Abstract
Introduction
Velopharyngeal insufficiency (VPI) is an unfavorable outcome of primary palatoplasty. Clinical and surgical risk factors for VPI are well described, yet the impact of social determinants of health (SDoH) on VPI is less understood. This study aimed to estimate the effect of income, ethnicity, and geographical location on VPI.
Methods
Non-syndromic patients who had primary palatoplasty at British Columbia Children's Hospital between 2005 and 2015 were retrospectively reviewed. Patient demographics, including income, ethnicity, and distance to hospital were collected, as well as primary palatoplasty details, VPI diagnosis, and any secondary speech surgery. Logistic regression models were used to examine associations with VPI.
Results
A total of 209 patients were included: 30% (n = 63) developed VPI. Complete cleft palate was a significant predictor of VPI (odds ratio (OR) 4.35, P ≤ .001). Indigenous identity, sex assigned at birth, average income, and distance to the hospital were not predictors of VPI. For those with complete cleft palate, there was a 24% greater likelihood of developing VPI for every month older the patient was at the time of initial palate repair (OR 1.27, P = .018).
Conclusions
Cleft palate severity and age at primary palatoplasty for patients with complete cleft palate had the largest effect on VPI. SDoH factors (income and geographical location) were not associated with VPI; however, there was a trend between Indigenous identity and the development of VPI. The effect of Indigenous identity was limited by incomplete data and may be an area of future research. These findings are reassuring for a multidisciplinary clinic with dedicated social work support in a public healthcare system.
Introduction
Cleft palate is one of the most common congenital craniofacial anomalies, which results from the failure of midline fusion of the palatal shelves. 1 A cleft palate is surgically repaired to enable normal maxillary growth, feeding, appearance, hearing, and speech. However, for some patients, the initial palatoplasty does not provide adequate velopharyngeal function. In these cases, the palate is unable to close off the nasopharynx from the oropharynx resulting in velopharyngeal insufficiency (VPI), a diagnosis characterized by hypernasal speech. VPI occurs in up to 40% of children with cleft palate, and most will undergo secondary surgical procedures. 2 A better understanding of the factors that contribute to VPI can inform strategies to lower the rate of occurrence.
To date, research on risk factors for VPI surgery has mainly focused on patient characteristics as well as surgical timing, technique, and surgeon experience. A systematic review found VPI to be strongly associated with Veau IV clefts, syndromes, and Pierre Robin Sequence, moderately associated with 22q11.2, and not associated with biological sex. 3 Moreover, younger patient age (<9 months) at the time of initial cleft palate repair has been correlated with a higher rate of secondary surgery.4,5 Although rates of VPI vary by individual surgeon, surgeon experience (ie, frequency of palatoplasties) has been found to be independent of VPI, and there are mixed results on the role and effect of surgical technique.4,6–8
Social determinants of health (SDoH) are non-medical factors (ie, geographic home, economic stability, ethnicity, and social supports) that can influence a patient's overall health and treatment outcomes.9–12 Previous research has identified that low socioeconomic status (SES) leads to poorer health outcomes, specifically pertaining to orofacial clefts 13 ; however, there are few reports on the association of SDoH and VPI. 3 Previous studies have found that patients from a lower SES have more missed appointments 14 and later primary palatoplasty repairs.15,16 Furthermore, patient ethnicity was also shown to be associated with older age at primary cleft palate repair resulting in increased rates of secondary palate surgery for VPI or fistula.4,15,17 Choa et al (2014) and Sitzman et al (2017) found no associations between patients’ ethnicity or deprivation (defined by low social class, lack of car ownership, overcrowding and male unemployment in UK patients, via Carstairs index) and speech outcomes following primary palatoplasty, while de Araújo et al (2022) found patients with lower SES or those who lived further away from the hospital center had poorer speech outcomes.6,16,18
Although there is limited and conflicting evidence on the effect of SDoH on speech outcomes after palatoplasty, this has not been studied in Canada, which has one of the highest rates of orofacial cleft in the world (∼1:790 live births). 19 A better understanding of SDoH and speech outcomes following initial palate repair may be particularly relevant for patients attending the British Columbia Children's Hospital (BCCH) Cleft Palate—Craniofacial program as previous research has shown approximately one in two families struggle financially or do not have adequate social support, and a third lack a primary healthcare provider. 20 Moreover, nearly 25% of pediatric patients in BC live in remote regions, which can limit access to specialized care. 21 Therefore, this study aimed to estimate the associations between SDoH factors (income, geographic location, and ethnicity) on the development of VPI in non-syndromic children with cleft palate with or without cleft lip. The findings from this study may highlight a need to address health inequities and resource allocation related to VPI for children living in Canada.
Methods
This study was designed as a retrospective chart review of patients who had undergone a primary palatoplasty between January 1, 2005 and December 31, 2015 at a single institution. This study was approved by the University of British Columbia Children's & Women's Research Ethics Board (H23-03846). A waiver of consent was obtained to access patient records for data collection.
Study Population
Eligible patients were identified from the BCCH Cleft Palate—Craniofacial Clinic database. Patients who underwent a primary palatoplasty at BCCH and completed their follow-up in BC were included. Exclusion criteria were: primary palatoplasty not completed at BCCH as well as patients with incomplete follow-up. Patients with known syndromes affecting palate and speech development such as Pierre Robin sequence, 22q11.2 deletion syndrome and Stickler syndrome were also excluded.
Data Collection
Data was collected on predetermined variables in three main domains: patient demographics, surgical factors, and speech outcomes with the primary outcome being the development of VPI.
Patient Demographics
Patient demographic variables consisted of sex assigned at birth, age at primary palatoplasty, patient's home health authority, shortest driving distance to the hospital, income based on postal code, and Indigenous identity. Median after-tax income of households in 2015 by postal code was collected based on Statistics Canada reports for the 2016 Census. 22 Ethnicity was collected from patient charts, and Indigenous identity (including First Nations, Métis, Inuit, and others) was self-reported by the family and then indicated by pediatrician notes, social worker notes, or through the involvement of an Indigenous care coordinator.
Primary Palatoplasty
Surgical factors consisted of cleft palate severity (complete vs incomplete cleft palate), age at primary palatoplasty, type of cleft palate repair, early and late complications of surgery, and the frequency and type of secondary speech surgeries performed. Cleft palate severity was defined as complete or incomplete, with complete cleft palate involving both the hard and soft palate. Post-operative complications were collected and defined as early (eg, infection, bleeding) or late (eg, dehiscence, fistula, and obstructive sleep apnea). Unrepaired alveolar cleft was excluded from the definition of a fistula.
Speech Outcomes
Secondary speech outcome variables consisted of the development of VPI including the velopharyngeal port gap size and closure pattern based on nasoendoscopy or videofluoroscopy, and if speech therapy was received. The development of VPI was determined based on SLP perceptual speech assessment, clinical notes, nasoendoscopy, and/or videofluoroscopy closest to 18 months post-palatoplasty.
Data Analysis
Descriptive statistics were performed for the various variables, including cleft palate severity, sex assigned at birth, age at primary palate surgery, shortest driving distance to the hospital, median yearly income, and Indigenous identity. Categorical variables were reported as frequencies and percentages. Continuous variables were reported as medians with interquartile ranges (IQRs) due to their skewed distributions. Logistic regression models were then used to examine associations with VPI, with multivariable analyses reporting odds ratios (ORs), 95% confidence intervals (CIs), and P-values. A P-value of less than .05 was considered statistically significant. Subgroup analyses were conducted separately for patients with complete and incomplete cleft palates. The effect of driving distance, income, and Indigenous identity on the timing of primary palatoplasty was analyzed using linear regression. All analyses were conducted using R Statistical Software version 4.4.1. 23
Results
Patient Demographics
A total of 258 patients were screened for inclusion. Forty-nine were excluded for the following reasons: primary palatoplasty not completed at our center (n = 25), presence of a syndrome (n = 6), submucous cleft palate (n = 6), insufficient documentation or data (n = 9), no cleft palate (n = 1), primary palate repair occurred before study period (n = 1), or lost to follow-up (n = 1, moved out of province after primary palate surgery). Therefore, 209 patients (female n = 89 and male n = 120) met inclusion criteria. Notably, one patient moved during their period of active care but moved back to BC where follow-up was provided. This patient was included in the study. The median age at primary palatoplasty for all patients was 10 months, as seen in Table 1. Overall, 131 patients (63%) had a complete cleft palate. The median driving distance for patients to reach the hospital was 50 km. The majority of patients were from the Fraser Health or Vancouver Coastal Health Authority, also seen in Table 1. The median after-tax income of households based on postal code was $62,589 CAD. Ethnicity data was not available for 164 (78%) of the study population. A total of 24 patients were Indigenous (11%). Of the 24 patients who were Indigenous, 19 had a complete cleft palate (79%). The median length of follow-up for the study was 4.3 years of age, IQR (2.9, 5.9), and clinically patients are followed until skeletal maturity.
Demographic Data, Cleft Diagnosis, and Primary Palatoplasty Details for All Patients (n = 209).
Categorical data are n (%), continuous data are median [IQR].
Other early complications include diarrhea, fever, lethargy and dehydration, poor fluid intake, and post-operative nasal regurgitation (one case each for a total of five “other” early complications).
Abbreviations: ICU, intensive care unit; CAD, Canadian dollars; km, kilometers; IQR, interquartile range.
Just over a quarter of patients (n = 57, 27%) were seen by social work and received support(s). Psychosocial support was the most common (n = 43/57, 75%), followed by travel/accommodation/parking (n = 9/57, 16%), food (n = 6/57, 11%), or advocating for speech therapy or genetic counseling (n = 3/57, 5%).
Primary Palatoplasty
As noted in Table 1, 76% of all patients had a hard palate repair, the most frequent technique was Veau/2-flap repair (n = 136, 86%), followed by Von Langenbeck repair (n = 21 13%) and hybrid (n = 1, 1%), respectively. All patients had soft palate repair with 160 (77%) having an intravelar veloplasty and 49 (23%) having a Furlow repair. Twenty patients (10%) had one or more early complications, and 25 patients (12%) had one or more late complications, as seen in Table 1. The age of a patient at primary palatoplasty was associated with Indigenous ethnicity (P = .004), and not associated with median driving distance to the hospital (P = .720) or income (P = .485), Supplemental Table 1.
Speech Outcomes
Overall, 63 patients (30%) developed VPI, Table 2. The median age at VPI diagnosis was 4.14 years, Table 3. The velopharyngeal closure pattern was identified for 55 of the 63 patients with VPI. Of these, 36 (64%) patients had a coronal closure pattern. For the eight patients without closure pattern data, one never had surgery, and seven either did not have a successful nasoendoscopy or no clear closure pattern was discernable. A higher rate of VPI was found in patients with a complete cleft palate compared to those with incomplete cleft palate (40% vs 13%), Table 4. Indigenous identity was not statistically significant (OR 2.15, CI: 0.46 to 11.4, P = .3) as a predictor for VPI, but had a wide CI making interpretation challenging. Sex assigned at birth, shortest driving distance to BCCH, and average yearly income based on postal code was not associated with the development of VPI (Table 4). Upon subgroup analysis by cleft severity, seen in Table 4, in patients with incomplete cleft palate, greater driving distance to BCCH was associated with higher odds of VPI. For each additional 100 km of driving distance, the odds of VPI increased by 18% (OR:1.18, P = .033). A complete cleft palate and older age at the time of primary palatoplasty were statistically significant predictors for the development of VPI. We further analyzed the timing/age at which primary palatoplasty was performed. As detailed previously, the mean age at primary palatoplasty was 10 months for the cohort; however, for each additional month primary palatoplasty was delayed from the mean, the odds of developing VPI increased by 27% (OR: 1.27, P = .018). For those with an incomplete cleft palate, older age at surgery did not impact speech outcomes. Of the 63 patients who had a diagnosis of VPI, 62 (98%) went on to have secondary speech surgery. One patient had no desire for further investigation or intervention. The majority of patients had a pharyngeal flap (52%), Table 3.
Speech Details Post-Primary Palatoplasty for All Patients (n = 209).
One patient had no desire for further investigation or intervention.
Abbreviations: VPI, velopharyngeal insufficiency.
Speech Details Post-Primary Palatoplasty for Patients with VPI Diagnosis (n = 63).
Categorical data are n (%), continuous data are median [IQR].
One patient received part of their treatment in another province.
The nasoendoscopy was not successful or no clear closure pattern was discernable.
One patient had no desire for further investigation or intervention.
Abbreviations: VPI, velopharyngeal insufficiency; IQR, interquartile range.
Predictive Variables for VPI.
Categorical data are n/row total (rates %). Odds ratios, confidence intervals, and P-values were calculated using multivariable logistic regression. Statistically significant P-values are bolded.
Abbreviations: VPI, velopharyngeal insufficiency; OR, odds ratio; CI, confidence interval.
Discussion
Overall, 30% (n = 63) of patients developed VPI, and of those patients, 98% (n = 62) underwent a secondary surgery for speech. This is consistent with previous studies addressing the rate of VPI development after palaptoplasty.2,24 SES, geographical location, and Indigenous identity were not associated with the development of VPI; however, complete cleft palate and older age at the time of complete cleft palate repair were associated with the development of VPI.
Social Determinants of Health
Our study found no association between ethnicity, SES, and geographical location with the development of VPI. This finding corroborates the results of Choa et al (2014) and Sitzman et al (2017), and was in contrast to de Araújo et al (2022) who found that patients’ home geographic region and SES were associated with poor speech outcomes.6,16,18 This difference may be explained by de Araújo et al (2022) finding that geography and SES were also associated with an older age at primary palate repair (≥3 years of age), whereas in our study population, geographic location and income were not associated with age at initial palate repair. 16 Although there was a statistically significant association between Indigenous identity and age of primary palatoplasty, this analysis was limited by missing data (no ethnicity data for 78% of study population). Our study found there was an association for patients with incomplete cleft palate and driving distance from BCCH. Where for each 100 km the odds of VPI increased by 18%. However, this association was only seen in the incomplete cleft group. There were 10 patients from the entire study population (5%) who developed VPI and had an incomplete cleft palate. Therefore, it is hard to draw clinical conclusions about driving distance based on the small population. Nonetheless, it's possible our sample size was not adequately powered to show the true effect that geographical location has on speech outcomes. Patients living in more remote communities often face barriers such as travel costs, lack of information, limited access to hospitals, and less financial support. 16 This study did not look specifically at how many patients moved within BC during the study period and how the resources available may vary based on geographical location. There was one patient who was excluded from the study due to incomplete follow-up and 9 patients excluded due to insufficient data. It is possible this could be associated with SDoH factors, but no concrete conclusions can be made. Additionally, people with lower SES may face barriers to accessing care due to taking time off work and out-of-pocket costs for private speech therapy. Even though these barriers exist, our clinic may help patients reduce or overcome them with social work support, as 27% of patients were seen by social work, which may be an alternative explanation for our null findings.
Indigenous identity was also not associated with the development of VPI, although there was a trend toward statistical significance, suggesting this relationship warrants further exploration. Due to the wide CI, the direction of the trend could change with more complete data. One possible explanation for the potential relationship between Indigenous identity and VPI was the significantly higher rate of complete cleft palate in patients who identified as Indigenous and the significant association of complete cleft palate with the development of VPI seen in our study. Moreover, it is possible that children who are Indigenous have increased barriers to accessing speech therapy; it is well documented that First Nations, Metis and Inuit have experienced and continue to experience structural and systemic racism that impedes their access to equitable healthcare. 25 Importantly, analyses related to ethnicity were limited, as the majority of records (78%) did not describe a patient's self-identified ethnicity as this was not routinely collected during the time period of the study. Although patients were seen in follow-up for a median of 4.3 years of age and clinically followed to skeletal maturity, no additional information about ethnicity was gathered throughout this time.
Cleft Severity and Surgery
Patients with complete cleft palate were more than twice as likely to develop VPI compared to those with an incomplete cleft palate. This study did not analyze if certain palatoplasty techniques were more likely to lead to VPI; however, it is well known that cleft severity often drives choice of technique, and there remain conflicting findings on the advantages of one technique over another.7,8
Timing of Surgery
Of those patients with a complete cleft palate, for each additional month of age at which palatoplasty was performed, the chance of developing VPI increased by 24%. The impact of age at primary palatoplasty was not seen in patients with an incomplete cleft palate. This may be clinically relevant as it is important to deliver timely care to all patients with cleft palate, but with a special consideration on the severity of the cleft. Performing primary palatoplasty early for those with a complete cleft palate could reduce the number of patients that develop VPI, which could help to improve speech outcomes. Other studies have found that performing surgery earlier (at < 24 months old, or at 12 months vs 36 months) results in significantly better articulation.26,27 In our study, only one patient had primary palatoplasty before 9 months of age (8 months at time of surgery), so we were unable to directly compare our results to Sitzman et al (2017) finding that surgery before 9 months of age results in a higher rate of secondary surgery. 6
Limitations
This study was limited by some incomplete data. Specifically, we had challenges with collecting the ethnicity of patients. For 164 (78%) patients, there was no data on ethnicity. In our local healthcare system, documenting patients’ self-reported ethnicity was not routinely collected during the time period of this study. Therefore, it cannot be determined if all Indigenous patients were correctly identified and captured. Lastly, as this study was limited to a single institution, it may not be generalizable to other centers.
Conclusions
In summary, cleft palate severity followed by age at primary palatoplasty for patients with complete cleft palate had the largest effect on the development of VPI. SDoH (income, geographic location, and ethnicity) did not impact the rate of VPI for a Canadian population of non-syndromic children with cleft palate with or without cleft lip. These results are reassuring within the context of a publicly funded health care system and corroborate studies done in the United Kingdom.6,18 The potential association of Indigenous identity and VPI was limited by incomplete ethnicity data and could be explored in a future study.
Supplemental Material
sj-docx-1-psg-10.1177_22925503261446316 - Supplemental material for Predictors of Poor Speech Outcomes Following Primary Palatoplasty for Patients with Cleft Palate: Do Social Determinants of Health Impact Velopharyngeal Insufficiency?
Supplemental material, sj-docx-1-psg-10.1177_22925503261446316 for Predictors of Poor Speech Outcomes Following Primary Palatoplasty for Patients with Cleft Palate: Do Social Determinants of Health Impact Velopharyngeal Insufficiency? by Frances Scheepers, Bhairvi T. Mathur, Sheryl Palm, Travis Gordon, Rebecca Courtemanche, Stephanie Cooper, Sophia Shayan, Ye Shen and Jugpal S. Arneja in Plastic Surgery
Footnotes
Ethical Statement
This project was approved by the University of British Columbia Children's and Women's Research Ethics Board (H23-03846). All procedures followed were in accordance with the ethical standards of the responsible committee on human experimentation (institutional and national) and with the Helsinki Declaration of 1975, as revised in 2008.
Consent to Participate
A waiver of consent was granted from the research ethics board.
Author Contributions
FS, BTM, RC, TG, SP, SC, SS and JSA were responsible for the study conceptualization and design, data interpretation, manuscript writing and editing. YS contributed greatly to data analysis.
Funding
The author(s) disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: for this study, Tara Mathur was awarded a summer studentship from the British Columbia Children's Hospital Office of Pediatric Surgical Evaluation and Innovation; Dr Arneja was supported by a University of British Columbia Clinical Faculty Research Support Award.
Declaration of Conflicting Interests
The authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Study-Related Presentations
Bhairvi Tara Mathur, Frances Scheepers, Sheryl Palm, Rebecca Courtemanche, Travis Gordon, Ye Shen, Jugpal Arneja. Predictors of Poor Speech Outcomes Following Primary Palatoplasty. Western Medical Research Conference. January 16-18, 2025. Carmel, California. Oral Presentation.
Supplemental Material
Supplemental material for this article is available online.
References
Supplementary Material
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