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Abstract

Background

Pediatric laryngotracheal stenosis often requires open airway reconstruction. While these surgeries establish an airway for adequate ventilation, many patients develop subsequent dysphonia. Numerous studies have reported outcomes related to voice.

Objective

This study aims to evaluate dysphonia in pediatric patients following open airway reconstruction, focusing on acoustic parameters, perceptual voice quality, and voice-related quality of life.

Methods

A comprehensive search using the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines across 6 databases identified articles involving pediatric patients who underwent open airway reconstruction and reported postoperative vocal acoustic parameters, perceptual voice quality, voice-related quality of life, or vocal mechanics. Articles were assessed for bias risk, and common outcomes were synthesized qualitatively and quantitatively using meta-analyses.

Results

Among 4089 articles, 21 were included, involving 497 pediatric patients. Laryngotracheoplasty was the most common procedure followed by cricotracheal resection. The Consensus Auditory-Perceptual Evaluation of Voice (CAPE-V) scale was frequently used to assess voice quality, with a mean score of 55.6 [95% confidence intervals (CIs): 47.9-63.3]. Voice-related quality of life was measured using the pediatric Voice Handicap Index (pVHI) and Pediatric Voice-Related Quality of Life Survey, with mean scores of 35.6 (95% CI: 21.4-49.7) and 83.7 (95% CI: 74.1-93.2), respectively. The fundamental frequency was 210.5 (95% CI: 174.6-246.3). Other common findings included supraglottic phonation, anterior commissure blunting, posterior glottic diastasis, and abnormal vocal cord mobility.

Conclusion

Pediatric patients experiencing dysphonia after open airway reconstruction exhibited moderately decreased voice quality and reduced voice-related quality of life. However, there was inconsistency in study protocols and outcome measures used. Preserving voice quality during airway reconstruction is crucial to avoid negative impacts on quality of life.

Graphical abstract

Keywords

pediatric airway reconstruction dysphonia quality of life

Background

Pediatric laryngotracheal stenosis most often occurs in the subglottis and may be congenital or acquired. It most commonly occurs in patients with a history of endotracheal intubation.¹ Depending on the severity, these patients may require interventions, ranging from endoscopic procedures to open airway surgery or a combination thereof. Three main categories of open airway reconstruction surgeries (OARS) exist: expansion surgery such as laryngotracheoplasty (LTP) with anterior and/or posterior cartilage grafts, resection surgery such as cricotracheal resection (CTR), and slide tracheoplasty.² These OARS are intended to establish an airway of adequate caliber to enable appropriate ventilation and obviate tracheostomy placement or allow decannulation if a tracheostomy is already in place. However, OARS are associated with subsequent voice abnormalities in approximately half of patients.³

While airway symptoms and decannulation are the primary concerns in the early years following OARS, concerns about voice typically arise in adolescence, a period marked with new peers who may notice and comment on an abnormal voice. Dysphonia may also become more concerning as one’s increased awareness of career goals and choices develop.⁴ Dysphonia has the potential to negatively affect not only physical aspects of voicing and communication, but also social and emotional implications. In fact, adolescents with dysphonia report feelings of anger, sadness, embarrassment, and nervousness due to their voice.⁵ Furthermore, it is demonstrated that high school teachers have more negative perceptions and attitudes toward adolescents with voice disorders compared to those with normal voices, even across several personality traits and attributes including likeability, sociability, and employability.⁶

Numerous studies on children with a history of OARS have reported outcomes related to vocal parameters, objective voice quality scores, as well as voice-related quality of life scores.^7-27 The present study aims to systematically review the literature on the voice outcomes of children who have had open airway reconstructive surgery. Specifically, this review’s objectives are to (1) identify relevant studies and critically assess their research quality; (2) perform meta-analyses on important outcome measures; (3) synthesize findings from the studies to draw general conclusions, uncover knowledge gaps, and guide future research.

Methods

This study used the Preferred Reporting Items for Systematic Reviews and Meta-analysis (PRISMA) guidelines.²⁸ A computerized comprehensive search of the literature was performed by a medical librarian and an otolaryngologist specializing in pediatric airway on PubMed, Embase, CINAHL, Web of Science, Medline, and the EBM Reviews databases. These terms included “Pediatric,” “Voice,” “Dysphonia,” and “Open airway surgery.” Additional search terms were identified based on keyword lists and references found in the articles. Medical subject headings were also tailored for certain databases (Supplemental Table 1).

To select articles for analysis, the inclusion criteria were as follows: (1) study must include subjects aged 18 years and younger having undergone OARS, which includes laryngofissure, LTP with or without anterior or posterior grafts, CTR, or slide tracheoplasty; (2) report qualitative or quantitative voice assessments, such as those related to vocal acoustic characteristics, voice quality, voice-related quality of life, or vocal mechanics; and (3) either French or English language full text. Articles were excluded if no abstract was available or if they were classified as review articles or meta-analyses. Articles were also excluded if they only presented patients who were all included in another article, such as articles presenting preliminary results on a set of patients who were all included in a later article. An initial screening of titles and abstracts of all identified articles was performed by 2 independent reviewers. Some duplicate articles were also removed at this stage. The remaining articles were reviewed by these 2 independent investigators using the articles’ full text and evaluated for inclusion based on the inclusion and exclusion criteria detailed above. Any disagreements between the 2 reviewers in this regard were resolved by discussion between the reviews and a mutual decision was made.

The articles retained for analyses all underwent quality assessments using the Risk of Bias Assessment tool for Non-randomized Studies (RoBANS).²⁹ This validated tool is harmonized with other tools, including Cochrane’s Risk of Bias tool. It contains 6 domains, each classified as either “High,” “Low,” or “Unclear” risk of bias: selection, confounding, measurement, blinding, incomplete outcome data, and selective outcome reporting, The overall risk of bias for an article was deemed “High” if any of the 6 domains were classified as “High” or “Unclear.” Otherwise, it was deemed “Low.” Data were extracted from the articles, including the study population and its characteristics, study design, as well as results pertaining to postoperative voice outcomes. Voice outcomes were divided into the following categories: vocal acoustics, voice quality, voice-related quality of life, and others.

For quantitative outcomes reported in at least 4 studies, meta-analysis was performed. Outcomes are reported with 95% confidence intervals (CIs). Statistical heterogeneity was evaluated using the I² statistic. Meta-analysis and plots were conducted using Comprehensive Meta-Analysis software V3 (Biostat).

Results

The search identified a total of 4089 articles. After the final review, 21 articles were selected for inclusion (Figure 1).^7-27 Table 1 presents a summary of the articles included in this study. Most articles were case series, mostly retrospective, and there was one case report. There were 497 patients included in the articles. Based on the 330 patients (from 18 articles) for which sex information was available, 46.9% were male. The mean age was 10.4 years among the 403 patients (from 18 articles) where age information was available.

Figure 1.

PRISMA flowchart. This Preferred Reporting Items for Systematic Reviews and Meta-Analyses flowchart presents the number of articles through the identification, screening, and selection process of the systematic review. Four thousand eighty-nine records were identified from the database search. Of these, 1144 reports were deemed potentially relevant after title and abstract screening and sought for retrieval. Following retrieval, the remaining 163 reports were assessed by full-text review. Among these, 21 articles were included in this study.

Table 1.

Summary of Included Studies.

Authors and year	Study type	n	Male (%)	Age, mean (y)	Type of surgery	Open airway surgeries, mean (n)
de Alarcon et al (2009)¹¹	Retrospective case series	42	NA	7.1	LTR, CTR, Endoscopic procedures	NA
Bergeron et al (2019)⁷	Retrospective cohort study	21	33.3	14	LTR, CTR, Tracheoplasty, Endoscopic procedures	2.6
Bergeron et al (2022)⁸	Retrospective case series	34	41.2	13.6	LTR, CTR, Tracheoplasty, Endoscopic procedures	1.8
Bliss et al (2015)⁹	Case report	1	0.0	15	LTR	1
Cohen et al (2019)¹⁰	Prospective observational	11	63.6	8.54	LTR, CTR	1.55
Elders et al (2021)¹²	Prospective cohort study	48	NA	14.4	LTR, CTR	1.79
Francois et al (1997)¹³	Case-control	10	40.0	7.81	LTR	NA
Geneid et al (2011)¹⁴	Retrospective cohort study	10	100.0	NA	LTR	NA
George et al (2010)¹⁵	Retrospective case series	77	NA	NA	Partial CTR	NA
Kelchner et al (2010)¹⁷	Retrospective case series	21	57.1	8	LTR, CTR	2.2
Kelchner et al (2010)¹⁶	Prospective case series	50	36.0	11	LTR	NA
Krival et al (2007)¹⁸	Retrospective case series	16	31.3	13.5	LTR, CTR	NA
MacArthur et al (1994)¹⁹	Retrospective case series	9	44.4	6	LTR	2
Maunsell et al (2021)²⁰	Prospective case series	20	55.0	4.75	LTR, CTR	2.3
Pech et al (1995)²¹	Retrospective case series	23	73.0	11.5	LTR, CTR	NA
Pullens et al (2017)²²	Prospective case series	55	45.5	11	LTR, CTR	1.8
Rovo et al (2020)²³	Prospective case series	7	42.9	NA	Slide tracheoplasty	1.14
Tirado et al (2011)²⁴	Prospective case series	12	58.3	9.66	LTR	1.33
Weinrich et al (2007)²⁵	Prospective cohort study	12	33.3	9.67	LTR	NA
Zacharias et al (2016)²⁶	Retrospective case series	11	18.2	18	LTR	NA
Zalzal et al (1993)²⁷	Retrospective case series	7	71.4	3.83	LTR	2

Sample size (N) does not include healthy volunteers.

Abbreviations: CTR, cricotracheal resection; LTR, laryngotracheal reconstruction; N, numbers; NA, information not available in the article’s text.

LTR was the most common OARS (19/21 articles). Information about the use of anterior and/or posterior cartilage grafts was seldom available in the articles. Out of 21, 11 articles included patients having undergone CTR. The number of open airway surgeries performed was available in 12 articles, with a mean across them of 1.79 open airway surgeries. As the articles often report summarized results and not individual patient data, it was not possible to compare outcomes between different groups such as sex, age groups, single versus multiple airway surgeries, or different types of airway surgeries

An overview of the different outcome measures used in each study is presented in Table 2.

Table 2.

Outcomes Assessed by Study.

Authors	Perceptual voice quality	Quality of life scales	Acoustic parameters	Other outcomes
de Alarcon et al¹¹	CAPE-V	pVHI
Bergeron et al⁷	CAPE-V	pVHI	MPT; F0; Airflow; Pressure; Intensity
Bergeron et al⁸	CAPE-V	pVHI	MPT; Airflow; Pressure; Intensity
Bliss et al⁹	GRBAS	pVHI	MPT
Cohen et al¹⁰	CAPE-V; GRBAS	PVRQOL
Elders et al¹²	DSI	pVHI	MPT; Jitter; Lowest intensity; Highest frequency
Francois et al¹³			MPT; F0	Subjective voice quality
Geneid et al¹⁴		PVRQOL; PVOS; HRQOL 16D and 17D
George et al¹⁵				Subjective dysphonia grades
Kelchner et al¹⁷	CAPE-V		MPT; F0; Airflow; Pressure; Intensity
Kelchner et al¹⁶	CAPE-V
Krival et al¹⁸	CAPE-V		F0	Laryngeal findings
MacArthur et al¹⁹			F0; Jitter	Subjective voice qualities; Laryngeal findings
Maunsell et al²⁰	CAPE-V	PVRQOL	F0; Jitter; Shimmer; Harmonic to noise ratio; Spectral emphasis
Pech et al²¹			MPT; Jitter; Airflow; Intensity; Pitch; Phonatory quotient	Subjective voice qualities
Pullens et al²²	DSI; VAS	pVHI
Rovo et al²³		QOL Questionnaire	Jitter; Shimmer; Harmonic to noise ratio; Pitch
Tirado et al²⁴	CAPE-V	PVRQOL; HUI3; IFS; VAS	F0	Laryngeal findings
Weinrich et al²⁵	CAPE-V, strain subscale		Airflow; Pressure
Zacharias et al²⁶	CAPE-V	pVHI	MPT; F0; Intensity
Zalzal et al²⁷			F0; Jitter	Laryngeal findings

Abbreviations: CAPE-V, Consensus Auditory-Perceptual Evaluation of Voice; DSI, Dysphonia Severity Index; F0, fundamental frequency; GRBAS, Grade, Roughness, Breathiness, Asthenia, Strain scale; HRQOL 16D and 17D, Healthy-Related Quality of Life Measures; HUI-3, Health Utilities Index mark 3; IFS, Impact on Family Life Scale; MPT, mean phonation time; pVHI, Pediatric Voice Handicap Index; PVOS, Pediatric Voice Outcomes Scale; PVRQOL, Pediatric Voice-Related Quality Of Life; QOL, quality of life; VAS, Visual Analog Scale; VHI, Voice Handicap Index.

Table 3 highlights the meta-analysis of the main voice findings.

Table 3.

Meta-Analysis of the Main Voice Findings for Patients With Open Airway Surgery.

Voice findings	Mean (95% CI)	SE
Vocal and aerodynamic assessment
Fundamental frequency (Hz)	210.5 (174.6-246.3)	18.3
Jitter (%)	1.5 (0.8-0.25)	0.3
Phonation time (s)	7.7 (5.7-9.8)	1
Maximum intensity (Hz)	79.9 (77.6-82.1)	1.1
Airflow (L/min)	180.8 (139.4-222.24)	21.1
Peak air pressure (cm/H₂O)	10.9 (9.1-12.8)	0.9
Perceptual assessment
CAPE-V	55.612 (47.9-63.3)	3.9
Voice-related quality of life
pVHI	35.6 (21.4-49.7)	7.2
PVRQOL	83.7 (74.1-93.2)	4.9

Abbreviations: CAPE-V, Consensus Auditory-Perceptual Evaluation of Voice; CI, confidence interval; cm, centimeter; Hz, hertz; L, liter; min, minute; pVHI, Pediatric Voice Handicap Index; PVRQOL, Pediatric Voice-Related Quality of Life; s, second; SE, standard error.

Perceptual Assessment of Voice

The Consensus Auditory-Perceptual Evaluation of Voice (CAPE-V) scale was the most used measure of perceptual voice quality, being used in 10 articles. The Grade, Roughness, Breathiness, Asthenia, Strain (GRBAS) scale and Dysphonia Severity Index (DSI) were used in 2 articles each. One article reported only the Strain subscale of the CAPE-V. Other measures of perceptual voice quality included subjective dysphonia severities or voice qualities, as well as a Visual Analog Scale judged by parents (Table 3). The mean CAPE-V was 55.6 (95% CI: 47.9-63.3). The forest plot for CAPE-V is shown in Figure 2. Voice was subjectively qualified as more strained, hoarse, weak, and breathy, with an overall moderately deviated voice quality.^13,14,19,21

Figure 2.

Forest plots for CAPE-V, pVHI, phonation time, and fundamental frequency. The forest plots illustrate the results of the meta-analysis conducted on 4 selected outcome measures. Individual study findings are represented by black squares and horizontal lines, denoting their values and corresponding 95% confidence intervals. The meta-analysis estimate is represented by a blue diamond, spanning the width of the 95% confidence interval. CAPE-V, Consensus Auditory-Perceptual Evaluation of Voice; Hz, hertz; pVHI, Pediatric Voice Handicap Index; s, seconds.

Voice-Related Quality of Life

The Pediatric Voice Handicap Index (pVHI) was the most used scale (used in 7 articles), while the Pediatric Voice-Related Quality of Life Survey (PVRQOL) was used less commonly (4 articles). Several other general Quality of life (QOL) and voice-related QOL scales were used for individual studies (Table 3). The mean pVHI was 35.6 (95% CI: 21.4-49.7), while the mean PVRQOL was 83.7 (95% CI: 74.1-93.2). The forest plots for these variables are shown in Figure 2.

Vocal and Aerodynamic Assessment

The most frequently reported outcomes in this category were fundamental frequency (9 articles), maximum phonation time (8 articles), and jitter (6 articles). Airflow, peak air pressure, and maximum intensity were other variables commonly reported. Table 3 presents the results of the meta-analysis including means and standard errors for vocal and aerodynamic variables. Figure 2 demonstrates the forest plots for fundamental frequency and phonation time. The mean fundamental frequency was 210.5 (95% CI: 174.6-246.3) and the mean phonation time was 7.7 seconds (95% CI: 5.7-9.8).

Structural Findings

Several studies reported laryngeal structural findings or commented on the laryngeal function and the mechanisms of phonation identified on endoscopy or dynamic imaging. Post airway reconstruction dysphonia patients were often grouped based on their site of phonation, either glottic or supraglottic. On endoscopy, MacArthur et al found abnormal vocal cord integrity in 78% of patients, anterior commissure blunting in 44%, posterior notching in 44%, abnormal vocal cord mobility in 44%, and abnormal vocal cord contact in 44% of patients.¹⁹ Francois et al found anterior commissure blunting in 60% of patients and impaired arytenoid movements in 30%.¹³ Tirado et al found decreased vocal cord contact in 3/8 patients, and asymmetry with displaced arytenoids in 3/8 patients.²⁴ Krival et al noted anteroposterior compression in glottic phonators and that 56% of patients had supraglottic or mixed phonation patterns.¹⁸ Krival et al, Kelchner et al, and Zacharias et al found that supraglottic phonators had a range of vibrating tissues and distinct patterns of supraglottic compression.^17,18,26 On high-speed videoendoscopy, Zacharias et al found that the source of vibration was the ventricular folds in 67% of patients.²⁶ Elders et al performed static and dynamic magnetic resonance imaging and identified that 81% of patients had vocal cord thickening, 72% had impaired vocal cord movements (mostly incomplete adduction), 60% had supraglottic collapse, and 21% had subglottic collapse.¹² Bergeron et al performed dynamic voice computed tomography and found that nearly all patients had some degree of supraglottic collapse that prevented perfect visualization of the larynx.⁷

All articles were judged to have a high overall risk of bias according to the RoBANS tool (Table 4). This was often due to the nature of the articles being retrospective case series or the presence of potentially significant confounding variables such as neonatal, medical, or surgical comorbidities. Most articles, however, were judged to have a low risk of bias in the categories of measurement bias, blinding, incomplete outcome data, and selective outcome reporting.

Table 4.

Risk of Bias Assessment Using RoBANS tool.

Study	Participant selection	Confounding variables	Measurement bias	Blinding of outcome assessment	Incomplete outcome data	Selective outcome reporting	Overall risk of bias^a
de Alarcon et al (2009)¹¹	High	High	Low	Low	High	Low	High
Bergeron et al (2019)⁷	High	High	Low	Low	Low	Low	High
Bergeron et al (2022)⁸	High	High	Low	Low	Low	Low	High
Bliss et al (2015)⁹	High	High	Low	High	Low	High	High
Cohen et al (2019)¹⁰	High	High	Low	Low	High	High	High
Elders et al (2021)¹²	Low	High	Low	Low	Low	Low	High
Francois et al (1997)¹³	Unclear	High	High	Low	Low	Low	High
Geneid et al (2011)¹⁴	High	High	Low	Low	Low	High	High
George et al (2010)¹⁵	High	High	High	Low	Low	Low	High
Kelchner et al (2010)¹⁷	High	High	Low	Low	High	Low	High
Kelchner et al (2010)¹⁶	Low	High	Low	Low	Low	Low	High
Krival et al (2007)¹⁸	High	High	Low	Low	Low	Low	High
MacArthur et al (1994)¹⁹	High	High	High	Low	High	Low	High
Maunsell et al (2020)²⁰	High	High	Low	Low	High	Low	High
Pech et al (1995)²¹	High	High	High	High	Low	Low	High
Pullens et al (2017)²²	High	High	Low	Low	High	Unclear	High
Rovo et al (2020)²³	Low	High	High	Low	Low	Low	High
Tirado et al (2011)²⁴	Low	Unclear	Low	Low	Low	Low	High
Weinrich et al (2007)²⁵	Low	High	Low	Low	Low	Low	High
Zacharias et al (2016)²⁶	High	High	Low	Low	Low	Low	High
Zalzal et al (1993)²⁷	High	High	High	Unclear	High	Low	High

Risk of bias was assessed considering biases in voice outcomes.

Abbreviations: RoBANS, risk of bias assessment tool for nonrandomized studies.

Overall risk of bias was deemed “High” if any of the 6 RoBANS domains were classified as “High” or “Unclear” and was otherwise deemed “Low.”

Discussion

This systematic review and meta-analysis assessed 21 articles regarding voice outcomes following pediatric open airway reconstruction, representing 497 patients in total. Overall, vocal quality was found to be moderately decreased in children following airway reconstruction, as demonstrated by the mean CAPE-V score. Furthermore, voice-related quality of life in this population is notably affected, as shown by the mean pVHI and PVRQOL scores. However significant, these major impacts were not surprising in a population having undergone almost 2 open airway surgeries per patient.

Vocal quality seems to be most frequently quantified with the CAPE-V, with 10 articles reporting it. The CAPE-V is a validated rating scale for the standardized perceptual evaluation of voice by clinicians.³⁰ It includes 5 subscales including roughness, breathiness, strain, pitch, and loudness, as well as an overall severity rating, which ranges from 0 to 100, with a higher score indicating a more severely deviant voice quality. In this case, the mean CAPE-V overall severity score of 55.6 is consistent with a moderately altered voice quality in children following open airway reconstruction. More specifically, this patient population’s voice is typically more hoarse, strained, weak, and breathy. For this study population whose mean age was 10.1 years, where normative fundamental frequency is around 250 to 270 Hz,^31,32 and a mean postoperative fundamental frequency of 210.5 Hz suggests a lower voice pitch than average. Maximum phonation time was found to be 7.7 seconds in this study population, which is slightly lower than normative values for children of the same age of around 8.98 seconds.³³

Several mechanisms for decreased voice quality in children following airway reconstruction have been proposed, including (1) vocal folds with abnormal mobility, vertical asymmetry, or scarring; (2) supraglottic compression and phonation; (3) posterior glottic diastasis; (4) epiglottic petiole prolapse; and (5) anterior commissure blunting.^2,8,34-38 Among studies which reported laryngeal structural findings, anterior commissure blunting was common, as were vocal cord abnormalities such as paralysis or incomplete adduction, as well as supraglottic compression and phonation. It is suggested that patterns of supraglottic compression may be compensatory and mitigate dysphonia.^17,39 Widely splayed vocal cords with posterior glottic diastasis, often the result of a large posterior graft, was also common. All these findings may contribute to altered voice qualities such as strained, weaker, breathier voices. Supraglottic phonation in particular may produce a more hoarse, rough voice.^2,18

Ultimately, this dysphonia can negatively influence these children’s quality of life, as demonstrated in this meta-analysis. To assess QOL, the pVHI was the most used scale, followed by the PVRQOL. Few studies used other QOL scales. Thus, meta-analyses were only performed for the pVHI and PVRQOL. The pVHI is a validated 23-item questionnaire to be completed by parents, divided into functional, physical, and emotional subcategories, intended to reflect the handicap inflicted by an abnormal voice.⁴⁰ The scale ranges from 0 to 92, with a higher score reflecting greater handicap, or lower voice-related quality of life. In this study, the mean pVHI was 35.6, suggesting moderate voice-related handicap in children following airway reconstruction. The PVRQOL is a validated 10-item questionnaire to evaluate QOL in children with dysphonia.⁴¹ Its scale ranges from 0 to 100, with a lower score indicating worse QOL. The result for the mean PVRQOL was 83.7, once again suggesting a moderately decreased voice-related quality of life in these children. These results are somewhat expected since it is known that in general, children with dysphonia may feel dissatisfied with their voice, frustrated, angry, sad, embarrassed or nervous about their voices.^5,42 It has been shown that listeners, including teachers, may have more negative perceptions toward children (and adults) with dysphonia, including stereotyping with negative perceptions of children’s unrelated attributes such as intelligence, academic performance, and personality traits.^43-45 Such biases may lead to educational, social, or vocational difficulties in adolescence.⁶ Globally, the effect of dysphonia in children, including following airway reconstruction, must not be neglected due to the demonstrated negative influence on voice-related quality of life.

Careful consideration should be made in an attempt to minimize the risk of dysphonia following airway reconstruction. The different types of airway reconstruction procedures may affect voice differently. CTR has been suggested to result in lower voice quality.¹¹ Other factors which may affect the risk of and degree of dysphonia include the preoperative extent and degree of stenosis,^15,22 duration of tracheostomy prior to LTR,¹⁹ complete laryngofissure,¹⁹ inadequate reapproximation of vocal folds after laryngofissure,⁴⁶ excessively wide posterior glottic grafts,^27,46 use and duration of stent placement,^19,21,27 and the total number of open airway reconstructions.^17,19,47 Awareness of these factors can help prevent or limit the degree of dysphonia. It has been proven that a formal evaluation in a multidisciplinary voice clinic prior to surgery can greatly affect the outcomes.^8,48

This study has several limitations. First, the articles selected were mostly retrospective case series and were at high risk of bias, often due to selection and confounding. This may ultimately bias the outcome measures reported in this study. The degree of dysphonia, for all reported outcomes in this study, could be overestimated since certain patients from the articles with milder dysphonia would be less likely to consult and/or participate in research for voice alterations. Conversely, dysphonia could be underestimated since some patients with moderate or severe dysphonia may be lost to follow-up and not included in research studies, but this is less likely than the former proposition. Furthermore, this population is notably known to have significant medical and surgical comorbidities, which may bias the results globally regarding the independent effect that OARS has on voice. Furthermore, some patient sets from the studies analyzed may have partly overlapped with each other, particularly for studies at the same centers or cities. We tried to account for this by excluding studies where we could ascertain that all patients were already accounted for in another study that was included. Finally, the studies were heterogeneous regarding their main study objectives, patient populations, types of airway reconstruction, the timing of follow-up and outcome measurements, as well as outcome measures reported. The overall conclusions that can be drawn from this study are thus of low-quality evidence but nonetheless provide important takeaways from the current literature. Despite this heterogeneity, our decision to undertake a meta-analysis was driven by a meticulous evaluation of the existing literature, which revealed a significant gap in comprehensive analyses that could provide robust insights into voice outcomes following open airway reconstruction. With a dataset encompassing more than 20 studies and almost 500 patients, our analysis offers a more powerful perspective on this research area, enabling us to derive conclusions with greater statistical power and value than individual studies alone. By providing a comprehensive overview, we also aim to promote a deeper understanding of this topic and stimulate further research, emphasizing the need for the use of more rigorous protocols, prospective studies, and validated consistent outcome measures. Airway surgery patients are undoubtedly a complex and diverse group, and this analysis reflects the intricate nature of their varied needs and demands. Finally, the conclusions and takeaways from this analysis bear importance and clinical relevance for otolaryngologists.

Future research should prioritize the use of formalized, validated, reliable, and consistent outcome measures for voice quality and voice-related quality of life in this patient population. In this regard, this study identified the CAPE-V, pVHI, fundamental frequency, and maximum phonation time as the most frequently used outcome measures. Standardization would help compare findings across different settings and studies. Ideally, future studies should aim to use a prospective protocol with a predefined timing of postsurgical assessments.

Conclusions

This systematic review and meta-analysis demonstrated that patients with dysphonia following airway reconstruction have moderately decreased voice quality as expressed by the CAPE-V, as well as reduced voice-related quality of life as shown by the pVHI and PVRQOL. However, there is a general lack of consistency in study protocols and outcome measures used in this population. Future studies should focus on using prospective, standardized protocols and outcome measures consistent with the literature. Careful consideration should be made during airway reconstruction to attempt to preserve voice quality and voice-related quality of life in this population.

Supplemental Material

sj-docx-1-ohn-10.1177_19160216241266570 – Supplemental material for Characterizing Dysphonia After Pediatric Open Airway Reconstruction: Systematic Review and Meta-Analysis

Supplemental material, sj-docx-1-ohn-10.1177_19160216241266570 for Characterizing Dysphonia After Pediatric Open Airway Reconstruction: Systematic Review and Meta-Analysis by Zachary Dahan, Alix Pincivy, Carol Nhan and Mathieu Bergeron in Journal of Otolaryngology - Head & Neck Surgery

Footnotes

Acknowledgements

None.

Author Contributions

Z.D. conducted the literature search, performed data extraction, and redacted the manuscript. A.P. assisted with identifying search terms and retrieving articles for the systematic review. C.N. and M.B. both screened and reviewed articles for inclusion. M.B. also assisted with data analysis and manuscript drafting. All authors read and approved the final manuscript.

Availability of Data and Materials

The datasets used and/or analyzed during the current study are available from the corresponding author on reasonable request.

Consent for Publication

Not applicable.

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.

Ethics Approval and Consent to Participate

Not applicable.

ORCID iDs

Alix Pincivy

Mathieu Bergeron

Supplemental Material

Supplemental material for this article is available online.

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