Abstract
Background
Obstructive sleep apnea (OSA) is a troublesome complication following surgical management of velopharyngeal insufficiency (VPI). The purpose of this study is to evaluate the comparative incidence of OSA following VPI surgery in the United States.
Methods
A retrospective analysis of cleft and non-cleft pediatric patients who underwent VPI surgery was performed using the IBM® MarketScan® Commercial Database. Patients were tracked longitudinally from 2007–2016 to evaluate the incidence of OSA. Multivariable regression was used to evaluate predictors of postoperative OSA and surgical revision.
Results
1,098 patients with an average age of 6.3 ± 4.0 years underwent a pharyngeal flap (61.0%), sphincter pharyngoplasty (22.2%), or palatal lengthening ± island flaps (16.8%). Diagnoses were predominantly cleft lip and/or palate (52.8%) and congenital oropharyngeal anomalies (42.6%). Eighty patients (7.3%) developed OSA at an average of 10.2 months postoperatively. Predictors of OSA included older age (p=0.014) and head and neck neoplasm (p=0.011). OSA rate following sphincter pharyngoplasty was 11.1% compared to 7.2% after pharyngeal flap. However, this difference was not significant on multivariable regression. Compared to sphincter pharyngoplasty, pharyngeal flap was associated with a lower risk of further surgery for OSA or palatal revision (OR 0.43, p=0.010). Of patients with cleft lip/palate, 35 developed OSA (6.0%) without a significant association with procedure type.
Conclusions
In this national claims database analysis of cleft and non-cleft pediatric patients, the rate of OSA following VPI surgery was not significantly different for pharyngeal flap compared to sphincter pharyngoplasty. Further investigation is needed to explore associations specifically within the cleft lip/palate population.
Background
Surgery for velopharyngeal insufficiency (VPI) is performed to improve speech; however, obstructive sleep and respiratory complications can be unintended consequences. The aperture of the velopharyngeal port can be conceptualized as a continuum, with VPI on one end representing an opening that is too wide, and obstructive sleep apnea (OSA) on the other as a complication of an opening that is too narrow.1 The goal of the craniofacial surgeon is to reach an equilibrium between these two ends of the spectrum, achieving improved resonance, proper nasal air escape, and adequate articulation without the respiratory compromise of OSA.2
Currently, no consensus on the pharyngoplasty procedure that affords the lowest rate of postoperative OSA exists. Palatoplasty, which involves palate muscle repositioning and lengthening through procedures such as the Furlow palatoplasty and buccal myomucosal (island) flaps,3,4 has demonstrated a lower rate of postoperative OSA compared to pharyngoplasty in VPI surgery.5–8 However, a palatal lengthening procedure is best suited for mild or moderate cases of VPI with misoriented palatal muscles and/or a small anterior to posterior gap; more severe cases require pharyngoplasty, either with a pharyngeal flap or sphincter pharyngoplasty.2,9 The pharyngeal flap consists of a flap of mucosa and superior pharyngeal constrictor muscle that is elevated from the posterior pharyngeal wall and sutured to the nasal layer of the velum in order to divide the pharyngeal port into two smaller ports.10 Sphincter pharyngoplasty creates dynamic port obstruction through elevation and transposition of bilateral myomucosal flaps to decrease the cross-sectional area of the velopharyngeal port.11
While early limited retrospective studies comparing pharyngoplasty methods have suggested a higher rate of OSA following pharyngeal flaps,12,13 more recent randomized controlled trials (RCTs) have demonstrated equivocal outcomes.14 Based on our clinical experience, we hypothesized that pharyngeal flaps confer no greater risk of postoperative OSA than sphincter pharyngoplasty. The purpose of this study was to leverage a nationwide database with longitudinal patient data to compare postoperative rates of OSA following VPI surgery in the pediatric population and to identify factors to target for risk reduction.
Methods
Database
Using the IBM® MarketScan® Research Databases, we performed a retrospective analysis of all patients who underwent VPI surgery from 2007 to 2016 in the United States. The MarketScan Research Databases contain de-identified patient-level healthcare claims data for millions of commercially-insured individuals.15 Data was drawn from the MarketScan Commercial Database, specifically the Inpatient Services and Outpatient Services datasets, which consist of 663 million and 8.7 billion claims, respectively. Each individual in a MarketScan database has a unique enrollee identifier, enabling linkage and longitudinal patient tracking. Studies using deidentified datasets that are hosted by the Stanford Center for Population Health Sciences (PHS) are covered under an umbrella Institutional Review Board (IRB) application. Individual IRB approval was not required for this study as it does not involve human subjects research. IBM MarketScan Databases are available to purchase by Federal, non-profit, academic, pharmaceutical, and other researchers. Use of the data is contingent on completing a data use agreement and purchasing the data needed to support the study. More information about licensing the IBM MarketScan Databases is available at: https://www.ibm.com/us-en/marketplace/marketscan-research-databases.
Variables
Using the Inpatient Services dataset, Current Procedural Terminology (CPT) codes (See Table, Supplemental Digital Content 1, which shows the list of Current Procedural Terminology (CPT) Codes) were used to identify individuals younger than 18 years who underwent surgery for VPI, including: palatal lengthening with or without island flap(s) (CPT 42220, 42227), sphincter pharyngoplasty (CPT 42950), and pharyngeal flap (CPT 42225, 42226). Using both the Inpatient Services and Outpatient Services datasets, claims with a diagnosis of OSA were extracted based on International Classification of Diseases, Ninth Revision (ICD-9) and Tenth Revision (ICD-10) diagnosis codes for obstructive sleep apnea (327.23 and G47.33, respectively). For cases where an enrollee identifier was associated with multiple claims with an OSA diagnosis, the first claim was selected and considered to be the date of OSA onset. The datasets were merged by enrollee number to identify patients who were diagnosed with OSA following surgery for VPI. Secondary VPI and revision operations were identified by CPT code denoting subsequent VPI surgery or revision palatoplasty (CPT 42215). Exclusion criteria included: diagnosis of OSA, sleep disturbance, or other obstructive respiratory disturbance at the time of, or prior to, primary VPI surgery; multiple different VPI procedures performed simultaneously; or fewer than 6 months follow up. Patients who were diagnosed with OSA following a second VPI surgery were also excluded in order to limit the analysis to those diagnosed following primary VPI surgery and prior to any secondary procedures.
Predictor variables extracted from the database included: age at primary VPI surgery, gender, United States geographic region, type of VPI surgery, provider specialty (plastic/maxillofacial surgery, otolaryngology, dentistry, and other), secondary VPI or palatal revision surgery, and underlying diagnosis as determined by ICD-9 and ICD-10 diagnosis codes associated with the surgical encounter. Non-mutually exclusive diagnostic categories included: congenital oropharyngeal anomalies (e.g. congenital VPI); cleft lip and/or palate; syndromic conditions (e.g. Velocardiofacial syndrome [VCFS], DiGeorge’s syndrome, Pierre Robin Sequence); anomalies of the jaw; lesion, wound, or non-neoplastic disease of the oropharynx; neoplasm of the head and neck; speech disorder; cognitive impairment and/or developmental delay; and neuromuscular disorder. Patients who were diagnosed with a postoperative fistula and/or site dehiscence within 5 years of VPI surgery were also identified by ICD diagnosis code (See Table, Supplemental Digital Content 2 for all ICD coding schemes. ICD-9 and ICD-10 codes reproduced in this table were utilized to group patients into diagnostic categories, and identify patients with outcomes of interest, e.g. obstructive sleep apnea, fistula, and/or dehiscence).
Statistical Analysis
Chi-squared tests and t-tests were used for univariable analysis of predictor variables by procedure type, and development of postoperative OSA and palate fistula and/or surgical site wound dehiscence. In instances in which cell numbers were less than 5 and/or the data was not normally distributed, Fisher’s exact tests and Wilcoxon rank sum tests were utilized in place of chi-squared and t-tests, respectively. Multivariable logistic regression modeled predictors of OSA and revision surgery, using a parsimonious approach of predictor variables based on factors found to be significant on univariable analysis and/or clinically relevant variables (i.e. known predictors). Each diagnosis was coded as an independent variable in the regressions. Subgroup univariable and multivariable analysis was additionally performed for the cleft lip/palate population. Logistic model fitness was assessed using the c-statistic. P-values of less than 0.05 were considered significant. All analyses were conducted using Stata/SE Version 15.0 (StataCorp LLC, College Station, TX). All tabulated data less than 10 were omitted due to the risk of individual identification.
Results
The sample included 1,098 patients who underwent VPI surgery (Table 1). Average age was 6.3 ± 4.0 years and 51.7% of patients were male. Underlying diagnoses were predominantly cleft lip and/or palate (52.8%) and/or congenital oropharyngeal anomalies (42.6%). The most common VPI surgery performed was the pharyngeal flap (61.0%), followed by sphincter pharyngoplasty (22.2%) and palatal lengthening with or without island flaps (16.8%). Fifty-seven patients (5.2%) underwent secondary VPI or revision surgery at a median of 10.4 months (interquartile range [IQR] 17.5, range 0.03–71.6 months) following primary VPI surgery. No significant differences in the type of secondary VPI procedure performed was found with pharyngeal flap and palatal lengthening +/− island flaps as the primary VPI procedure. However, patients who underwent a secondary VPI operation after sphincter pharyngoplasty were significantly more likely to undergo revision sphincter pharyngoplasty (See Table, Supplemental Digital Content 3, which shows Secondary VPI and Revision Surgery by Primary VPI Procedure Type. Primary VPI surgery refers to initial procedure performed for VPI. Secondary VPI surgery refers to revision surgery to address persistent or recurrent VPI following primary VPI surgery. VPI, velopharyngeal insufficiency). Twenty-one patients (1.9%) developed a new fistula or dehiscence postoperatively after primary VPI surgery at a median of 21.9 months (N<10; IQR 26.3, range 0.5–49.5 months) for fistula and 2.1 months (N=12; IQR 7.2, range 0.2–56.6 months) for dehiscence; of these, a small minority underwent a secondary VPI or palatal revision procedure.
Table 1.
Demographics of VPI Surgery Cohort (N=1,098)
| Characteristic | N (%) |
|---|---|
| Age, years | |
| 0–2 | 133 (12.11) |
| 3–5 | 443 (40.35) |
| 6–8 | 253 (23.04) |
| 9–11 | 130 (11.84) |
| 12–14 | 74 (6.74) |
| 15–17 | 65 (5.92) |
| Gender | |
| Male | 568 (51.73) |
| Female | 530 (48.27) |
| Region | |
| Northeast | 137 (12.48) |
| North Central | 271 (24.68) |
| South | 460 (41.89) |
| West | 215 (19.58) |
| Unknown | 15 (1.37) |
| Diagnosis* | |
| Cleft lip and/or palate | 580 (52.82) |
| Congenital anomalies of oropharynx | 468 (42.62) |
| Lesion or disease of oropharynx | 140 (12.75) |
| Speech disorder | 111 (10.11) |
| Syndromic | 56 (5.10) |
| Velocardiofacial syndrome | 30 (2.73) |
| DiGeorge’s syndrome | † |
| Congenital anomaly of skull/face | 22 (2.00) |
| Anomaly of jaw | 11 (1.00) |
| Neoplasm of head and neck | † |
| Developmental delay/cognitive impairment | † |
| Neuromuscular disorder | 0 |
| Other | 14 (1.28) |
| Provider Specialty | |
| Plastic/maxillofacial surgery | 607 (55.28) |
| Otolaryngology | 159 (14.48) |
| Dentistry | 30 (2.73) |
| Other | 302 (27.50) |
| VPI Procedure | |
| Palatal lengthening +/− island flap(s) | 184 (16.76) |
| Sphincter pharyngoplasty | 244 (22.22) |
| Pharyngeal flap | 670 (61.02) |
| Revision Procedure | |
| Palatal lengthening +/− island flap(s) | † |
| Sphincter pharyngoplasty | 18 (1.64) |
| Pharyngeal flap | 29 (2.64) |
| Revision palatoplasty | 18 (1.64) |
VPI, velopharyngeal insufficiency
Percentages do not total 100% due to possibility of multiple diagnoses
Exact value excluded due to cell size <10 to protect patient confidentiality
Eighty patients (7.3%) were diagnosed with OSA (Figure 1). The median time from VPI surgery to OSA diagnosis was 10.2 months (IQR 19.3, range 0.10–104.9 months). The distribution of months until OSA diagnosis is illustrated in Figure 2. As demonstrated in Table 2, on univariable analysis, OSA was more common following sphincter pharyngoplasty (11.1%) than pharyngeal flap (7.2%) or palatal lengthening with or without island flaps (p=0.003). Secondary VPI or revision procedures were least likely following pharyngeal flap (3.6%) compared to sphincter pharyngoplasty (8.2%) or palatal lengthening (7.1%; p=0.009). Procedure type varied significantly by age, with the majority of palatal lengthening procedures performed in younger age groups (0–5 years) compared to sphincter pharyngoplasties and pharyngeal flaps in older age groups (3–8 years, p<0.001). Diagnoses also varied by procedure type, including proportionately more patients undergoing palatal lengthening procedures having a diagnosis of cleft lip and/or palate (p<0.001), compared to higher proportions of underlying diagnoses of congenital oropharyngeal anomalies (p<0.001), speech disorder (p=0.008), and head and neck neoplasms (p=0.012) with other procedures.
Figure 1. Flowchart of Database Analysis and Cohort Selection.
Dx, diagnosis. OSA, obstructive sleep apnea. S/p, status post. VPI, velopharyngeal insufficiency. Computer graphic reproduced and modified with permission from Easel.ly.
Figure 2.
Distribution of Months from Velopharyngeal Insufficiency Surgery to Diagnosis of Obstructive Sleep Apnea (OSA).
Table 2.
Comparison by Procedure Type, All Patients (N=1,098)
| Characteristic | Sphincter, N=244 (%) | Pharyngeal Flap, N=670 (%) | Lengthening, N=184 (%) | p-value |
|---|---|---|---|---|
| Age, years | <0.001 | |||
| 0–2 | 14 (5.74) | 35 (5.22) | 84 (45.65) | |
| 3–5 | 90 (36.89) | 297 (44.33) | 56 (30.43) | |
| 6–8 | 65 (26.64) | 170 (25.37) | 18 (9.78) | |
| 9–11 | 32 (13.11) | 85 (12.69) | 13 (7.07) | |
| 12–14 | 23 (9.43) | 45 (6.72) | † | |
| 15–17 | 20 (8.20) | 38 (5.67) | † | |
| Gender | ||||
| Male | 124 (50.82) | 347 (51.79) | 97 (52.72) | 0.926 |
| Female | 120 (49.18) | 323 (48.21) | 87 (47.28) | |
| Region | <0.001 | |||
| Northeast | 17 (6.97) | 109 (16.27) | 11 (5.98) | |
| North Central | 65 (26.64) | 163 (24.33) | 43 (23.37) | |
| South | 113 (46.31) | 291 (43.43) | 56 (30.43) | |
| West | 47 (19.26) | 94 (14.03) | 74 (40.22) | |
| Unknown | † | 13 (1.94) | 0 (0) | |
| Diagnosis* | ||||
| Cleft lip and/or palate | 88 (36.07) | 340 (50.75) | 152 (82.61) | <0.001 |
| Congenital anomalies of oropharynx | 139 (56.97) | 301 (44.93) | 28 (15.22) | <0.001 |
| Lesion or disease of oropharynx | 39 (15.98) | 81 (12.09) | 20 (10.87) | 0.208 |
| Speech disorder | 18 (7.38) | 84 (12.54) | 11 (5.98) | 0.008 |
| Syndromic | † | 42 (6.27) | † | 0.081 |
| Anomaly of jaw | † | † | † | 0.254 |
| Neoplasm of head and neck | † | † | 0 (0) | 0.012 |
| Other | † | † | † | 0.009 |
| Provider specialty | <0.001 | |||
| Plastic/maxillofacial surgery | 138 (56.56) | 379 (56.57) | 90 (48.91) | |
| Otolaryngology | 40 (16.39) | 107 (15.97) | 12 (6.52) | |
| Dentistry | 0 (0) | 25 (3.73) | † | |
| Other | 66 (27.05) | 159 (23.73) | 77 (41.85) | |
| Postoperative OSA | 27 (11.07) | 48 (7.16) | † | 0.003 |
| Postoperative fistula | † | † | † | 0.355 |
| Postoperative dehiscence | † | † | 0 (0) | 0.157 |
| Revision procedure(s) | 20 (8.20) | 24 (3.58) | 13 (7.07) | 0.009 |
OSA, obstructive sleep apnea
Percentages do not total 100% due to possibility of multiple diagnoses
Exact value excluded due to cell size <10 to protect patient confidentiality
As shown in Table 3, on multivariable regression, significant predictors of postoperative OSA included younger age (odds ratio [OR] 0.91, 95% confidence interval [CI] 0.85 – 0.98, p=0.014), neoplasm of the head and neck (OR 13.08, 95% CI 1.80 – 95.14, p=0.011), and VPI procedure. Specifically, a lower rate of OSA following palatal lengthening with or without island flaps was observed compared to sphincter pharyngoplasty (OR 0.25, 95% CI 0.09 – 0.74, p=0.012). Model p-value was 0.005 with a c-statistic of 0.70. The only significant predictor of secondary VPI or revision procedure(s) was pharyngeal flap, with a lower odds following this procedure compared to sphincter pharyngoplasty (OR 0.43, 95% CI 0.23 – 0.82, p=0.010). Model p-value was 0.012 with a c-statistic of 0.71 (Table 4).
Table 3.
Multivariable Predictors of OSA after VPI Surgery, All Patients
| Characteristic | Odds ratio | 95% confidence interval | p-value |
|---|---|---|---|
| Age | 0.91 | 0.85 – 0.98 | 0.014 |
| Female | 0.96 | 0.60 – 1.54 | 0.877 |
| Region | |||
| North | Ref | - | - |
| North Central | 0.94 | 0.41 – 2.14 | 0.879 |
| South | 1.19 | 0.56 – 2.51 | 0.654 |
| West | 0.80 | 0.31 – 2.07 | 0.639 |
| Diagnosis | |||
| Cleft lip and/or palate | 0.88 | 0.49 – 1.57 | 0.657 |
| Congenital anomalies of oropharynx | 1.83 | 0.97 – 3.46 | 0.061 |
| Lesion or disease of oropharynx | 0.94 | 0.37 – 2.43 | 0.902 |
| Syndromic | 1.84 | 0.73 – 4.64 | 0.199 |
| Neoplasm of head & neck | 13.08 | 1.80 – 95.14 | 0.011 |
| Speech disorder | 0.54 | 0.18 – 1.61 | 0.269 |
| VPI Procedure | |||
| Sphincter pharyngoplasty | Ref | - | - |
| Pharyngeal flap | 0.68 | 0.40 – 1.16 | 0.155 |
| Palatal lengthening +/− island flap(s) | 0.25 | 0.09 – 0.74 | 0.012 |
| Provider specialty | |||
| Plastic/maxillofacial surgery | Ref | - | - |
| Otolaryngology | 0.63 | 0.27 – 1.42 | 0.266 |
| Dentistry | 1.32 | 0.29 – 6.07 | 0.719 |
| Other | 0.89 | 0.50 – 1.58 | 0.694 |
| Postoperative fistula or dehiscence | 0.80 | 0.16 – 4.13 | 0.793 |
OSA, obstructive sleep apnea. Ref, reference. VPI, velopharyngeal insufficiency
Table 4.
Multivariable Predictors of Revision Procedure(s), All Patients
| Characteristic | Odds ratio | 95% confidence interval | p-value |
|---|---|---|---|
| Age | 0.98 | 0.91 – 1.06 | 0.607 |
| Female | 0.64 | 0.37 – 1.12 | 0.122 |
| Region | |||
| North | Ref | - | - |
| North Central | 0.93 | 0.23 – 3.72 | 0.918 |
| South | 3.08 | 0.92 – 10.33 | 0.068 |
| West | 1.60 | 0.41 – 6.21 | 0.494 |
| Diagnosis | |||
| Cleft lip and/or palate | 0.87 | 0.45 – 1.70 | 0.688 |
| Congenital anomalies of oropharynx | 0.94 | 0.49 – 1.77 | 0.838 |
| Syndromic | 1.27 | 0.36 – 4.44 | 0.711 |
| VPI Procedure | |||
| Sphincter pharyngoplasty | Ref | - | - |
| Pharyngeal flap | 0.43 | 0.23 – 0.82 | 0.010 |
| Palatal lengthening +/− island flap(s) | 0.89 | 0.37 – 2.11 | 0.784 |
| Provider specialty | |||
| Plastic/maxillofacial surgery | Ref | - | - |
| Otolaryngology | 0.48 | 0.16 – 1.44 | 0.188 |
| Other | 0.98 | 0.53 – 1.80 | 0.949 |
| Postoperative OSA | 1.33 | 0.54 – 3.32 | 0.530 |
| Postoperative fistula or dehiscence | 1.66 | 0.37 – 7.50 | 0.511 |
OSA, obstructive sleep apnea. Ref, reference. VPI, velopharyngeal insufficiency.
Among the 580 patients with cleft lip and/or palate, 35 (6.0%) were diagnosed with OSA at a median of 9.7 months (IQR 19.3, range 0.1 – 99.6 months) following VPI surgery (Table 5). Compared to the entire sample, this subgroup similarly demonstrated significant variability in underlying diagnosis; among patients in this subgroup with a diagnosis of congenital oropharyngeal anomalies, fewer underwent palatal lengthening (9.2%) compared to sphincter pharyngoplasty (43.2%) or pharyngeal flap (25.9%) (p<0.001). Unique to this subgroup, patients with syndromic cleft lip and/or palate were more likely to undergo pharyngeal flap (p=0.037). On multivariable regression, VPI surgical repair type was not significantly associated with OSA among cleft palate patients (model p-value 0.033, c-statistic 0.71, Table 6).
Table 5.
Comparison by Procedure Type, Cleft Lip/Palate Patients (N=580)
| Characteristic | Sphincter, N=88 (%) | Pharyngeal Flap, N=340 (%) | Lengthening, N=152 (%) | p-value |
|---|---|---|---|---|
| Age, years | <0.001 | |||
| 0–2 | † | 27 (7.94) | 78 (51.32) | |
| 3–5 | 35 (39.77) | 153 (45.00) | 42 (27.63) | |
| 6–8 | 24 (27.27) | 78 (22.94) | 16 (10.53) | |
| 9–11 | 11 (12.50) | 43 (12.65) | † | |
| 12–14 | † | 16 (4.71) | † | |
| 15–17 | † | 23 (6.76) | † | |
| Gender | ||||
| Male | 41 (46.59) | 178 (52.35) | 80 (52.63) | 0.599 |
| Female | 47 (53.41) | 162 (47.65) | 72 (47.37) | |
| Region | <0.001 | |||
| Northeast | † | 64 (18.82) | † | |
| North Central | 23 (26.14) | 70 (20.59) | 34 (22.37) | |
| South | 44 (50.00) | 167 (49.12) | 53 (34.87) | |
| West | 15 (17.05) | 36 (10.59) | 59 (38.82) | |
| Unknown | 0 (0) | † | 0 (0) | |
| Diagnosis | ||||
| Congenital anomalies of oropharynx | 38 (43.18) | 88 (25.88) | 14 (9.21) | <0.001 |
| Lesion or disease of oropharynx | † | 18 (5.29) | 12 (7.89) | 0.502 |
| Speech disorder | † | 27 (7.94) | † | 0.093 |
| Syndromic | † | 15 (4.41) | † | 0.037 |
| Anomaly of jaw | 0 (0) | † | † | 0.500 |
| Provider specialty | <0.001 | |||
| Plastic/maxillofacial surgery | 65 (73.86) | 222 (65.29) | 78 (51.32) | |
| Otolaryngology | † | 30 (8.82) | † | |
| Dentistry | 0 (0) | 16 (4.71) | † | |
| Other | 17 (19.32) | 72 (21.18) | 65 (42.76) | |
| Postoperative OSA | † | 22 (6.47) | † | 0.152 |
| Postoperative fistula | 0 (0) | † | † | 0.217 |
| Postoperative dehiscence | † | † | 0 (0) | 0.437 |
| Revision procedure(s) | † | 18 (5.29) | † | 0.966 |
OSA, obstructive sleep apnea
Exact value excluded due to cell size <10 to protect patient confidentiality
Table 6.
Multivariable Predictors of OSA after VPI Surgery, Cleft Lip/Palate Patients
| Characteristic | Odds ratio | 95% confidence interval | p-value |
|---|---|---|---|
| Age | 0.94 | 0.84 – 1.05 | 0.252 |
| Diagnosis | |||
| Congenital anomalies of oropharynx | 2.92 | 1.41 – 6.06 | 0.004 |
| Syndromic | 1.16 | 0.14 – 9.41 | 0.886 |
| VPI Procedure | |||
| Sphincter pharyngoplasty | Ref | - | - |
| Pharyngeal flap | 0.84 | 0.35 – 2.02 | 0.694 |
| Palatal lengthening +/− island flap(s) | 0.39 | 0.11 – 1.35 | 0.136 |
| Provider specialty | |||
| Plastic/maxillofacial surgery | Ref | - | - |
| Otolaryngology | 0.74 | 0.16 – 3.38 | 0.700 |
| Other | 1.63 | 0.77 – 3.44 | 0.202 |
OSA, obstructive sleep apnea. Ref, reference. VPI, velopharyngeal insufficiency.
Discussion
In our nationwide analysis of 1,098 patients who underwent VPI surgery, we demonstrate no statistically significant difference in the rate of postoperative OSA among sphincter pharyngoplasty (11.1%) compared to pharyngeal flap (7.2%) patients on multivariable analysis. Though only significant on univariable analysis, both of these pharyngeal procedures had a higher rate of postoperative OSA compared to palatal lengthening, as one would expect since the resting velopharyngeal inlet is less narrowed with the latter procedure. Our results suggest that pharyngeal flap and sphincter pharyngoplasty are equivalent for OSA outcomes. This finding contradicts preexisting literature suggesting that OSA is more common following pharyngeal flap compared to sphincter pharyngoplasty.12,13 Additionally, we show that pharyngeal flap patients were less likely to require a second VPI or palatal revision operation compared to those undergoing sphincter pharyngoplasty (OR 0.43, p=0.010). This finding is not likely due to differential rates of fistula or dehiscence, as the distribution of postoperative fistula or dehiscence did not significantly vary by procedure type.
Many retrospective studies have published rates of OSA following pharyngeal flap6,7,16–18 and sphincter pharyngoplasty5,19,20 alone or in comparison to palatal lengthening. However, only a handful of studies over the past three decades have directly compared OSA rates between pharyngoplasty procedures. Early retrospective cohort studies reported higher rates of postoperative OSA following pharyngeal flaps,12,13 which met statistical significance in a more recent study of 39 cleft patients.21 The retrospective design and small cohort sizes of these studies limit the strength of their conclusions, suggesting a need to review higher level evidence. Randomized controlled trials by Abyholm et al.14 and Ysunza et al.22 both compare rates of OSA following VPI surgery; the results are combined in Collins et al.’s meta-analysis that presents a collective 133 patients.23 With follow up times of 3–4 months, Collins et al. reported a marginally higher incidence of OSA after pharyngeal flap compared to sphincter pharyngoplasty, without any conclusions about statistical significance.23 More recently, de Blacam et al. performed a systematic review of surgery for VPI, including 83 studies and 4,011 patients; only 42 (50.6%) studies reported OSA outcomes, with a finding of no statistically significant variation in OSA rate or need for further surgery among procedure types.24 Five studies with a collective sample size of 40 patients included more than one VPI procedure type; the remainder reported outcomes for a single type of procedure. Among these 40 patients, multiple VPI procedures may have been performed by the same surgeon, and thus surgeon-specific factors could be controlled but none were reported. Similarly, our analysis cannot control for surgeon-specific factors or other factors impacting the decision for sphincter pharyngoplasty versus pharyngeal flap.
In much of the literature on VPI surgical outcomes, speech defines the success of VPI surgery. With OSA relatively out of focus, there is scant information on predictors of postoperative OSA in the literature. In a retrospective study of 58 patients undergoing sphincter pharyngoplasty, Witt et al. observed that patients with microretrognathia, perinatal respiratory dysfunction, younger age, and upper respiratory infection appeared to be at increased risk for acute postoperative OSA.25 Ettinger et al.’s retrospective analysis adds prior tonsillectomy and adenoidectomy to this list of risk factors (relative risk 2.4, p=0.04), which the authors theorize is secondary to palatopharyngeal muscle scarring.1 Besides procedure type, we identify younger age (OR 0.91, p=0.014) as a protective factor; this is in contrast to Witt et al.’s observation, yet consistent with prior findings of increasing age as a risk factor for OSA in the pediatric and adult population.26,27 We also show that head and neck neoplasm is a risk factor for postoperative OSA (OR 13.08, p=0.011). Though relatively rare in children, head and neck cancer and its treatment are known risk factors for OSA due to changes to pharyngeal musculature, and chronic edema and fibrosis secondary to radiation therapy.28
The cleft palate population is central to any discussion of VPI, as approximately 20% of cleft palate patients may require surgery for VPI following palate repair.29,30 This rate more than doubles for cleft palate patients that develop a palatal fistula.30 Indeed, the majority of patients in our sample had a diagnosis of cleft lip and/or palate. Cleft patients may be at increased risk of OSA due to abnormal dimensions of the maxilla and mandible, a small nasopharyngeal space, and scarring from prior procedures.31,32 However, our results demonstrate that cleft lip/palate is not significantly predictive of postoperative OSA. Furthermore, we found no significant association between VPI procedure and postoperative OSA. In a study of middle-aged adult patients with a history of repaired cleft palate, approximately half had undergone subsequent pharyngeal flap surgery with no significant difference in the rate of OSA between patients with and without a pharyngeal flap.17 This study suggests that aforementioned underlying congenital airway abnormalities may be the culprit, which is reflected in our analysis: a diagnosis of congenital anomaly of the oropharynx was a predictive variable for postoperative OSA in the cleft lip/palate population (OR 2.92, p=0.004). We hypothesize that the severity of the cleft is also related, though this cannot be captured with our methodology.
Syndromic patients are known to be at high risk for OSA due to both structural and neuromuscular oropharyngeal abnormalities. VCFS patients can have functional hypotonia of the palate and upper airway, in addition to larger velopharyngeal gap sizes that require obturation with a wider pharyngeal flap.33–35 Due to the severity of the palatopharyngeal disproportion, some authors propose predominant use of pharyngeal flaps in the VCFS population.36,37 Our analysis reflects this decision, showing that a higher proportion of patients undergoing pharyngeal flap surgery were syndromic cleft patients (4.4%) compared to sphincter pharyngoplasty or palatoplasty, without any demonstrated impact on the incidence of postoperative OSA.
Beyond risk of postoperative OSA, predicted speech outcome plays a critical role in selection of surgical technique. One school of thought is that technique selection should be based on velopharyngeal gap size and closure pattern; some authors advocate for use of pharyngeal flaps in patients with adequate lateral wall motion and a circular or sagittal closure pattern, versus sphincter pharyngoplasty for patients with coronal patterns.2,9,38 Though this mantra widely guides treatment, prospective RCTs have demonstrated no significant differences in speech outcomes.14,22 The width of the pharyngeal flap and level of inset may be adjusted to accommodate coronal closure patterns.7,39 The skill and comfort level of the surgeon may also influence outcomes.7,40 Thus, we suggest that selection of surgical technique should not be based solely on velopharyngeal anatomy; in light of evidence questioning its relevance to speech outcome, we advocate for consideration of other factors, such as likelihood of postoperative OSA and need for surgical revision. In our analysis, the equivalence of the pharyngeal flap for OSA outcomes and more favorable risk profile for revision surgery suggests that this procedure should be given serious consideration during surgical planning, irrespective of velopharyngeal anatomy.
Over the past several years, the senior author’s practice has shifted away from performing sphincter pharyngoplasties to instead tailoring the pharyngeal flap to meet the anatomical and functional needs of the cleft patient. If the patient presents with a small velopharyngeal port gap during speech by nasoendoscopy, or malpositioned levator veli palatini muscles (e.g. as evidenced by arching of the palate with phonation and inverted v-deformity of the muscle bulge), and has not had a Furlow palatoplasty for primary cleft palate repair, then palatal lengthening is often the first-line VPI surgery pursued. In other cases, and particularly with VCFS and neuromuscular disorders, we have found that a superiorly-based pharyngeal flap can be customized to meet the needs of most patients, irrespective of closure pattern. Our team practice is to obtain a baseline polysomnogram when pre-existing OSA is suspected, which can impact our decision on flap size with smaller flaps performed when OSA is present. We have observed a very low incidence of persistent OSA following pharyngeal flap procedures irrespective of flap width, which is consistent with prior studies demonstrating no association between pharyngeal flap width and postoperative OSA.37,41 In the event that OSA or hyponasality occur following pharyngeal flap and fail to resolve, Katzel et al. have shown that surgical takedown performed 5 or more years after the initial surgery can address these issues without compromising speech outcomes.42
OSA following VPI surgery may be transient, yet this study is unable to distinguish between temporary and persistent symptomatology. Swelling of the upper airway is likely to cause some degree of acute OSA in the majority of patients,43 though this is only clinically significant if it fails to resolve. Edema and associated symptoms of OSA can resolve within days,25 or more commonly by 3 to 6 months.43,44 Our study identifies OSA in the postoperative patient by the initial appearance of an ICD code signifying OSA, but cannot detect whether this condition resolved. Our results may thus overestimate the incidence of postoperative OSA, either due to over-coding the OSA diagnosis or failing to exclude transient cases, though this should apply indiscriminately to all procedure types. Alternatively, we may underestimate the incidence of OSA, in addition to the diagnoses of fistula and dehiscence, if these diagnoses are inadequately documented in administrative records.
Additional limitations are related to the retrospective design with utilization of an administrative claims database, which collectively introduces possible selection bias, confounding errors, and coding errors. Although the database has large numbers, the data can only be as specific as ICD and CPT codes on which the claims are based. Thus, we are lacking detail on factors such as velopharyngeal gap size and closure pattern, cleft morphology, speech outcomes, surgeon skill, and clinical significance of OSA (i.e. whether any treatment, such as positive pressure ventilation, was required). In addition, we cannot determine the method of OSA diagnosis. Polysomnography is the diagnostic gold standard, yet symptoms are commonly used for diagnosis despite being poor predictors of OSA.45 This is a ubiquitous problem, as the majority of studies of OSA following VPI surgery do not base the diagnosis on polysomnography.24 Furthermore, a lack of inquiry regarding sleep on the part of the physician may lead to underreporting of symptoms that should trigger polysomnography evaluation. Future studies should strive for consistency in OSA diagnosis with consistent questions about nocturnal breathing and routine use of polysomnography. If polysomnography is too resource- and time-intensive, then the use of validated questionnaires that are expeditious and sensitive assessments of OSA can be utilized.24,46–48
Conclusions
Among cleft and non-cleft patients undergoing surgery for VPI in this national database analysis, the rate of OSA following VPI surgery was not significantly different for pharyngeal flap compared to sphincter pharyngoplasty. Despite prior published reports of lower rates of OSA following sphincter pharyngoplasty, this study showed equivalence among a population of patients with and without cleft lip and palate, and thus craniofacial surgeons should not be dissuaded from this procedure for fear of OSA. This study is not without the inherent limitations of large administrative database analysis. However, our findings of no significant difference in postoperative OSA rates, but a significantly higher rate of secondary VPI and palatal revision surgery following sphincter pharyngoplasty, suggest a need for surgeons to consider these factors when choosing a surgical method to address VPI.
Supplementary Material
Supplemental Digital Content 1. See Table, which shows a list of Current Procedural Terminology (CPT) Codes. The CPT codes reproduced in this table were utilized to identify patients who underwent primary and/or secondary surgery for velopharyngeal insufficiency, and/or revision palatal surgery.
Supplemental Digital Content 2. See Table, which shows the list of International Classification of Diseases, Ninth Revision (ICD-9) and Tenth Revision (ICD-10) Diagnosis Codes. ICD-9 and ICD-10 codes reproduced in this table were utilized to group patients into diagnostic categories, and identify patients with outcomes of interest (e.g. obstructive sleep apnea, fistula, and/or dehiscence). *Includes all ICD codes within category. †Includes all ICD codes within category, with exception of isolated codes that have been extracted and are included in an alternative grouping.
Supplemental Digital Content 3. See Table, which shows Secondary VPI and Revision Surgery by Primary VPI Procedure Type. Primary VPI surgery refers to initial procedure performed for VPI. Secondary VPI surgery refers to revision surgery to address persistent or recurrent VPI following primary VPI surgery. VPI, velopharyngeal insufficiency. *Significant difference (p<0.001) among the type of secondary VPI procedure. †Excluded due to cell size ≤ 10 to protect patient confidentiality.
Acknowledgements:
Data for this project were accessed using the Stanford Center for Population Health Sciences Data Core. The PHS Data Core is supported by a National Institutes of Health National Center for Advancing Translational Science Clinical and Translational Science Award (UL1 TR001085) and from Internal Stanford funding. The content is solely the responsibility of the authors and does not necessarily represent the official views of the NIH.
Financial Disclosure Statement:
No commercial associations or financial disclosures for any author. No funding was received for this work.
Footnotes
IRB Approval: Studies using deidentified datasets that are hosted by the Stanford Center for Population Health Sciences (PHS) are covered under the PHS umbrella IRB 40974. Individual IRB approval was not required for this study as it does not involve human subjects research.
Accepted for presentation: American Association of Plastic Surgeons (AAPS) 100th Annual Meeting, Miami, FL (May 2021)
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Associated Data
This section collects any data citations, data availability statements, or supplementary materials included in this article.
Supplementary Materials
Supplemental Digital Content 1. See Table, which shows a list of Current Procedural Terminology (CPT) Codes. The CPT codes reproduced in this table were utilized to identify patients who underwent primary and/or secondary surgery for velopharyngeal insufficiency, and/or revision palatal surgery.
Supplemental Digital Content 2. See Table, which shows the list of International Classification of Diseases, Ninth Revision (ICD-9) and Tenth Revision (ICD-10) Diagnosis Codes. ICD-9 and ICD-10 codes reproduced in this table were utilized to group patients into diagnostic categories, and identify patients with outcomes of interest (e.g. obstructive sleep apnea, fistula, and/or dehiscence). *Includes all ICD codes within category. †Includes all ICD codes within category, with exception of isolated codes that have been extracted and are included in an alternative grouping.
Supplemental Digital Content 3. See Table, which shows Secondary VPI and Revision Surgery by Primary VPI Procedure Type. Primary VPI surgery refers to initial procedure performed for VPI. Secondary VPI surgery refers to revision surgery to address persistent or recurrent VPI following primary VPI surgery. VPI, velopharyngeal insufficiency. *Significant difference (p<0.001) among the type of secondary VPI procedure. †Excluded due to cell size ≤ 10 to protect patient confidentiality.