Supraglottic collapse as noted during a microlaryngoscopy (a arytenoids, p pyriform sinus)
Posterior Glottic Diastasis
Laryngotracheoplasty (LTP)
LTP is a surgical procedure to expand the airway diameter by placing an anterior and/or posterior costal cartilage graft (ACCG, APCCG, PCCG), most often costal cartilage or thyroid ala cartilage. This procedure can be performed as a double-stage surgery (placement of a tracheostomy) or as a single-stage surgery (removal or non-placement of a tracheostomy). Multiple factors may impact voice outcomes, such as prelaryngeal muscle dissection during surgery, baseline subglottic stenosis severity, and laryngeal nerve injury.
Cricotracheal Resection (CTR)
CTR involves the excision of the anterolateral cricoid plate and anastomosis of the distal tracheal ring to the proximal thyroid ala with suture lines placed in the posterior cricoid mucosa to reapproximate the trachealis to the more proximal cricoid plate [5]. This procedure should be reserved for experienced surgeons as results are highly surgeon dependent. Furthermore, this procedure may significantly alter the voice. In particular, CTR decreases the fundamental frequency of connected speech and vowel phonation and changes the acoustic signal type [6].
Slide Tracheoplasty
Slide tracheoplasty is a surgery to increase tracheal diameter. The trachea is opened anteriorly and posteriorly. It is then slid up onto itself and reconnected to make the trachea shorter, but wider. This procedure is classically performed for long-segment congenital tracheal stenosis. Limited data exist regarding specific voice outcomes after such procedures, but vocal fold paralysis has been reported in approximately 5% of patients [7].
Epidemiology
Complete laryngofissure
Cricotracheal resection
Posterior grafting
Higher grade of subglottic stenosis (SGS)
Revision airway surgery
History of multiple airway reconstructions
Patients with low-grade stenosis (grade 1–2), single-stage procedure, and fewer comorbidities are less likely to have post-reconstruction dysphonia [9, 10].
Pathophysiology
Abnormal vocal fold mobility
Persistent subglottic stenosis
Anterior commissure blunting
Posterior glottic diastasis
Prolapsed petiole
Vertical asymmetry of vocal folds
Vocal fold scaring
Supraglottic compression
Presentation
Most patients will report some degree of dysphonia after airway reconstruction. Typically, voice outcomes after airway reconstruction include roughness, breathiness, supraglottic phonation, or inappropriate pitch [1, 14]. These children often present when they are becoming more social or making decisions about future careers. In younger children (ages 5–10 years), issues may arise as children begin participation in sports and other social activities. As children reach middle school age, they may have more issues related to being in multiple different classroom settings with different peer groups and teachers who are not familiar with their voice. Concerns include peers noticing this dysphonia as well as difficulty being heard in a noisy environment, embarrassment about their voices, and reduced willingness to participate in class [15]. In adolescents, the voice is important for social interactions, defining their identity, and determining their future occupation. Teenagers report embarrassment and fear of peer responses to their voice, as well as frustration and social isolation [15].
Otolaryngologist and Speech-Language Pathologist Approach
Patients with dysphonia after airway reconstruction are typically complex, and there are a variety of parameters that must be evaluated. As such, the evaluation is best performed in a multidisciplinary fashion with both an otolaryngologist and speech-language pathologist well-versed in voice pathology. Multidisciplinary voice evaluation frequently influences the course of treatment for patients with airway reconstruction and dysphonia [14, 16].
History
The history for these patients is largely obtained from the parents, but the child’s perspective is also very important when discussing the management of voice concerns. Many children will have had voice issues throughout their life related to prior intubations, scarring in the airway, and prior surgical interventions. At the time of presentation for voice evaluation, it is necessary to determine the child’s airway history including intubation history, history or presence of a tracheostomy, and prior surgical interventions. Furthermore, attention should also focus on current medications and relevant comorbidities such as pulmonary or swallowing disorders. Details of the surgical interventions performed, such as if anterior or posterior grafts were placed or if a laryngofissure was performed, are useful in helping predict potential causes of the dysphonia. Reviewing operative notes and operative videos, if available, may also be helpful to determine potential causes of the dysphonia.
As with any history, the clinicians should determine the nature and course of the dysphonia, alleviating and aggravating factors, and if prior interventions (e.g., voice therapy, injections, surgery) have been attempted. If they have had prior interventions, the specifics of those and their outcomes should be elicited. The family’s perception of the voice quality (e.g., weak, breathy, rough, raspy, harsh, deep) and how it is affecting the child’s quality of life in all environments (e.g., home, school, social, work) is important. Families will sometimes report that the child has “two voices” – typically a weaker one (presumably the glottic voice) and a stronger but deeper one (presumably a supraglottic voice). They may describe this as a “duck” voice or “superhero” voice. For a younger preverbal patient, a formal preoperative voice evaluation may be challenging; however, the breathing pattern, presence of stridor, and babbling can be documented.
Indices such as the pediatric Voice Handicap Index (pVHI) [17] or Voice-Related Quality of Life (VRQL) [18] can assist in elucidating the perceived impact of dysphonia. Similar factors that may cause laryngeal irritation and exacerbate voice problems in other patients, such as laryngopharyngeal reflux, allergic rhinitis, chronic cough, asthma, sleep-disordered breathing, smoke exposure, and vocal misuse/overuse, must be evaluated, as well.
Additionally, operating on the larynx can significantly and disparately impact voice, airway, and swallowing. As such, the current airway status (e.g., tracheostomy, decannulated), presence of stridor or dyspnea on exertion, and time of last airway evaluation should be noted. Lastly, patients who undergo open airway reconstruction are also likely to experience some degree of postoperative dysphagia symptoms and delayed return to oral intake. Cough, choking events, and aspiration pneumonias should be documented. The patient’s current feeding status and swallowing safety should also be assessed.
Examination
The examination is typically completed in conjunction with the speech-language pathologist. Combined evaluation has been proven to be beneficial for decision-making regarding voice management and potential surgical interventions [14]. A general head and neck examination should be completed. Special attention should be paid to the intelligibility, voice quality, effort for voice production, and voice range. Specific perceptual instruments, such as the grade, roughness, breathiness, asthenia, and strain (GRBAS) [19] scale or Consensus Auditory-Perceptual Evaluation of Voice (CAPE-V) [20], are used to document the quality and severity of the dysphonia. These scales assist in establishing a baseline and monitoring progress over time. Interestingly, a prior study showed only weak-to-fair correlation between the parent-reported pVHI and expert ratings of voice quality using the CAPE-V [21]. The presence of diplophonia and if two discrete voices can be elicited should be documented.
The otolaryngologist should pay attention to the presence of stridor or respiratory distress; however, most children presenting for voice evaluation after airway reconstruction have typically overcome this challenge. Finally, patients who have undergone airway reconstruction may have other factors that can affect their voice outcome or ability to participate in therapy, such as syndromes, other congenital abnormalities, or developmental delay that should be noted.
Instrumental Assessment
Endoscopic Evaluation
Laryngoscopy and videostroboscopy should be performed. Again, the speech-language pathologist and otolaryngologist serve complimentary roles. Depending on the child’s age, ability to cooperate, and anatomy, flexible transnasal and/or rigid transoral 70-degree stroboscopy may be completed. Whereas one or the other is often adequate for evaluation of common laryngeal lesions, both transnasal and transoral exams may be necessary to fully evaluate the anatomy and function in these post-airway reconstruction patients. The use of a distal chip telescope will improve image quality, and recording the examination is useful for reference. Careful attention should be paid to attempting to determine the sound generator for phonation (e.g., glottic or supraglottic or both), presence of a posterior glottic gap, if the vocal folds are level, the degree of scarring, the mobility of the vocal folds and arytenoids, and the degree of effort/strain with vocalization. Of note, examination of the glottis during phonation is often difficult due to the degree of supraglottic collapse and/or squeeze seen in these patients as well as postsurgical anatomical variation. Parameters of vibratory patterns should also be evaluated via stroboscopic exam.
Rigid endoscopy in the operating room alone is not adequate for evaluation of vocal pathology. However, the mobility of the arytenoids, presence of posterior glottic scarring or diastasis, and other structural anomalies can be assessed and may add important information to the clinical picture.
Acoustic and Aerodynamic Evaluation
Post-airway reconstruction patients should undergo acoustic and aerodynamic evaluation. This is typically completed by the speech-language pathologist, and detailed descriptions of these exams can be found in other chapters. Briefly, acoustic analysis provides information regarding the fundamental frequency; jitter, shimmer, and noise-to-harmonic ratio; and frequency range and may also include spectral/cepstral measures such as cepstral peak prominence. Aerodynamic measures provide information regarding glottal efficiency by determining the average airflow rate and estimated subglottic pressures. Not all patients will be able to produce a type I (periodic) signal and a measurement of fundamental frequency. Common features seen in post-airway reconstruction patients are a lower pitch and reduced pitch range, breathiness, and a reduced maximum phonation time [14]. These assessments help provide a baseline and can be used to measure response to voice or surgical therapies over time. A prior study suggests that the majority of children are able to complete the acoustic and aerodynamic assessments with a significant proportion of post-airway reconstruction patients having severe dysphonia [22]. Recording enough voicing segments may be challenging and sometimes impossible; protocols should be tailored to the patient’s capability.