Surgical Outcomes & Revision Rates of Velopharyngeal Insufficiency in Children (PDF)

American Journal of Otolaryngology–Head and Neck Medicine and Surgery 45 (2024) 104341 Contents lists available at ScienceDirect American Journal of Otolaryngolo...

American Journal of Otolaryngology–Head and Neck Medicine and Surgery 45 (2024) 104341 Contents lists available at ScienceDirect American Journal of Otolaryngology–Head and Neck Medicine and Surgery journal homepage: www.elsevier.com/locate/amjoto Surgical outcomes and revision rates for velopharyngeal insufficiency (VPI) in syndromic and non-syndromic children: A systematic review and meta-analysis Evan S. Chernov a, b, April N. Taniguchi a, c, Shaun A. Nguyen a, *, Sarah R. Sutton a, d, Phayvanh P. Pecha a, Krishna G. Patel a, Melissa Montiel e, William W. Carroll a a Medical University of South Carolina, Department of Otolaryngology-Head and Neck Surgery, 135 Rutledge Avenue, Charleston, SC 29425, USA b SUNY Upstate Medical University, School of Medicine, 766 Irving Ave, Syracuse, NY 13210, USA c University of Central Florida, School of Medicine, 6850 Lake Nona Blvd, Orlando, FL 32827, USA d University of Nevada, Reno, School of Medicine, 1664 N Virginia St, Reno, NV 89557, USA e Medical University of South Carolina, Department of Speech Language Pathology, 135 Rutledge Avenue, Charleston, SC 29425, USA A R T I C L E I N F O A B S T R A C T Keywords: Purpose: To evaluate pre- and post-operative resonance, surgical technique, revision rate, and revision indication Velopharyngeal insufficiency among syndromic and non-syndromic children with velopharyngeal insufficiency (VPI). Pediatric Materials and methods: A systematic review was conducted through July 2022. Children surgically treated for VPI Resonance were included. A meta-analysis of single means, proportions, comparison of proportions, and mean differences Syndromic Non-syndromic with 95 % confidence interval [CI] was conducted. Surgery Results: Twenty-three articles (n = 1437) were included in the analysis. The most common surgery was Sphincter Pharyngoplasty (SP), 62.6 % [31.3–88.9] for syndromic and 76.3 % [37.5–98.9] for non-syndromic children. Among all surgical techniques, for syndromic and non-syndromic children, 54.8 % [30.9–77.5] and 73.9 % [61.3–84.6] obtained normal resonance post-operatively, respectively. Syndromic patients obtained normal resonance post-operatively in 83.3 % [57.7–96.6] of Combined Furlow Palatoplasty and Sphincter Phar­ yngoplasty (CPSP), 72.6 % [54.5–87.5] of Pharyngeal Flap (PF), and 45.1 % [13.2–79.8] of Sphincter Phar­ yngoplasty (SP) surgeries. Non-syndromic patients obtained normal resonance post-operatively in 79.2 % [66.4–88.8] of PF and 75.2 % [61.8–86.5] of SP surgeries. The revision rate for syndromic and non-syndromic patients was 19.9 % [15.0–25.6] and 11.3 % [5.8–18.3], respectively. The difference was statistically significant, 8.6 % [2.9–15.0, p = 0.003]. Syndromic patients who underwent PF were least likely to undergo revision surgery as compared to SP and CPSP, 7.7 % [2.3–17.9] vs. 23.7 % [15.5–33.1] and 15.3 % [2.8–40.7], respectively. Conclusions: Syndromic children had higher revision rates and were significantly less likely to obtain normal resonance following primary surgery than non-syndromic patients. Among syndromic children, PF and CPSP have been shown to improve resonance and reduce revision rates more so than SP alone. 1. Introduction [3–6]. While speech therapy may be useful for concomitant speech disorders, anatomical limitations of VPI prevent proper velopharyngeal Velopharyngeal Insufficiency (VPI) has been shown to negatively closure which cannot be overcome with therapy. If conservative impact quality of life in children. VPI commonly presents with nasal management fails, there are several surgical options including Pharyn­ air emission, abnormal resonance with hypernasality, and sometimes geal Flap (PF), Sphincter Pharyngoplasty (SP), Furlow Palatoplasty (FP), nasal regurgitation of oropharyngeal contents [1,2]. VPI occurs in pa­ and Combined Furlow Palatoplasty and Sphincter Pharyngoplasty tients with cleft palate at rates around 20–30 % following palatoplasty (CPSP) [1,7]. New adjuncts include buccal myomucosal flaps and pos­ and has associations with 22q11.2 microdeletion syndrome (22qDS) terior pharyngeal wall augmentation (including injection * Corresponding author at: Department of Otolaryngology—Head and Neck Surgery, Medical University of South Carolina, 135 Rutledge Avenue, MSC 550, Charleston, SC 29425, USA. E-mail address: [email protected] (S.A. Nguyen). https://doi.org/10.1016/j.amjoto.2024.104341 Received 27 February 2024; Available online 1 May 2024 0196-0709/© 2024 Elsevier Inc. All rights reserved. E.S. Chernov et al. American Journal of Otolaryngology–Head and Neck Medicine and Surgery 45 (2024) 104341 pharyngoplasty). Without adequate management, social limitations and well as analyze the differences in revision indications among syndromic emotional impacts are common. and non-syndromic children. Given the anatomic and neurologic variation among syndromic and non-syndromic patients as well as the range of surgical techniques used 2. Materials and methods for VPI correction, the post-operative complications and success rates may vary. Many diagnostic modalities exist for measuring VPI, leading 2.1. Search criteria to subjective methods by which providers determine the necessity of primary and revision surgery. While VPI surgical revision risk factors This study was conducted according to the Preferred Reporting Items can be found for syndromic and non-syndromic pediatric patients, a for Systematic Reviews and Meta-analyses guidelines. To identify comprehensive comparison of these populations does not, to our studies for inclusion, two authors (A.N.T and S.R.S) created a search knowledge, exist. strategy for four databases: CINAHL (EBSCO), Cochrane Library (Wiley), A more comprehensive investigation into syndromic and non- PubMed (US National Library of Medicine, National Institutes of syndromic patients’ VPI surgeries is needed to understand the post- Health), and SCOPUS (Elsevier). Databases were searched from date of operative complications that are deemed severe enough to re-operate. inception through July 29, 2022. The search strategies used a combi­ Following the PICOS (Participant, Intervention, Comparison, Outcome, nation of subject headings and keywords for the following concepts: and Study design) statement, we aimed to offer an updated systematic Velopharyngeal Insufficiency, surgery, and pediatric. As outlined in review and meta-analysis of surgical outcomes for VPI correction, as Supplemental Appendix A, the PubMed search strategy was modified for Identification of studies via databases and registers Records identified from: Identification CINAHL (n = 103) Records removed before Cochrane (n = 45) screening: PubMed (n = 495) Duplicate records removed (n SCOPUS (n = 361) = 168) Records screened (n = 836) Records excluded (n = 766) Reports sought for retrieval Reports not retrieved (n = 70) Screening (n = 0) Reports assessed for eligibility Reports excluded: (n = 70) Adult population (n = 32) Wrong Intervention (n = 7) Lack of sufficient information (n = 4) Wrong Outcomes (n = 2) Wrong study design (n = 1) Wrong patient population(n = 1) Included Studies included in review (n = 23) Fig. 1. PRISMA Flow Diagram. From: Page MJ, McKenzie JE, Bossuyt PM, Boutron I, Hoffmann TC, Mulrow CD, et al. The PRISMA 2020 statement: an updated guideline for reporting systematic reviews. BMJ 2021; 372: n71. https://doi.org/10.1136/bmj.n71. 2 E.S. Chernov et al. American Journal of Otolaryngology–Head and Neck Medicine and Surgery 45 (2024) 104341 the three other databases. To identify additional articles, the references with some studies defining the categories as 7 of relevant articles were hand-searched. References were exported into mm, respectively. Table 1 illustrates the resonance scales that individual review management software, Covidence, (Veritas Health Innovation studies used to display hyponasality and hypernasality as well as this Ltd), for study selection as shown in Fig. 1. study’s conversion to a uniform 0–5 scale for comparative analysis. 2.2. Selection criteria 2.5. Statistical analysis Any studies assessing surgical intervention for VPI and revision rates Meta-analysis of single means (age) was performed by Comprehen­ for children (0–18 years) were included in the analysis. All study designs sive Meta-Analysis version 4 (Biostat Inc., Englewood, NJ, USA). Meta- except for systematic reviews and meta-analyses were considered for analysis of continuous measures (syndromic revision, non-syndromic inclusion. The patient population of interest focused on determining the revision) between pre-operative resonance versus post-operative reso­ following conditions: syndromic, non-syndromic, velopharyngeal nance groups were performed with RevMan v5.4.2 (The Cochrane closure pattern, and cleft palate. Exclusion criteria included non-English Collaboration, 2020). Meta-analysis of proportions (revision rates, studies, reviews, duplicates, inaccessible articles, or those with missing revision surgery indications and outcomes, …) was performed using statistical data. Studies were ineligible if post-operative speech out­ MedCalc 20.110 (MedCalc Software Ltd., Ostend, Belgium; http comes and/or revision rates were not measured. Two reviewers (A.N.T and S.R.S) independently assessed abstracts to identify articles that met the inclusion criteria and then applied exclusion criteria to full text re­ Table 1 Resonance scales. view. Conflicts were resolved by a third author (S.A.N.). Study Study’s resonance scale Conversion to universal scale 2.3. Data extraction Alvarez Carvajal Normal (0) Normal (0) 2018 Hyponasality (0) Hyponasality (0) Data extraction was conducted independently by three reviewers (E. Mild hypernasality (1) Mild hypernasality (1) S.C, A.N.T, and S.R.S). Data extracted from studies included author, Moderate hypernasality (2) Moderate hypernasality (3) publication year, country of study, study design, patient characteristics, Severe hypernasality (3) Severe hypernasality (5) surgery type, pre- and post-op resonance, and revision indication. Dis­ Bezuhly 2012 Within normal limits (0) Within normal limits (0) agreements were resolved by S.A.N. Mild hypernasality (1) Mild hypernasality (1) Moderate hypernasality (2) Moderate hypernasality (3) Articles that met criteria were critically appraised to assess level of Severe hypernasality (3) Severe hypernasality (5) evidence using the Oxford Center for Evidence-Based Medicine criteria Bohm 2013 Within acceptable limits (0) Within acceptable limits (0). The Risk of Bias in Non-Randomized Studies—of Interventions Hyponasality (0) Hyponasality (0) (ROBINS-I) tool was used to evaluate non-randomized studies (Fig. 2) Mild hypernasality (1) Mild hypernasality (1) Mild-moderate Mild-moderate. Two authors (A.N.T and S.R.S) performed independent risk as­ hypernasality (2) hypernasality (2) sessments on all studies. Risk of bias assessment included: bias due to Mixed resonance (2) Mixed resonance (2) confounding, bias in selection of participants into the study, bias in Moderate hypernasality (3) Moderate hypernasality (3) classification of interventions, bias due to deviations from intended in­ Moderate-severe Moderate-severe terventions, bias due to missing data, bias in measurement of outcomes, hypernasality (4) hypernasality (4) Severe hypernasality (5) Severe hypernasality (5) and bias in selection of the reported results. Each aspect was graded as Bohm 2019 Within acceptable limits (0) Within acceptable limits (0) low, unclear, or high. Hyponasality (0) Hyponasality (0) Mild hypernasality (1) Mild hypernasality (1) 2.4. Definitions Mild-moderate Mild-moderate hypernasality (2) hypernasality (2) Mixed resonance (2) Mixed resonance (2) In this study, surgical success was determined based on individual Moderate hypernasality (3) Moderate hypernasality (3) studies’ criteria such as: normalized speech [11,12], no or mild residual Moderate-severe Moderate-severe hypernasality [13–16], relief of airway obstruction for hyponasal pa­ hypernasality (4) hypernasality (4) Severe hypernasality (5) Severe hypernasality (5) tients [12,13], low Speech Language Pathologist-3 (SLP-3) scores Elsherbiny 2020 No hypernasality (0) No hypernasality (0) [17,18], velopharyngeal competence on speech evaluation , and Mild hypernasality (1) Mild hypernasality (1) proper velopharyngeal closure on nasendoscopy and/or fluoroscopy Inconsistent hypernasality Inconsistent hypernasality. When analyzing surgical revision rates, studies whose inclusion (1) (1) criteria required revision were excluded to avoid biasing the data. Moderate hypernasality (2) Moderate hypernasality (3) Severe hypernasality (3) Severe hypernasality (5) Velopharyngeal gap, assessed with nasendoscopy, refers to the space Kaye 2022a Normal resonance (0) Normal resonance (0) between the posterior aspect of the soft palate and the posterior Mild hypernasality (1) Mild hypernasality (1) pharyngeal wall. It is typically categorized as small, moderate, or large Mild-moderate Mild-moderate hypernasality (2) hypernasality (2) Mixed resonance (2) Mixed resonance (2) Moderate hypernasality (3) Moderate hypernasality (3) Moderate-severe Moderate-severe hypernasality (4) hypernasality (4) Severe hypernasality (5) Severe hypernasality (5) Sie 1998b Normal resonance Normal resonance (0) Hyponasality Hyponasality (0) Mild hypernasality Mild hypernasality (1) Moderate hypernasality Moderate hypernasality (3) Severe hypernasality Severe hypernasality (5) Widdershoven Never hypernasal Never hypernasal (0) 2008b Sometimes hypernasal Sometimes hypernasal (2) Always hypernasal Always hypernasal (4) a Numerical scale value in parentheses. b Fig. 2. Risk of bias assessment. Study did not include numerical scale; displayed verbally in results. 3 E.S. Chernov et al. American Journal of Otolaryngology–Head and Neck Medicine and Surgery 45 (2024) 104341 s://www.medcalc.org; accessed on 2023). The revision rates and success 3. Results rates were expressed as a percentage with 95 % confidence intervals [CI]. Each measure was weighted according to the number of patients 3.1. Overview of included studies affected. The weighted-summary proportion was calculated by the Freeman–Tukey transformation. Heterogeneity among studies was As shown in Fig. 1, the literature search yielded 836 unique articles assessed using χ2 and I2 statistics. I2 < 50 % indicated acceptable het­ with title and abstract screening excluding 766 reports. Full text review erogeneity, and therefore the fixed-effects model was used. Otherwise, excluded 47 articles, resulting in 23 studies for data extraction and the random-effects model was performed. In addition, a comparison of analysis [4,11–18,22–35]. The articles selected for inclusion were pub­ weighted proportions was done to compare revision surgery outcomes lished between 1986 and 2022 reporting on 1437 patients with a mean between gender, PF vs. SP. Finally, Egger’s tests with funnel plots were age of 7.6 years (range, 0.4 to 18.8). The studies (Table 2) were level 3 performed to further assess the risk of publication bias [20,21]. Potential and 4 based on Oxford Levels of Evidence. As displayed in Fig. 2, po­ publication bias was evaluated by visual inspection of the funnel plot, as tential sources of bias were most pronounced for bias in measurement of bias results in asymmetry of the funnel plot, and Egger’s test, which outcomes and bias due to confounding. A funnel plot with Egger’s test statistically examines this asymmetry (Supplemental Appendix Fig. B.1). (0.1, [− 0.9–1.2], p = 0.80) demonstrated all studies were within the A p-value of