postoperatively.
Downloaded from aor.sagepub.com at UNIV OF GEORGIA LIBRARIES on May 28, 2015
74
Annals of Otology, Rhinology & Laryngology 124(1)
Figure 1. A broad posterior cricoid plate is visible during suspension laryngoscopy.
Figure 3. Direct laryngoscopy, 1 week following posterior cricoid reduction.
Figure 2. Following the removal of a central strip of cartilage, the posterior cricoid is reapproximated using a No. 4-0 polydioxanone suture.
Figure 4. The right arytenoid complex is grasped and pulled medially, while curved scissors are used to create an inferiorly based flap composed of a portion of the lateral epiglottic cartilage and aryepiglottic fold.
A soft diet was initiated after a bedside swallow study was performed on postoperative day 1. Voice rest was recommended for 1 week, after which repeat endoscopy was performed (Figure 3).
Aryepiglottic/Arytenoid Flap A single dose of perioperative antibiotics was given. Laryngoscopy and bronchoscopy were performed and the airway was sized to confirm adequacy of the airway lumen prior to reduction. With the patient ventilating spontaneously under general anesthesia, a Zeitels laryngoscope was placed and the larynx was suspended.9 An operating microscope was then brought into the field and a potassium titanyl phosphate (KTP) laser was used to ablate the mucosa
of the posterior glottis at the intended flap recipient site. The aryepiglottic fold and arytenoid were then injected with 1% lidocaine with 1:100 000 epinephrine. Microlaryngeal forceps were used to provide counter tension while curved scissors were used to create an inferiorly based flap composed of a portion of the lateral epiglottic cartilage and aryepiglottic fold (Figure 4). A back-cut was then created on the posterior face of the ipsilateral arytenoid, allowing the bulk of the arytenoid apex, the cuneiform and corniculate cartilage, and the aryepiglottic flap to be rotated into the posterior glottis at the site of diastasis (Figure 5). After achieving hemostasis at the harvest site using the KTP laser, the flap was secured using a No. 4-0 polydioxanone suture in an interrupted fashion. The first suture was placed to
Downloaded from aor.sagepub.com at UNIV OF GEORGIA LIBRARIES on May 28, 2015
75
Sidell et al
performed on postoperative day 1. Voice rest was recommended for 1 week, after which repeat endoscopy was performed.
Case Descriptions Patient 1
Figure 5. A back-cut created on the posterior face of the ipsilateral arytenoid allows the bulk of the arytenoid complex to be rotated into the posterior glottis at the site of diastasis.
Patient 1 was a 10-year-old female born at 24 weeks’ gestation. She was intubated for 1 month during infancy but had not undergone airway procedures prior to presentation. Evaluation in the pediatric voice clinic demonstrated mounding in the interarytenoid space and incomplete approximation of the arytenoids and vocal folds. During phonation, severe lateral supraglottic compression was noted. Perceptually, the voice was breathy and low in pitch. Preoperative microlaryngoscopy demonstrated a large posterior glottic gap with mature scar tissue surrounding a broad, flattened posterior cricoid plate. The patient’s airway was sized with a 6.5 endotracheal tube with a free leak. An endoscopic posterior cricoid reduction was performed and approximately 2.5 mm of cartilage was removed from the central cricoid. At the conclusion of the operation, the patient was sized with a 6.0 endotracheal tube. She was observed in the specialty airway unit and discharged on postoperative day 2, without symptoms of respiratory distress and tolerating a soft diet.
Patient 2
Figure 6. A magnified view of the right arytenoid complex spanning the posterior glottis. No. 4-0 polydioxanone suture is used to reapproximate the mucosa of the lateral flap base with the mucosal defect in the aryepiglottic fold.
inset the distal end of the flap into the defect, allowing the cartilaginous component of the flap to approximate the raw mucosal surface previously denuded by the laser. Additional sutures were placed to advance additional flap bulk into the defect as needed and to reapproximate the mucosa of the lateral flap base with the mucosal defect in the aryepiglottic fold (Figure 6). If the patient was unable to maintain spontaneous ventilation, a laser-safe endotracheal tube was placed during the procedure. The airway was again sized following flap placement. Patients were monitored in the specialty airway unit or the PICU until discharge. A proton pump inhibitor was given for 1 month postoperatively. A soft diet was initiated after a bedside swallow study was
Patient 2 was an 18-year-old male born at 26 weeks’ gestation. He underwent prolonged intubation during infancy. He had a history of laryngofissure and double-staged LTP with anterior and posterior costal cartilage grafting in the first year of life. He was severely dysphonic upon presentation, and microlaryngoscopy and bronchoscopy demonstrated vocal fold asymmetry and a poorly aligned anterior commissure. At 17 years of age, he underwent revision laryngofissure with anterior/posterior cricoid split to realign the vocal folds. Subsequent evaluation in the pediatric voice clinic demonstrated improved vocal volume and strength; however, the patient continued to require disability services for breathy dysphonia. Stroboscopy demonstrated a moderately narrow glottic aperture but a large, persistent posterior gap during phonation. An endoscopic left arytenoid flap was subsequently performed. The patient was observed overnight in the PICU and transferred to the specialty airway unit on postoperative day 1. He was discharged on postoperative day 2, without respiratory symptoms and tolerating a regular diet.
Patient 3 Patient 3 was a 19-year-old female born at 26 weeks’ gestation. She underwent prolonged intubation during infancy,
Downloaded from aor.sagepub.com at UNIV OF GEORGIA LIBRARIES on May 28, 2015
76
Annals of Otology, Rhinology & Laryngology 124(1)
resulting in subglottic stenosis (SGS). She had a history of true vocal fold paralysis, LTP with posterior costal cartilage grafting, and bilateral fat injection of the vocal folds. Evaluation in the pediatric voice clinic demonstrated irregularity of the free edge of the vocal fold, a posterior glottic chink, and poor abduction of the vocal folds. Perceptually, she had severe dysphonia characterized by roughness and strain. Preoperative microlaryngoscopy and bronchoscopy demonstrated a large posterior glottic gap with only moderately abducted vocal folds. She subsequently underwent a right endoscopic aryepiglottic rotational advancement flap. The flap included a small portion of the right epiglottic cartilage, cuneiform and corniculate cartilages, and overlying mucosa. The patient was observed in the specialized airway unit and discharged on postoperative day 2, without airway symptoms and tolerating a regular diet.
Patient 4 Patient 4 was a 22-year-old female with a history of neonatal intubation and SGS. She presented at our institution after undergoing multiple prior laryngeal and tracheal operations. She subsequently underwent tracheal resection and cervical slide tracheoplasty for persistent symptomatic tracheal stenosis. After management of her tracheal airway, she received voice therapy for breathy dysphonia. Microlaryngoscopy and bronchoscopy demonstrated a large posterior glottic trough contributing to incomplete glottic closure. Her airway was sized with a 6.5 endotracheal tube with a free leak. An endoscopic posterior cricoid reduction was performed using a CO2 laser, and a 3-mm strip of the posterior cricoid plate was removed. The patient was observed in the specialized airway unit and discharged on postoperative day 3, without airway symptoms and tolerating a soft diet.
Patient 5 Patient 5 was an 18-year-old female born at 27 weeks’ gestation. She underwent prolonged intubation during infancy and subsequently presented with SGS. She underwent LTP with anterior costal cartilage grafting at 5 years of age. She presented to the pediatric voice clinic for evaluation of her longstanding dysphonia. Examination findings included posterior gap glottic closure that was difficult to visualize due to mixed supraglottic compression. Perceptually, her voice exhibited moderate breathy dysphonia with a soft volume and high pitch. Preoperative microlaryngoscopy demonstrated a broad posterior cricoid plate and widely patent subglottis and trachea. Her airway was sized with a 7.0 endotracheal tube with a free leak. An endoscopic posterior cricoid reduction was performed, and approximately 2.5 to 3 mm of cartilage was removed from the central cricoid. At the conclusion of
the operation, the airway was sized with a 6.5 endotracheal tube. The patient was observed postoperatively in the specialized airway unit and discharged on postoperative day 2, without airway symptoms and tolerating a soft diet.
Patient 6 Patient 6 was a 7-year-old male who was born at 35 weeks’ gestation with complete tracheal rings. He presented with a history of tracheal homograft and pericardial patch that was performed at 8 months of age, followed by an additional double-stage LTP. He then developed restenosis and required tracheostomy placement. Upon presentation, a thoracic slide tracheoplasty on cardiopulmonary bypass was performed to address his distal tracheal stenosis. This was followed by a double-stage LTP to re-establish his subglottic airway. In addition, a cervical and thoracic slide tracheoplasty via a transcervical approach was performed to address his proximal tracheal stenosis. During this time, the patient was evaluated in the voice therapy clinic, where he was noted to be aphonic. He communicated primarily by means of mouthing, tongue clicks, and gestures. His vocal folds were in a fixed abducted position and his subglottic airway was widely patent posteriorly. At the time of slide tracheoplasty, an open posterior cricoid reduction was performed to remove a segment of the previously placed posterior costal cartilage graft. Voice therapy following the procedure demonstrated significant improvement from his preoperative state, and supraglottic voicing was achieved.
Summary of Results Six patients received surgical management for PGD; 3 received an endoscopic posterior cricoid reduction, 1 received an open posterior cricoid reduction, and 2 received an arytenoid/aryepiglottic fold flap. Five patients had previously undergone LTP for SGS, and 1 (patient 1) had a history of prolonged intubation but had not undergone previous airway reconstruction. Patients 1 through 5 tolerated their operative procedure well, with a maximum hospital stay of 3 days postoperatively. Patient 6 underwent slide tracheoplasty at the time of definitive voice surgery and was discharged after 1 week. All patients were discharged without airway symptoms and tolerating an oral diet. There were no perioperative complications. Due to geographic and financial constraints, not all patients were able to undergo both postoperative voice analyses. Because patient 6 was aphonic preoperatively, only 5 patients had usable preoperative voice data for review. Of these, 4 returned for postoperative voice analysis (patients 1-4), and 3 returned for additional analysis following voice therapy (patients 1-3) (Tables 1 and 2).
Downloaded from aor.sagepub.com at UNIV OF GEORGIA LIBRARIES on May 28, 2015
77
Sidell et al Table 1. Preoperative and Postoperative Perceptual Voice Data. Patient
Operative Intervention
CAPE-V (Preop)
PCR AF AF PCR PCR PCR
43 53 74 58 30 Aphonic
1 2 3 4 5 6
CAPE-V (Postop)
CAPE-V (After Postop ST)
33 80 62 43
22 65 62
pVHI (Preop)
pVHI (Postop)
40 82 73 70 66 Aphonic
17 55 58 43
pVHI (After Postop ST) 4 14 17 49
Abbreviations: AF, arytenoid flap; CAPE-V, Consensus Auditory-Perceptual Evaluation of Voice; PCR, posterior cricoid reduction; Postop, postoperative; Preop, preoperative; pVHI, Pediatric Voice Handicap Index; ST, speech therapy.
Table 2. Preoperative and Postoperative Acoustic Voice Data.
Patient 1 2 3 4 5
Operative Intervention
Signal Type
MPT (Preop)
MPT (Postop)
PCR AF AF PCR PCR
I III II I II
7.95 6.33 9.26 2.7 5.2
9.7 5 16.62 4
MPT (After Postop ST) 9.3 10 18.66
Maximum Loudness (Preop), dB
Maximum Loudness (Postop), dB
Maximum Loudness (After Postop ST), dB
72.97 70.67 79.03 92.13 72.4
80.5 71.67 84.51 93.3
87.83 72.2 89.63
Abbreviations: AF, arytenoid flap; dB, decibels; MPT, maximum phonation time; PCR, posterior cricoid reduction; Postop, postoperative; Preop, preoperative; ST, speech therapy.
Of the 3 patients with complete voice data, 2 (patients 1 and 3) had improvements in their CAPE-V scores following surgery and voice therapy. Patient 4 also demonstrated improvement in her CAPE-V score postoperatively but did not return for voice evaluation following voice therapy. All patients had a reduction in postoperative pVHI scores that further improved following voice therapy. Patient 6, previously aphonic despite voice therapy, achieved a pVHI score of 49 following operative intervention and voice therapy (Table 1). Five patients had acoustic data available for review preoperatively. The majority of patients were classified as having a Titze11 type II or III signal. Three of 4 patients were able to improve MPT following surgery. All patients with available voice data had improvements in MPT and maximum loudness following surgery and voice therapy when compared to preoperative values (Table 2).
Discussion These case presentations are the first to describe the operative management of PGD secondary to LTP and/or prolonged intubation during childhood. Our findings revealed improvements in perceptual (CAPE-V), patient-reported (pVHI), and acoustic voice measures (MPT, signal type, loudness) in 3 of 4 children with voice data obtained from the initial postoperative evaluation; further improvements
were noted following a course of voice therapy. These 3 patients demonstrated improvement in overall CAPE-V outcomes, including subscores reflecting loudness, breathiness, pitch, and strain; however, an increase in roughness was also recognized. Given that the intent of reducing the posterior glottic aperture is to increase pressure generation during voicing, these findings are not surprising. The increase in pressure leads to improvements in breathiness and loudness, with the result that other preexisting voice abnormalities such as roughness may be more audible postoperatively. Alternatively, roughness may be a result of the surgical intervention itself in patients who have undergone arytenoid flap surgery. Regardless of this negative effect, specific components of the perceptual voice analysis such as increased volume and reduced strain are likely to produce an overall improvement in voice. Acoustic analysis demonstrated dramatic improvements in MPTs and voice volume for all patients with available postoperative data. The changes in perceptual voice outcomes following surgery may also reflect the compromise made in vocal quality to improve laryngeal efficiency (MPT) and increase maximum loudness (dB) (Table 2). We believe that these changes culminated in overall improvement in the fluidity and intelligibility of speech, as demonstrated by the improvement in overall patient-reported voice outcomes as measured by the pVHI following surgery and voice therapy.
Downloaded from aor.sagepub.com at UNIV OF GEORGIA LIBRARIES on May 28, 2015
78
Annals of Otology, Rhinology & Laryngology 124(1)
Although the small number of patients in this study does not permit meaningful statistical analysis, we found that postoperative improvement in overall pVHI scores was greater than changes in CAPE-V scores, suggesting that patients perceived a greater improvement in voice postoperatively than did the SLP. This finding may also reflect the limitations of the CAPE-V system in accurately assessing the overall intelligibility of voice as perceived by the patient population. An exception to the improvement seen in overall CAPE-V scores following surgery was observed in patient 2. Although the overall scores for this patient did not return to the preoperative baseline, some improvement in voice quality was noted following postoperative voice therapy. This result likely reflects the patient’s transition to supraglottic phonation, something that became possible only following surgery but would be expected to negatively influence certain voice attributes based on CAPE-V scoring. Despite a decline in CAPE-V scores following surgery, pVHI scores improved dramatically for this patient postoperatively and improved further following voice therapy. Furthermore, acoustic measures for this patient demonstrated a doubling of the MPT and increased sound intensity following surgery and voice therapy. This serves to emphasize the importance of a voice therapy regimen that is tailored specifically to the postsurgical changes of each individual’s airway anatomy. The primary factors influencing procedure selection were vocal fold mobility and maximum glottic aperture as seen on stroboscopy. Patients 2 and 3 demonstrated limited abduction of the vocal folds, raising concern for possible airway obstruction after posterior cricoid reduction. Targeted narrowing was instead created by means of an arytenoid flap, allowing improved posterior glottic competence while minimizing reduction of the overall cross-sectional area of the airway. Under ideal circumstances, all patients with PGD would undergo posterior cricoid reduction, which better restores the normal proportional relationships between the arytenoids and the cricoid that are disrupted by posterior expansion laryngotracheoplasty. In contrast, arytenoid flap reconstruction may improve glottic competence in the setting of PGD but may not allow uniform distribution of subglottic pressure along the free margin of the vocal fold during phonation. In patients 2 and 3, arytenoid flaps offered improved voice characteristics while maintaining adequate airway patency. Regardless of the specific operative intervention used to improve PGD, appropriate patient selection and careful clinical evaluation are of utmost importance. Whereas many children with PGD have undergone previous extensive medical and surgical interventions to increase the caliber of the airway, including posterior expansion procedures, the operative procedures described in this case series selectively narrowed a portion of the airway. Parental or patient reluctance to do this must be addressed by thorough explanations and good counseling. This point cannot be overemphasized.
Conclusion Our case series of 6 patients suggests that operative intervention will lead to improved voice, without compromising airway patency, in carefully selected pediatric patients with PGD secondary to prolonged intubation and/or LTP during childhood. Patients exhibited postoperative improvement in loudness and vocal endurance; however, they also exhibited an acceptable degree of compromise in voice quality. Declaration of Conflicting Interests The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding The author(s) received no financial support for the research, authorship, and/or publication of this article.
References 1. de Alarcón A. Voice outcomes after pediatric airway reconstruction. Laryngoscope. 2012;122:S84-S86. 2. Zalzal GH. Rib cartilage grafts for the treatment of posterior glottic and subglottic stenosis in children. Ann Otol Rhinol Laryngol. 1988;97:506-511. 3. Maddalozzo J, Holinger LB. Laryngotracheal reconstruction for subglottic stenosis in children. Ann Otol Rhinol Laryngol. 1987;96:665-669. 4. Smith ME, Marsh JH, Cotton RT, Myer CM III. Voice problems after pediatric laryngotracheal reconstruction: videolaryngostroboscopic, acoustic and perceptual assessment. Int J Pediatr Otorhinolaryngol. 1993;25:173-181. 5. Vijayasekaran S, Sances R, Cotton R, Elluru RG. Changes in the cricoarytenoid joint induced by intubation in neonates. Arch Otolaryngol Head Neck Surg. 2006;132:1342-1345. 6. Kelchner LN, Weinrich B, Brehm SB, Tabangin ME, de Alarcón A. Characterization of supraglottic phonation in children after airway reconstruction. Ann Otol Rhinol Laryngol. 2010;119(6):383-390. 7. Arviso LC, Klein AM, Johns MM III. The management of postintubation phonatory insufficiency. J Voice. 2012;26(4):530533. 8. Brehm SB, Weinrich B, Zieser M, et al. Aerodynamic and acoustic assessment in children following airway reconstruction: an assessment of feasibility. Int J Ped Otorhinolaryngol. 2009;73:1019-1023. 9. Zeitels SM, de Alarcón A, Burns JA, Lopez-Guerra G, Hillman RE. Posterior glottis diastasis: mechanically deceptive and often overlooked. Ann Otol Rhinol Laryngol. 2011;120(2):71-80. 10. Zur KB, Cotton S, Kelchner L, Baker S, Weinrich B, Lee L. Pediatric Voice Handicap Index (pVHI): a new tool for evaluating pediatric dysphonia. Int J Ped Otorhinolaryngol. 2007;71(1):77-82. 11. Titze I. Workshop on Acoustic Voice Analysis: Summary Statement. Iowa City, IA: National Center for Voice and Speech; 1995.
Downloaded from aor.sagepub.com at UNIV OF GEORGIA LIBRARIES on May 28, 2015
research-article2014
AORXXX10.1177/0003489414543100Annals of Otology, Rhinology & LaryngologySidell et al
Article
Surgical Management of Posterior Glottic Diastasis in Children
Annals of Otology, Rhinology & Laryngology 2015, Vol. 124(1) 72–78 © The Author(s) 2014 Reprints and permissions: sagepub.com/journalsPermissions.nav DOI: 10.1177/0003489414543100 aor.sagepub.com
Douglas R. Sidell, MD1, Stephanie Zacharias, PhD, CCC-SLP2,3,4, Karthik Balakrishnan1, Michael J. Rutter, MBChB, FRACS1,5,6, and Alessandro de Alarcón, MD, MPH1,3,4,5,6
Abstract Introduction: The purpose of this study was to report our clinical experience in the surgical management of patients with posterior glottic diastasis (PGD) secondary to prolonged intubation and/or laryngotracheoplasty (LTP) during childhood. Methods: We reviewed the charts of patients with a history of prolonged intubation and/or LTP who had undergone surgical correction for PGD at our institution between 2010 and 2014. We documented demographic data and pertinent information regarding medical and surgical histories. The Pediatric Voice Handicap Index (pVHI) and/or the Consensus Auditory-Perceptual Evaluation of Voice (CAPE-V) were used to assess patients both before and after undergoing treatment for voice disorders. Results: Six patients met our inclusion criteria. With 1 exception, all patients with complete voice data demonstrated improvements in perceptual, patient-reported, and acoustic voice measures. There were no perioperative complications. Conclusion: Our case series demonstrates that operative intervention can lead to improved voice in carefully selected patients with PGD secondary to prolonged intubation and/or LTP during childhood. Patients exhibited postoperative improvement in loudness and vocal endurance; however, they also exhibited a degree of compromise in voice quality. Keywords laryngotracheoplasty, posterior glottic diastasis, dysphonia, pediatric, voice disorders
Introduction A number of studies over the past several decades have shown that despite the lifesaving benefits of both laryngotracheoplasty (LTP) and prolonged intubation, many children who undergo either of these interventions are left with dysphonia.1-4 Although voice issues have long been an accepted iatrogenic consequence of these interventions, clinicians as well as caregivers have become increasingly sensitive to the long-term psychosocial effect of dysphonia.1 Of necessity, the initial priority for patients with airway obstruction during infancy or early childhood is to establish a patent airway. However, the ability to phonate adequately becomes increasingly important with age, particularly during the teenage years. During adolescence and early adulthood, voice becomes an integral component of social interaction, with abnormalities often affecting career decisions. Published reports from our own clinical practice and from other institutions indicate that children who have undergone LTP early in life or have incurred laryngeal injury resulting from prolonged intubation are now presenting during adolescence with the primary complaint of dysphonia.1-6
Advancements in the aerodynamic and acoustic assessment of the pediatric voice have led to the identification and characterization of specific voice abnormalities5-8 and a more in-depth understanding of iatrogenic dysphonia. Zeitels et al9 described persistent posterior glottic incompetence in adults with a history of laryngeal injury resulting 1
Division of Pediatric Otolaryngology–Head and Neck Surgery, Cincinnati Children’s Hospital Medical Center, Cincinnati, Ohio, USA 2 Division of Speech-Language Pathology, Cincinnati Children’s Hospital Medical Center, Cincinnati, Ohio, USA 3 Communication Sciences Research Center, Cincinnati Children’s Hospital Medical Center, Cincinnati, Ohio, USA 4 Centers for Pediatric Voice Disorders, Cincinnati Children’s Hospital Medical Center, Cincinnati, Ohio, USA 5 Aerodigestive and Esophageal Center, Cincinnati Children’s Hospital Medical Center, Cincinnati, Ohio, USA 6 Department of Otolaryngology–Head and Neck Surgery, University of Cincinnati College of Medicine, Cincinnati, Ohio, USA Corresponding Author: Alessandro de Alarcón, MD, MPH, Division of Pediatric Otolaryngology– Head and Neck Surgery, Cincinnati Children’s Hospital Medical Center, 3333 Burnet Avenue, MLC 2018, Cincinnati, OH 45229-3039, USA. Email: [email protected]
Downloaded from aor.sagepub.com at UNIV OF GEORGIA LIBRARIES on May 28, 2015
73
Sidell et al from intubation. These authors referred to this phenomenon as posterior glottic diastasis (PGD) and suggested its relevance in patients with a history of airway expansion procedures. Posterior glottic diastasis would be especially relevant in pediatric airway patients, who frequently undergo prolonged intubation and/or posterior grafts for airway expansion. To surgically correct PGD, Zeitels and his colleagues developed 2 operative procedures: the open partial posterior cricoid resection and an endoscopic advancement-rotation flap with interarytenoid interposition. The purpose of our study was to report our clinical experience applying these operative procedures to the management of patients with PGD secondary to prolonged intubation and/or LTP during childhood. To date, these operations have not been evaluated in this population.
Materials and Methods We reviewed the medical records of patients who had a history of LTP and/or prolonged intubation during childhood and who had undergone surgical correction for PGD between January 2010 and January 2014 at our institution. We obtained demographic data, including race, sex, and age at the time of surgery, and pertinent information regarding medical and surgical histories. All patients underwent a comprehensive voice evaluation preoperatively. They were asked to return for an additional evaluation postoperatively and a third evaluation following a course of postoperative voice therapy.
Comprehensive Voice Evaluation The Pediatric Voice Handicap Index (pVHI) and/or the Consensus Auditory-Perceptual Evaluation of Voice (CAPE-V) were used to assess patients both before and after undergoing treatment for voice disorders. These tests were administered using previously described protocols for a full voice evaluation.10 Acoustic data were obtained in a sound treated booth using the Computerized Speech Lab with Real Time Pitch software (model 4500; KayPENTAX, Lincoln Park, New Jersey, USA). A Shure SM48 unidirectional dynamic microphone was used with a constant microphone-to-mouth distance of 5 cm maintained in an off-axis position. A Quest 201 sound level meter positioned 15 cm from the patient’s mouth was used to collect intensity data. Phonatory tasks were performed 3 times and averaged to obtain maximum phonation times (MPTs) and intensity data. The vowel “ah,” sustained at a comfortable pitch, was collected and inspected visually to determine signal type using the classification system described by Titze.11 Each patient also underwent a transoral and/or transnasal digital stroboscopic examination. The decision to perform a
transnasal flexible endoscopy was based on the consensus of the voice team when considering the technical needs of the examination, the ability of the patient to cooperate, and the specific nasal anatomy of each patient. Flexible endoscopy was performed after application of oxymetazoline hydrochloride and tetracaine hydrochloride to the nasal passages. This study was approved by the institutional review board at Cincinnati Children’s Hospital Medical Center.
Diagnosis and Corrective Surgical Procedures Posterior glottic diastasis was suspected in patients with a history of airway expansion or prolonged intubation who presented with breathy dysphonia. Rigid endoscopy was performed to confirm the presence of a broad posterior cricoid plate and interarytenoid space contributing to a persistent posterior keyhole aperture. After confirming a diagnosis of PGD, patients in this study were managed using 1 of the 2 procedures described below. The decision to perform an arytenoid/aryepiglottic flap or posterior cricoid reduction was based on vocal fold mobility and the maximum crosssectional area of the larynx on stroboscopy. Patients with a narrow glottic aperture (vocal fold immobility) were managed with an arytenoid/aryepiglottic flap.
Endoscopic Posterior Cricoid Reduction A single dose of perioperative antibiotics was given. Laryngoscopy and bronchoscopy were performed and the airway was sized to confirm adequacy of the airway lumen prior to reduction. With the patient ventilating spontaneously under general anesthesia, a Zeitels laryngoscope was inserted into the laryngeal vestibule, splaying the false vocal folds and affording a view of the posterior cricoid plate (Figure 1). The laryngoscope was suspended and an operating microscope was brought into the field. The posterior cricoid plate was injected with 1% lidocaine with 1:100 000 parts epinephrine. A Blitzer knife and/or D-knife were used to excise an appropriate width of the posterior cricoid plate. In selected patients, a C02 laser was used to excise the posterior cricoid strip. The cricoid was then reapproximated using a No. 4-0 polydioxanone suture in an interrupted fashion (Figure 2). Complete closure was generally performed with 2 sutures. A vocal fold spreader was often necessary to visualize the proximal aspect of the posterior cricoid plate. The airway was again sized following reduction. The patient was then allowed to awaken. Patients were observed in the specialty airway unit or pediatric intensive care unit (PICU) postoperatively. A proton pump inhibitor was given for 1 month postoperatively.
Downloaded from aor.sagepub.com at UNIV OF GEORGIA LIBRARIES on May 28, 2015
74
Annals of Otology, Rhinology & Laryngology 124(1)
Figure 1. A broad posterior cricoid plate is visible during suspension laryngoscopy.
Figure 3. Direct laryngoscopy, 1 week following posterior cricoid reduction.
Figure 2. Following the removal of a central strip of cartilage, the posterior cricoid is reapproximated using a No. 4-0 polydioxanone suture.
Figure 4. The right arytenoid complex is grasped and pulled medially, while curved scissors are used to create an inferiorly based flap composed of a portion of the lateral epiglottic cartilage and aryepiglottic fold.
A soft diet was initiated after a bedside swallow study was performed on postoperative day 1. Voice rest was recommended for 1 week, after which repeat endoscopy was performed (Figure 3).
Aryepiglottic/Arytenoid Flap A single dose of perioperative antibiotics was given. Laryngoscopy and bronchoscopy were performed and the airway was sized to confirm adequacy of the airway lumen prior to reduction. With the patient ventilating spontaneously under general anesthesia, a Zeitels laryngoscope was placed and the larynx was suspended.9 An operating microscope was then brought into the field and a potassium titanyl phosphate (KTP) laser was used to ablate the mucosa
of the posterior glottis at the intended flap recipient site. The aryepiglottic fold and arytenoid were then injected with 1% lidocaine with 1:100 000 epinephrine. Microlaryngeal forceps were used to provide counter tension while curved scissors were used to create an inferiorly based flap composed of a portion of the lateral epiglottic cartilage and aryepiglottic fold (Figure 4). A back-cut was then created on the posterior face of the ipsilateral arytenoid, allowing the bulk of the arytenoid apex, the cuneiform and corniculate cartilage, and the aryepiglottic flap to be rotated into the posterior glottis at the site of diastasis (Figure 5). After achieving hemostasis at the harvest site using the KTP laser, the flap was secured using a No. 4-0 polydioxanone suture in an interrupted fashion. The first suture was placed to
Downloaded from aor.sagepub.com at UNIV OF GEORGIA LIBRARIES on May 28, 2015
75
Sidell et al
performed on postoperative day 1. Voice rest was recommended for 1 week, after which repeat endoscopy was performed.
Case Descriptions Patient 1
Figure 5. A back-cut created on the posterior face of the ipsilateral arytenoid allows the bulk of the arytenoid complex to be rotated into the posterior glottis at the site of diastasis.
Patient 1 was a 10-year-old female born at 24 weeks’ gestation. She was intubated for 1 month during infancy but had not undergone airway procedures prior to presentation. Evaluation in the pediatric voice clinic demonstrated mounding in the interarytenoid space and incomplete approximation of the arytenoids and vocal folds. During phonation, severe lateral supraglottic compression was noted. Perceptually, the voice was breathy and low in pitch. Preoperative microlaryngoscopy demonstrated a large posterior glottic gap with mature scar tissue surrounding a broad, flattened posterior cricoid plate. The patient’s airway was sized with a 6.5 endotracheal tube with a free leak. An endoscopic posterior cricoid reduction was performed and approximately 2.5 mm of cartilage was removed from the central cricoid. At the conclusion of the operation, the patient was sized with a 6.0 endotracheal tube. She was observed in the specialty airway unit and discharged on postoperative day 2, without symptoms of respiratory distress and tolerating a soft diet.
Patient 2
Figure 6. A magnified view of the right arytenoid complex spanning the posterior glottis. No. 4-0 polydioxanone suture is used to reapproximate the mucosa of the lateral flap base with the mucosal defect in the aryepiglottic fold.
inset the distal end of the flap into the defect, allowing the cartilaginous component of the flap to approximate the raw mucosal surface previously denuded by the laser. Additional sutures were placed to advance additional flap bulk into the defect as needed and to reapproximate the mucosa of the lateral flap base with the mucosal defect in the aryepiglottic fold (Figure 6). If the patient was unable to maintain spontaneous ventilation, a laser-safe endotracheal tube was placed during the procedure. The airway was again sized following flap placement. Patients were monitored in the specialty airway unit or the PICU until discharge. A proton pump inhibitor was given for 1 month postoperatively. A soft diet was initiated after a bedside swallow study was
Patient 2 was an 18-year-old male born at 26 weeks’ gestation. He underwent prolonged intubation during infancy. He had a history of laryngofissure and double-staged LTP with anterior and posterior costal cartilage grafting in the first year of life. He was severely dysphonic upon presentation, and microlaryngoscopy and bronchoscopy demonstrated vocal fold asymmetry and a poorly aligned anterior commissure. At 17 years of age, he underwent revision laryngofissure with anterior/posterior cricoid split to realign the vocal folds. Subsequent evaluation in the pediatric voice clinic demonstrated improved vocal volume and strength; however, the patient continued to require disability services for breathy dysphonia. Stroboscopy demonstrated a moderately narrow glottic aperture but a large, persistent posterior gap during phonation. An endoscopic left arytenoid flap was subsequently performed. The patient was observed overnight in the PICU and transferred to the specialty airway unit on postoperative day 1. He was discharged on postoperative day 2, without respiratory symptoms and tolerating a regular diet.
Patient 3 Patient 3 was a 19-year-old female born at 26 weeks’ gestation. She underwent prolonged intubation during infancy,
Downloaded from aor.sagepub.com at UNIV OF GEORGIA LIBRARIES on May 28, 2015
76
Annals of Otology, Rhinology & Laryngology 124(1)
resulting in subglottic stenosis (SGS). She had a history of true vocal fold paralysis, LTP with posterior costal cartilage grafting, and bilateral fat injection of the vocal folds. Evaluation in the pediatric voice clinic demonstrated irregularity of the free edge of the vocal fold, a posterior glottic chink, and poor abduction of the vocal folds. Perceptually, she had severe dysphonia characterized by roughness and strain. Preoperative microlaryngoscopy and bronchoscopy demonstrated a large posterior glottic gap with only moderately abducted vocal folds. She subsequently underwent a right endoscopic aryepiglottic rotational advancement flap. The flap included a small portion of the right epiglottic cartilage, cuneiform and corniculate cartilages, and overlying mucosa. The patient was observed in the specialized airway unit and discharged on postoperative day 2, without airway symptoms and tolerating a regular diet.
Patient 4 Patient 4 was a 22-year-old female with a history of neonatal intubation and SGS. She presented at our institution after undergoing multiple prior laryngeal and tracheal operations. She subsequently underwent tracheal resection and cervical slide tracheoplasty for persistent symptomatic tracheal stenosis. After management of her tracheal airway, she received voice therapy for breathy dysphonia. Microlaryngoscopy and bronchoscopy demonstrated a large posterior glottic trough contributing to incomplete glottic closure. Her airway was sized with a 6.5 endotracheal tube with a free leak. An endoscopic posterior cricoid reduction was performed using a CO2 laser, and a 3-mm strip of the posterior cricoid plate was removed. The patient was observed in the specialized airway unit and discharged on postoperative day 3, without airway symptoms and tolerating a soft diet.
Patient 5 Patient 5 was an 18-year-old female born at 27 weeks’ gestation. She underwent prolonged intubation during infancy and subsequently presented with SGS. She underwent LTP with anterior costal cartilage grafting at 5 years of age. She presented to the pediatric voice clinic for evaluation of her longstanding dysphonia. Examination findings included posterior gap glottic closure that was difficult to visualize due to mixed supraglottic compression. Perceptually, her voice exhibited moderate breathy dysphonia with a soft volume and high pitch. Preoperative microlaryngoscopy demonstrated a broad posterior cricoid plate and widely patent subglottis and trachea. Her airway was sized with a 7.0 endotracheal tube with a free leak. An endoscopic posterior cricoid reduction was performed, and approximately 2.5 to 3 mm of cartilage was removed from the central cricoid. At the conclusion of
the operation, the airway was sized with a 6.5 endotracheal tube. The patient was observed postoperatively in the specialized airway unit and discharged on postoperative day 2, without airway symptoms and tolerating a soft diet.
Patient 6 Patient 6 was a 7-year-old male who was born at 35 weeks’ gestation with complete tracheal rings. He presented with a history of tracheal homograft and pericardial patch that was performed at 8 months of age, followed by an additional double-stage LTP. He then developed restenosis and required tracheostomy placement. Upon presentation, a thoracic slide tracheoplasty on cardiopulmonary bypass was performed to address his distal tracheal stenosis. This was followed by a double-stage LTP to re-establish his subglottic airway. In addition, a cervical and thoracic slide tracheoplasty via a transcervical approach was performed to address his proximal tracheal stenosis. During this time, the patient was evaluated in the voice therapy clinic, where he was noted to be aphonic. He communicated primarily by means of mouthing, tongue clicks, and gestures. His vocal folds were in a fixed abducted position and his subglottic airway was widely patent posteriorly. At the time of slide tracheoplasty, an open posterior cricoid reduction was performed to remove a segment of the previously placed posterior costal cartilage graft. Voice therapy following the procedure demonstrated significant improvement from his preoperative state, and supraglottic voicing was achieved.
Summary of Results Six patients received surgical management for PGD; 3 received an endoscopic posterior cricoid reduction, 1 received an open posterior cricoid reduction, and 2 received an arytenoid/aryepiglottic fold flap. Five patients had previously undergone LTP for SGS, and 1 (patient 1) had a history of prolonged intubation but had not undergone previous airway reconstruction. Patients 1 through 5 tolerated their operative procedure well, with a maximum hospital stay of 3 days postoperatively. Patient 6 underwent slide tracheoplasty at the time of definitive voice surgery and was discharged after 1 week. All patients were discharged without airway symptoms and tolerating an oral diet. There were no perioperative complications. Due to geographic and financial constraints, not all patients were able to undergo both postoperative voice analyses. Because patient 6 was aphonic preoperatively, only 5 patients had usable preoperative voice data for review. Of these, 4 returned for postoperative voice analysis (patients 1-4), and 3 returned for additional analysis following voice therapy (patients 1-3) (Tables 1 and 2).
Downloaded from aor.sagepub.com at UNIV OF GEORGIA LIBRARIES on May 28, 2015
77
Sidell et al Table 1. Preoperative and Postoperative Perceptual Voice Data. Patient
Operative Intervention
CAPE-V (Preop)
PCR AF AF PCR PCR PCR
43 53 74 58 30 Aphonic
1 2 3 4 5 6
CAPE-V (Postop)
CAPE-V (After Postop ST)
33 80 62 43
22 65 62
pVHI (Preop)
pVHI (Postop)
40 82 73 70 66 Aphonic
17 55 58 43
pVHI (After Postop ST) 4 14 17 49
Abbreviations: AF, arytenoid flap; CAPE-V, Consensus Auditory-Perceptual Evaluation of Voice; PCR, posterior cricoid reduction; Postop, postoperative; Preop, preoperative; pVHI, Pediatric Voice Handicap Index; ST, speech therapy.
Table 2. Preoperative and Postoperative Acoustic Voice Data.
Patient 1 2 3 4 5
Operative Intervention
Signal Type
MPT (Preop)
MPT (Postop)
PCR AF AF PCR PCR
I III II I II
7.95 6.33 9.26 2.7 5.2
9.7 5 16.62 4
MPT (After Postop ST) 9.3 10 18.66
Maximum Loudness (Preop), dB
Maximum Loudness (Postop), dB
Maximum Loudness (After Postop ST), dB
72.97 70.67 79.03 92.13 72.4
80.5 71.67 84.51 93.3
87.83 72.2 89.63
Abbreviations: AF, arytenoid flap; dB, decibels; MPT, maximum phonation time; PCR, posterior cricoid reduction; Postop, postoperative; Preop, preoperative; ST, speech therapy.
Of the 3 patients with complete voice data, 2 (patients 1 and 3) had improvements in their CAPE-V scores following surgery and voice therapy. Patient 4 also demonstrated improvement in her CAPE-V score postoperatively but did not return for voice evaluation following voice therapy. All patients had a reduction in postoperative pVHI scores that further improved following voice therapy. Patient 6, previously aphonic despite voice therapy, achieved a pVHI score of 49 following operative intervention and voice therapy (Table 1). Five patients had acoustic data available for review preoperatively. The majority of patients were classified as having a Titze11 type II or III signal. Three of 4 patients were able to improve MPT following surgery. All patients with available voice data had improvements in MPT and maximum loudness following surgery and voice therapy when compared to preoperative values (Table 2).
Discussion These case presentations are the first to describe the operative management of PGD secondary to LTP and/or prolonged intubation during childhood. Our findings revealed improvements in perceptual (CAPE-V), patient-reported (pVHI), and acoustic voice measures (MPT, signal type, loudness) in 3 of 4 children with voice data obtained from the initial postoperative evaluation; further improvements
were noted following a course of voice therapy. These 3 patients demonstrated improvement in overall CAPE-V outcomes, including subscores reflecting loudness, breathiness, pitch, and strain; however, an increase in roughness was also recognized. Given that the intent of reducing the posterior glottic aperture is to increase pressure generation during voicing, these findings are not surprising. The increase in pressure leads to improvements in breathiness and loudness, with the result that other preexisting voice abnormalities such as roughness may be more audible postoperatively. Alternatively, roughness may be a result of the surgical intervention itself in patients who have undergone arytenoid flap surgery. Regardless of this negative effect, specific components of the perceptual voice analysis such as increased volume and reduced strain are likely to produce an overall improvement in voice. Acoustic analysis demonstrated dramatic improvements in MPTs and voice volume for all patients with available postoperative data. The changes in perceptual voice outcomes following surgery may also reflect the compromise made in vocal quality to improve laryngeal efficiency (MPT) and increase maximum loudness (dB) (Table 2). We believe that these changes culminated in overall improvement in the fluidity and intelligibility of speech, as demonstrated by the improvement in overall patient-reported voice outcomes as measured by the pVHI following surgery and voice therapy.
Downloaded from aor.sagepub.com at UNIV OF GEORGIA LIBRARIES on May 28, 2015
78
Annals of Otology, Rhinology & Laryngology 124(1)
Although the small number of patients in this study does not permit meaningful statistical analysis, we found that postoperative improvement in overall pVHI scores was greater than changes in CAPE-V scores, suggesting that patients perceived a greater improvement in voice postoperatively than did the SLP. This finding may also reflect the limitations of the CAPE-V system in accurately assessing the overall intelligibility of voice as perceived by the patient population. An exception to the improvement seen in overall CAPE-V scores following surgery was observed in patient 2. Although the overall scores for this patient did not return to the preoperative baseline, some improvement in voice quality was noted following postoperative voice therapy. This result likely reflects the patient’s transition to supraglottic phonation, something that became possible only following surgery but would be expected to negatively influence certain voice attributes based on CAPE-V scoring. Despite a decline in CAPE-V scores following surgery, pVHI scores improved dramatically for this patient postoperatively and improved further following voice therapy. Furthermore, acoustic measures for this patient demonstrated a doubling of the MPT and increased sound intensity following surgery and voice therapy. This serves to emphasize the importance of a voice therapy regimen that is tailored specifically to the postsurgical changes of each individual’s airway anatomy. The primary factors influencing procedure selection were vocal fold mobility and maximum glottic aperture as seen on stroboscopy. Patients 2 and 3 demonstrated limited abduction of the vocal folds, raising concern for possible airway obstruction after posterior cricoid reduction. Targeted narrowing was instead created by means of an arytenoid flap, allowing improved posterior glottic competence while minimizing reduction of the overall cross-sectional area of the airway. Under ideal circumstances, all patients with PGD would undergo posterior cricoid reduction, which better restores the normal proportional relationships between the arytenoids and the cricoid that are disrupted by posterior expansion laryngotracheoplasty. In contrast, arytenoid flap reconstruction may improve glottic competence in the setting of PGD but may not allow uniform distribution of subglottic pressure along the free margin of the vocal fold during phonation. In patients 2 and 3, arytenoid flaps offered improved voice characteristics while maintaining adequate airway patency. Regardless of the specific operative intervention used to improve PGD, appropriate patient selection and careful clinical evaluation are of utmost importance. Whereas many children with PGD have undergone previous extensive medical and surgical interventions to increase the caliber of the airway, including posterior expansion procedures, the operative procedures described in this case series selectively narrowed a portion of the airway. Parental or patient reluctance to do this must be addressed by thorough explanations and good counseling. This point cannot be overemphasized.
Conclusion Our case series of 6 patients suggests that operative intervention will lead to improved voice, without compromising airway patency, in carefully selected pediatric patients with PGD secondary to prolonged intubation and/or LTP during childhood. Patients exhibited postoperative improvement in loudness and vocal endurance; however, they also exhibited an acceptable degree of compromise in voice quality. Declaration of Conflicting Interests The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding The author(s) received no financial support for the research, authorship, and/or publication of this article.
References 1. de Alarcón A. Voice outcomes after pediatric airway reconstruction. Laryngoscope. 2012;122:S84-S86. 2. Zalzal GH. Rib cartilage grafts for the treatment of posterior glottic and subglottic stenosis in children. Ann Otol Rhinol Laryngol. 1988;97:506-511. 3. Maddalozzo J, Holinger LB. Laryngotracheal reconstruction for subglottic stenosis in children. Ann Otol Rhinol Laryngol. 1987;96:665-669. 4. Smith ME, Marsh JH, Cotton RT, Myer CM III. Voice problems after pediatric laryngotracheal reconstruction: videolaryngostroboscopic, acoustic and perceptual assessment. Int J Pediatr Otorhinolaryngol. 1993;25:173-181. 5. Vijayasekaran S, Sances R, Cotton R, Elluru RG. Changes in the cricoarytenoid joint induced by intubation in neonates. Arch Otolaryngol Head Neck Surg. 2006;132:1342-1345. 6. Kelchner LN, Weinrich B, Brehm SB, Tabangin ME, de Alarcón A. Characterization of supraglottic phonation in children after airway reconstruction. Ann Otol Rhinol Laryngol. 2010;119(6):383-390. 7. Arviso LC, Klein AM, Johns MM III. The management of postintubation phonatory insufficiency. J Voice. 2012;26(4):530533. 8. Brehm SB, Weinrich B, Zieser M, et al. Aerodynamic and acoustic assessment in children following airway reconstruction: an assessment of feasibility. Int J Ped Otorhinolaryngol. 2009;73:1019-1023. 9. Zeitels SM, de Alarcón A, Burns JA, Lopez-Guerra G, Hillman RE. Posterior glottis diastasis: mechanically deceptive and often overlooked. Ann Otol Rhinol Laryngol. 2011;120(2):71-80. 10. Zur KB, Cotton S, Kelchner L, Baker S, Weinrich B, Lee L. Pediatric Voice Handicap Index (pVHI): a new tool for evaluating pediatric dysphonia. Int J Ped Otorhinolaryngol. 2007;71(1):77-82. 11. Titze I. Workshop on Acoustic Voice Analysis: Summary Statement. Iowa City, IA: National Center for Voice and Speech; 1995.
Downloaded from aor.sagepub.com at UNIV OF GEORGIA LIBRARIES on May 28, 2015
