Revealing a Cleft in the Community: A Systematic Review of Sociodemographic Disparities Affecting the Care of Patients With Cleft Lip

Abstract

Introduction:

Comprehensive management of cleft lip with or without cleft palate (CL/P) often begins prenatally, continues into adulthood, and involves multidisciplinary collaboration. This study aims to review the current literature and summarize sociodemographic disparities that impact access to care, management, and postoperative outcomes in patients with CL/P.

Methods:

A systematic review was conducted using Pubmed, Embase, and Medline databases. Studies discussing sociodemographic disparities regarding access to care, management, and treatment outcomes for patients with CL/P were included. Studies performed outside the US and published before the year 2000 were excluded.

Results:

Of 3782 studies initially identified, 26 met the inclusion criteria. Disparities discussed included access to care (n = 11), missed appointments (n = 2), use of nasoalveolar molding [NAM] (n = 3), surgical timing (n = 8), and surgical outcomes (n = 8). Minority race or ethnicity, particularly black, Hispanic, Asian, and Native-American, as well as public insurance were associated with impaired access to care. Other factors causing impaired access to care were non-English primary language and high cost. Predictors for missed appointments were black race and Medicaid insurance. Decreased use of or success with NAM was associated with Asian race, public insurance, and long travel distance to a cleft care center. Factors associated with delayed CL surgery were Asian or black race, Hispanic ethnicity, public insurance, and non-English primary language. Surgical outcomes were assessed using various esthetic, speech, and quality of life measures; factors linked to worse outcomes included black or latin race and public insurance.

Conclusion:

Patients with CL/P from minority racial or ethnic groups, particularly black, Asian, and Hispanic, as well as those with public insurance are disproportionately impacted by health disparities. Disparities related to age, geographic location, and primary language have also been reported, among others. Statewide or institutional social programs may help address these disparities.

Keywords

cleft lip health disparities sociodemographic systematic review access to care missed appointments nasoalveolar molding surgical timing outcomes

Introduction

Cleft lip (CL) is one of the most common congenital anomalies in the world. In the United States, isolated CL (iCL) affects 1 out of every 2800 births while combined cleft lip and palate (CL+P) occurs in 1 out of 1600 live births.¹ Cleft lip with or without cleft palate (CL/P) can be diagnosed as early as 20 weeks gestational age during routine prenatal ultrasound screening.^2,3

Comprehensive management of CL/P is multidisciplinary and involves collaboration between craniofacial surgeons, dentists, otolaryngologists, speech and language pathologists (SLP), and geneticists. Management can begin with family and genetic counseling in the prenatal period and often continues into early adulthood. Primary CL repair is routinely performed between 3 and 6 months of life.^4,5 Early primary CL repair is crucial to promote feeding and growth, permit eventual speech production, and improve cosmetic appearance in this population.^6,7 Other important aspects of effective CL management include pre- and/or postoperative nasoalveolar molding (NAM), cleft rhinoplasty, revision surgeries if needed, myringotomy, speech therapy, oral hygiene, and dental care. Children with unilateral iCL have been reported to have an average total number of 5.3 surgeries during their childhood and early adulthood, while those with CL+P require an average of over 9 surgeries before their mid-20s.⁸

Given the prolonged, multidisciplinary treatment course needed for patients with CL/P as well as the benefits of early surgical repair, effective comprehensive management may be limited by sociodemographic barriers to care. While previous studies have reported different types of health disparities in the management of CL, there have been no systematic reviews summarizing these disparities. Therefore, the aim of this study is to perform a systematic review of the current literature to identify and describe sociodemographic disparities affecting access to, management of, and postoperative outcomes in patients with CL/P.

Methods

This study was conducted according to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines.⁹

Search Strategy

Literature searches were performed on October 25, 2022 using the Pubmed, Ovid Medline, and Embase databases. The search strategies for each database are provided in the Supplement and included all relevant keywords for “cleft lip” and “health disparities” (Supplemental File 1). Results included studies published online ahead of print publication. Prior to study screening and selection, the search strategy results were restricted to English language only.

Study Selection

Studies were screened by title and abstract using predefined inclusion and exclusion criteria. Screening was performed by 2 independent reviewers (CO, NM), and any differences were resolved by consensus. Duplicate studies were removed. Full text articles were then reviewed by the 3 independent reviewers (CO, AB, JB) for final inclusion. The following inclusion criteria were utilized: studies discussing any sociodemographic disparities regarding access to care, overall management, or treatment outcomes in patients with CL/P, published in and discussing disparities specifically within the US, and with a full text available in English. Review or summary articles, case reports, epidemiologic studies, opinion pieces, studies published before the year 2000, and those with no available full text article were excluded. The PRISMA guidelines were utilized for the entire study selection process (Figure 1).

Figure 1.

Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) study flow diagram.

Data Extraction

Data extraction was performed by 3 independent reviewers (CO, AB, JB) using standardized, predefined templates. Extracted data included the following: author information, publication year, study type, number of patients specifically with CL as well as demographic information such as sex, race, and/or ethnicity, and insurance status. For any studies discussing isolated CP (iCP) in addition to CL/P, only demographic information specific to patients with CL/P was documented. In addition, the patient age was uniquely defined in each study reviewed, with some using age categories and others reporting the mean or median age. Due to this inconsistency, patient age reported in each study was not documented for our review.

Data Organization

Studies were stratified into 5 predefined categories according to the type of health disparity discussed: (1) “Access to care,” (2) “Missed appointments,” (3) “Use of NAM,” (4) “Surgical timing,” and (5) “Surgical outcomes.” Studies were categorized as “Access to care” if they examined any barriers to CL/P care, including access to healthcare personnel or comprehensive cleft care centers, surgical evaluation or management, clinic visits, or speech therapy. “Missed appointments” studies focused on patients with appointments that were scheduled but not attended as well as patients who were lost to follow-up after initial evaluation. Any study primarily discussing utilization and outcomes after preoperative NAM were characterized as “Use of NAM.” “Surgical timing” studies focused on timely CL repair, which was defined uniquely by each study. Finally, studies discussing any type of postoperative outcome, including esthetic, speech, or oral health outcomes as well as postoperative complications and quality of life (QOL) were categorized as “Surgical outcomes.” Data was organized and stored on a secure, cloud-based spreadsheet (Google LLC; Mountain View, California).

Analysis

Due to considerable study heterogeneity, no quantitative meta-analysis was performed.

Results

Initial literature search yielded 3782 articles, 226 of which were screened out as non-English. After screening by title and abstract, 3478 articles were excluded. Forty-one articles were then identified as duplicates and removed. Thirty-seven articles were included for full text review. Of these 37 articles, 7 studies were excluded due to lack of an available full text, 2 studies were excluded because health disparities were not discussed, 1 article referred to a non-US based patient population, and 1 article utilized an irrelevant study design (evaluation of crowdfunding campaigns). Ultimately, 26 articles were reviewed in full.

Of all articles meeting final inclusion criteria, 13 (50%) were retrospective cohort studies, and 21 (80.8%) were published after 2010 (Table 1). Eleven (42.3%) articles discussed access to care, 2 (7.7%) discussed missed appointments, 3 (11.5%) discussed use of NAM, and 8 (30.8%) articles each discussed surgical timing or surgical outcomes. Of note, many studies failed to report demographic data for patients with CL/P separately from patients with iCP. For instance, only 9 studies specifically reported the number of black patients with CL/P. In contrast, most other studies reported the number of black patients with either iCP or CL/P, while some studies combined black patients with other racial minorities.

Table 1.

Study Characteristics and Demographic Information.

Study author	Year	Study type	Patients with cleft lip	Female sex	Racial group or ethnicity						Insurance
Study author	Year	Study type	Patients with cleft lip	Female sex	White	Black	Asian or Pacific Islander	Native American or American Indian	Other	Hispanic	Public	Private	None
Bennett et al	2018	Cross-sectional	60								9 (15%)	51 (85%)
Broder et al	2012	Cross-sectional	-
Cassell et al	2014	Cross-sectional	245	104 (42%)	199 (81%)				46 (19%)		104 (42%)	155 (47%)	14 (6%)
Cassell et al	2013	Cross-sectional	157	104 (43%)	199 (81%)				46 (19%)		104 (45%)	115 (50%)
Cassell et al	2007	Retrospective Cohort	644	280 (43%)							291 (45%)	353 (55%)
Cassell et al	2009	Retrospective Cohort	355	138 (39%)							355 (100%)
Conrad et al	2021	Retrospective Cohort	214	84 (39%)
Damiano et al	2009	Cross-sectional	175
Gibson et al	2021	Retrospective Cohort	106									78%	22%
Hopper et al	2009	Database	2558	35%	7%	14%	8%		11%	2%	42%
Kalmar et al	2022	Retrospective Cohort	8954	3385 (38%)	5988 (66.9%)						4856 (54.2%)	3729 (41.6%)
Kimia et al	2020	Retrospective Cohort	149	61 (41%)	98 (66%)	26 (17%)	7 (4%)		18 (12%)	12 (8%)	84 (56%)	65 (44%)
Lynn et al	2020	Retrospective Cohort	165	72 (43.6%)	130 (78.8%)	7 (4.2%)	10 (6.1%)		18 (10.9%)	9 (5.5%)	104 (63%)
Lynn et al	2018	Retrospective Cohort	116
Marston et al	2019	Case Control	58	14 (24%)	37 (64%)	11 (19%)	2 (3%)		1 (2%)	7 (12%)
Mullen et al	2021	Database	6021	3852 (64%)	3416 (57%)	532 (9%)	285 (5%)	34 (1%)	749 (12%)	1005 (16%)
Peck et al	2021	Geospatial	103 776
Ruff et al	2018	Prospective Cohort	917
Sharif-Askary et al	2018	Geospatial	322
Sischo et al	2012	Retrospective Cohort	83	79 (92%)	59 (71%)	8 (10%)	8 (10%)		3 (4%)	5 (6%)	28 (34%)	59 (71%)
Stoneburner et al	2021	Retrospective Cohort	503	180 (36%)	144 (29%)	15 (3%)	58 (12%)		275 (55%)	236 (47%)	297 (59%)	191 (38%)
Thompson et al	2017	Database	8385	3179 (38%)	3547 (42%)	419 (5%)	405 (5%)		476 (6%)	1944 (23%)	3646 (43%)	4026 (48%)
Wagner et al	2022	Retrospective Cohort	473
Williams et al	2003	Retrospective Cohort	331
Wolfswinkel et al	2022	Geospatial	-
Zaluzec et al	2019	Retrospective Cohort	89		11 (12%)	22 (25%)			4 (4%)	52 (58%)	76 (85%)	13 (15%)

Note. All values represent n (%). For blank cells, information was not provided or could not be deduced from the article.

Access to Care

The most common factors associated with increased or decreased access to care were race/ethnicity and insurance status (Table 2).

Table 2.

Studies Discussing Disparities in Access to Care.

Decreased access to care			Increased access to care
Author	Year of publication	Factors discussed	Author	Year of publication	Factors discussed
Cassell et al	2014	• Cost: high• Familial obligations• Geographic: long travel times• Job/career• Logistical: long waiting room hours, delays to schedule appointment	Cassell et al	2007	• Sex: male• Insurance: Medicaid• Social services: maternity care coordination
Kalmar et al	2022	• Income: below median	Cassell et al	2013	• Education: partial college
Lynn et al	2020	• Race/ethnicity: black	Kalmar et al	2022	• Race/ethnicity: white
Peck et al	2021	• Geographic: location in the South	Peck et al	2021	• Geographic: location in the Northeast
Ruff et al	2018	• Race/ethnicity: Asian or Hispanic/Latino	Ruff et al	2018	• Age: increasing• Insurance: private
Williams et al	2003	• Maternal age: ≥25 yRace/ethnicity: white	Wagner et al	2022	• Insurance: private• Race/ethnicity: white
Wolfswinkel et al	2022	• Race/ethnicity: American Indian and Native American
Zaluzec et al	2019	• Language: Non-English (for CL+P but not for iCL)

Race and Ethnicity

Several studies demonstrated that patients from minority racial groups have decreased access to care. In their 2020 retrospective, single-institution cohort study consisting of 165 patients with CL/P undergoing surgical repair, Lynn et al performed a multivariate analysis and found that black patients were first evaluated by a SLP 55 weeks later than non-black patients (P = .03).¹⁰ In 2018, Ruff et al performed a prospective cohort study which included 917 patients with CL, and they found that Asian or Hispanic patients had significantly fewer surgeries than their white counterparts.¹¹ Furthermore, a recent geospatial study demonstrated that 47.1% of American Cleft Palate Craniofacial Association (ACPA) accredited centers situated in areas highly populated by American-Indian or Native American people were not staffed by craniofacial surgeons, compared to just 21.1% of ACPA centers nationwide.¹²

Two other studies demonstrated that white patients had better access to care compared to other races. In their retrospective, single-center study including 473 patients with CL/P, Wagner et al examined health disparities before and after implementation of a cleft nurse navigator (CNN).¹³ They noted that white patients had significantly more communication with nursing staff and had earlier cleft care than non-white patients before CNN implementation. After implementation, however, these racial disparities were no longer observed. In 2022, Kalmar et al found that white patients were more likely than non-white patients to be treated by a high-volume surgeon, defined by surgeon case volume greater than the 80th percentile (adjusted odds ratio [OR] = 2.5, 95% confidence interval [CI] 1.6-3.9).¹⁴

While most studies demonstrated an association between minority race and decreased access to care, one study appears to challenge this finding. In a 2003 retrospective cohort study including 331 patients with CL/P from the Florida Birth Defects Registry, white patients were found to have lower rates of contact with state-run cleft management centers than black or Hispanic patients.¹⁵

Insurance Status

Two studies evaluated the impact of private insurance on access to care. Wagner et al demonstrated that patients with private insurance had their first outpatient cleft care appointment 12 days earlier than those with public insurance before implementation of a CNN (P < .001).¹³ This disparity was reduced but still remained after CNN implementation. In addition, Ruff et al showed that patients with private insurance had higher rates of esthetic procedures than those with non-private insurance.¹¹

In contrast, one study noted increased access to care for Medicaid-insured patients. In a 2007 study which included 644 patients with CL/P, Cassell et al reported that patients insured by Medicaid were more likely than non-Medicaid patients to be referred to the North Carolina Child Service Coordination Program, a state-run specialized service for children with birth defects.¹⁶

Other Risk Factors for Decreased Access to Care

Non-English primary language has been linked to older age at the first clinic visit for patients with CL+P but not for those with iCL.¹⁷ In a 2014 survey-based study, Cassell et al identified multiple barriers to care as reported by caregivers of children with CL/P, including high cost, familial or job obligations, and long travel distances.¹⁸ Remaining risk factors for decreased access to care reported by other studies were low income level, maternal age over 25 years, maternal education lower than college level, and geographic location in the Southern US.^14,15,19,20

Missed Appointments

Only 2 studies assessed health disparities related to missed appointments (Table 3). Lynn et al’s 2018 article defined missed appointment rate as the number of missed appointments divided by the number of scheduled appointments.²¹ They found that black patients had significantly higher rates of missed appointments compared to white patients (P = .00). In addition, this study demonstrated that patients insured by Medicaid, from unstable households, or those with multiple siblings had increased rates of missed appointments compared to their counterparts in a multivariate analysis.

Table 3.

Studies Discussing Disparities in Missed Appointments and Use of Nasoalveolar Molding.

Increased missed appointments			Decreased missed appointments
Author	Year of publication	Factors discussed	Author	Year of publication	Factors discussed
Lynn et al	2018	• Household: unstable or multiple siblingsInsurance: MedicaidRace/ethnicity: black	Lynn et al	2018	Being religious
			Sharif-Askary et al	2018	Age: olderSocioeconomic status: low
Decreased use of nasoalveolar molding			Increased use of nasoalveolar molding
Author	Year of publication	Factors discussed	Author	Year of publication	Factors discussed
Gibson et al^a	2021	• Maternal age: increasing	Gibson et al^a	2021	• Household: married/partnered parents• Insurance: private• Parental age: increasing
Kimia et al	2020	• Geographic: travel distance >90 miles• Race/ethnicity: Asian	Kimia et al	2020	• Insurance: private
Sischo et al	2012	• Household: multiple children	Sischo et al	2012	• Birth order: first or second

Assessed success and difficulty of nasoalveolar molding, rather than use of the device.

In a 2018 study, Sharif-Askary et al examined risk factors for “loss to follow-up” (LTFU), defined as missing 3 consecutive appointments despite attempts to resume follow-up or over 2.5 years with no attempted follow-up.²² They performed 2 multivariate analyses–with and without consideration of geospatial dependency. Results demonstrated that patients who were older at the time of last follow-up had a lower odds of LTFU, regardless of geospatial dependency (OR 0.81, 95% CI 0.75-0.88 without geospatial dependency; OR 0.81, 95% CI 0.77-0.85 with geospatial dependency). In addition, lower socioeconomic status (SES), which was determined based on each patient’s address, was associated with a slightly lower rate of LTFU when considering geospatial dependency (OR 0.98, 95% CI 0.97-0.99), but there was no significant difference in LTFU based on SES when excluding geospatial dependency from analysis.

Use of Nasoalveolar Molding

Three studies assessed the use of preoperative NAM in patients with CL/P, with all reporting factors associated with increased and decreased use (Table 3).

Risk Factors for Decreased Use or Success

Kimia et al defined use of NAM based on attendance to clinic appointments and adherence to treatment.²³ They demonstrated that Asian patients were significantly less likely than white patients to use NAM (OR 0.09, 95% CI 0.01-0.97). In addition, patients with a travel distance of greater than 90 miles to the cleft care center were less likely to utilize NAM than those with a shorter travel distance (OR 0.27, 95% CI 0.09-0.80). In their 2012 retrospective cohort study, Sischo et al noted that patients with multiple siblings were significantly less likely to receive NAM than those with no siblings (P < .05).²⁴ Gibson et al focused on the success and difficulty associated with NAM use.²⁵ This study found that increasing maternal age was associated with decreased success in cleft gap closure (P = .041).

Factors Associated With Increased Use or Success

Kimia et al reported that patients with private insurance were more likely than those with public insurance to use NAM.²³ Similarly, Gibson et al showed that private insurance was associated with fewer appliance wear issues compared to non-private insurance, indicating lower difficulty of use (P = .019).²⁵ Married or partnered parents also had fewer appliance issues than single or divorced parents (P = .018). Gibson et al further found higher rates of cleft gap closure, indicating greater treatment success, in patients with married or partnered parents compared to single or divorced parents (P = .018) as well as in those with older paternal age (P = .007). Lastly, Sischo et al demonstrated that patients who were their parents’ first or second born were more likely to receive NAM than those who were third born or higher.²⁴

Surgical Timing

Most of the studies that discussed surgical timing focused on risk factors for delayed surgery, with race and insurance status being the most commonly cited risk factors (Table 4).

Table 4.

Studies discussing disparities in surgical timing.

Delayed surgical timing			Appropriate surgical timing
Author	Year of publication	Factors discussed	Author	Year of publication	Factors discussed
Cassell et al	2009	• Maternal age: ≥30• Race: black	Cassell et al	2009	• Social services: maternity care coordination
Lynn et al	2020	• Household: Child Protective Services involvement• Race/ethnicity: Asian, black	Kalmar et al	2022	• Insurance: private• Race/ethnicity: white
Kalmar et al	2022	• Geographic: urban setting	Wagner et al	2022	• Insurance: private• Before CNN, no significant difference after CNN
Mullen et al	2021	• Race/ethnicity: Asian, Hispanic
Ruff et al	2018	• 1. Insurance: non-private
Stoneburner et al	2021	• Geographic: shorter travel distance• Insurance: government• Language: Spanish primary• Race/ethnicity: non-HispanicSex: female
Wagner et al	2022	• Race/ethnicity: non-white• Before implementing CNN, no significant difference after CNN
Zaluzec et al	2019	• Language: non-English• Race/ethnicity: Hispanic

Note. CNN = clinical nurse navigator.

Race and Ethnicity

Multiple studies in this review have suggested that non-white or minority race was often associated with delayed operative intervention. In the 2009 study by Cassell et al, which included 355 patients with CL, timely cleft surgery was defined as occurring within 18 months of life in children with CL/P.²⁶ They conducted a multivariate logistic regression, which demonstrated that non-Hispanic black patients had a significantly lower odds of timely surgery than non-Hispanic white patients (adjusted OR 0.30, 95% CI 0.14-0.67).

In their 2020 study, Lynn et al noted that Asian and black race were both significantly associated with older age at the time of surgical repair compared with white race.¹⁰ In a retrospective database review that included 6021 patients with CL, of which 285 patients (4.7%) were Asian, Mullen et al similarly demonstrated a higher likelihood of delayed CL repair in Asian compared to white patients.²⁷ Furthermore, Wagner et al showed that non-white patients received CL repair 16 days later than white patients before implementation of a CNN (P = .011), but this disparity decreased to 8 days after CNN implementation (P = .095).¹³ Kalmar et al also noted that white race was associated with significantly earlier CL repair than non-white race (P < .001), which remained true for patients with unilateral or bilateral CL.¹⁴

Several studies have examined the impact of Hispanic ethnicity on timeliness of surgery. Mullen et al reported an increased likelihood of delayed CL repair in Hispanic patients compared to Caucasians.²⁷ Likewise, Zaluzec et al found that Hispanic children had a significantly longer lag time, defined as time from first clinic visit to CL repair, than white children (3.8 vs 2.6 months, P = .05). In contrast to these findings, Stoneburner et al noted that non-Hispanic patients were significantly more likely to have delayed CL repair, defined as surgery after 6 months of life, compared to Hispanic patients.²⁸

Insurance Status

Two studies have cited non-private or public insurance as a risk factor for delayed CL repair. Stoneburner et al found that patients with government insurance were more likely to have delayed CL repair than those with private insurance.²⁸ In addition, Ruff et al noted that patients with non-private insurance were more likely to postpone recommended surgeries than those with private insurance.¹¹ Other studies have identified private insurance as a predictor of timely surgery. Both Kalmar et al and Wagner et al reported that patients with commercial or private insurance had significantly earlier CL repair than their publicly insured counterparts. However, Wagner et al further showed that this disparity was present before but was no longer observed after implementation of a CNN.

Other Risk Factors for Delayed Surgical Timing

Primary language spoken in the child’s household appeared to impact the timeliness of CL repair. Children were more likely to have delayed surgery if they were from non-English-speaking or primarily Spanish-speaking households.^17,28 Other risk factors for delayed surgical timing were maternal age over 30, involvement of Child Protective Services in the household, and living in an urban location.^10,14,26

Surgical Outcomes

Race and insurance status were repeatedly found to impact treatment outcomes (Table 5).

Table 5.

Studies discussing disparities in surgical outcomes.

Worse surgical outcomes			Improved surgical outcomes
Author	Year of publication	Factors discussed	Author	Year of publication	Factors discussed
Bennett et al	2018	• Cost: financial concerns• Education: higher level• Health literacy: poor communication with healthcare system	Conrad et al	2021	• Race: white• Socioeconomic status: higher
Broder et al	2012	• Insurance: non-private• Race/ethnicity: black: Latino	Damiano et al	2009	• Geographic: location in Arkansas (compared to New York or Iowa)• Household: 3 or more children
Hopper et al	2009	• Insurance: Medicaid• Setting: high-volume surgeon (for readmission)• Setting: low-volume surgeon (for serious inpatient complication)	Hopper et al	2009	• Age: older• Setting: high-volume hospital
Marston et al	2019	• Race/ethnicity: black
Mullen et al	2021	• Race/ethnicity: black, Hispanic
Thompson et al	2017	• Insurance: private• Race/ethnicity: Asian/Pacific Islander• Sex: female

Race and Ethnicity

Three studies showed that minority race, often black, was associated with worse surgical outcomes.^27,29,30 Marston et al demonstrated worse scar outcomes in black patients compared to white patients, and Mullen et al showed higher readmission rates in black and Hispanic patients compared to Asian and white patients.^27,30 In 2012, Broder et al utilized a survey instrument to assess various QOL measures.²⁹ Compared to Asian and white patients, black and Latino patients in this study had lower scores for emotional well-being (P < .001) and for school (P < .05), which was defined as their ability to engage in school-related tasks. Similarly, Conrad et al’s questionnaire-based study found that white race was predictive of better parent-reported functioning, despite controlling for the amount of surgeries undergone.³¹

Insurance Status

Two studies reported public insurance as a risk factor for worse surgical outcomes. In Broder et al’s study, non-private insurance was associated with lower functional and emotional well-being scores.²⁹ Hopper et al performed a retrospective database study consisting of 2558 patients who underwent primary CL repair. In this study, patients with Medicaid demonstrated a higher odds of readmission compared to those with non-Medicaid insurance.³² Thompson et al reported that private insurance was linked to a higher odds of CL revision.³³

Other Risk Factors for Worse Surgical Outcomes

Hopper et al showed that children undergoing primary CL repair at an older age were more likely to be discharged on the same day.³² In addition, Bennett et al demonstrated worse caregiver-reported speech and esthetic outcomes from caregivers who expressed concerns about healthcare costs as well as those who endorsed low health literacy.³⁴ Thompson et al reported that female patients were more likely to undergo revision CL repair than their male counterparts.³³ Other factors impacting surgical outcomes were household size and practice setting.^32,35

Discussion

Multiple health disparities affecting the overall management of CL/P as well as postoperative outcomes after CL repair have been reported. Most of the articles reviewed in this study were published in the last decade, suggesting a growing interest and awareness of this issue. A variety of sociodemographic factors were noted to impact the care of patients with CL/P, including race and ethnicity, insurance status, sex, primary language, and geographic location, among others. Disparities related to patient race and insurance status were most frequently reported. These represent possible targets for future advocacy efforts.

Patients from minority racial and/or ethnic groups, particularly black, Asian, and Hispanic, were disproportionately affected by disparities in all categories of CL/P management and outcomes defined by this study. One study demonstrated disparities in access to care specifically for Native-American or American-Indian patients.¹² However, it is important to note that this racial group was rarely included independently and was often combined with other racial groups due to a small sample size. As a result, it is possible that disparities for this racial group may be more prominent than reported in the literature. Nevertheless, exact reasons for the racial disparities observed in this study have not been well defined. Some studies speculate that differences in access to resources or cultural preferences may have a role.^11,26 Though not commonly reported specifically in patients with CL/P, potential explanations for racial disparities have been explored across other medical specialties. These potential explanations include racial differences in socioeconomic status, access to resources, health literacy, or cultural practices as well as implicit bias among providers.^36-38 Historical or ongoing systematic and discriminatory practices, such as residential segregation, that disproportionately impact racial minority groups may also play a role in the disparities observed in our review.³⁹ More than likely, the reasons for the racial disparities affecting patients with CL/P are multifactorial, involving a complex combination of the aforementioned factors.

Insurance status was another frequently reported disparity also affecting all categories defined in this study. Compared to those with private or commercial insurance, patients with public insurance more frequently encountered barriers to equitable care. These results suggest that publicly-insured children may be receiving a lower quality of cleft care than their privately insured counterparts.

Fortunately, some studies in this review have reported that disparities in CL/P management and outcomes may be mitigated by institutional or statewide social programs. Wagner et al’s study demonstrated improved racial and insurance disparities for both access to care and surgical timing after implementation of an institutional CNN.¹³ In their 2009 study, Cassell et al evaluated the impact of maternity care coordination services, a program offering medical, psychosocial, and financial support during and after pregnancy for women eligible for Medicaid in North Carolina. They found that children of caregivers who received these services had a higher odds of timely cleft surgery within 18 months of life.²⁶ Though these studies report favorable results, larger, multi-institutional studies are necessary to reinforce the efficacy of similar or innovative social programs for reducing disparities in CL/P care.

This study has several limitations. In particular, all studies included in this review were retrospective or cross-sectional, which brings into questions the generalizability of these results. In addition, many of the studies were limited to a single institution. Furthermore, some studies combined results for patients with iCP and those with CL/P rather than reporting disparities for patients with CL/P separately, which increases the risk of confounding bias for our review. Lastly, while we were able to characterize all studies based on predefined categories, each study had unique methods for defining the outcomes assessed. For example, appropriate surgical timing was defined differently in each study that evaluated this outcome. Variations in the definitions employed across different studies may have introduced bias into our results.

Conclusions

Patients with CL/P from minority racial or ethnic groups, particularly black, Asian, and Hispanic, as well as those with non-private insurance are disproportionately affected by health disparities and may be at risk of receiving inferior care. Disparities related to sex, geographic location, and primary language have also been reported. Statewide or institutional social programs can help address these disparities. Future research should focus on developing new strategies to promote equity in the management of patients with CL/P.

Supplemental Material

sj-docx-1-fac-10.1177_27325016241227614 – Supplemental material for Revealing a Cleft in the Community: A Systematic Review of Sociodemographic Disparities Affecting the Care of Patients With Cleft Lip

Supplemental material, sj-docx-1-fac-10.1177_27325016241227614 for Revealing a Cleft in the Community: A Systematic Review of Sociodemographic Disparities Affecting the Care of Patients With Cleft Lip by Chioma G. Obinero, Alexandra Boyd, Jose Barrera, Alyssa Valenti, Naikhoba Munabi, Thomas Imahiyerobo and Matthew R. Greives in FACE

Supplemental Material

sj-docx-2-fac-10.1177_27325016241227614 – Supplemental material for Revealing a Cleft in the Community: A Systematic Review of Sociodemographic Disparities Affecting the Care of Patients With Cleft Lip

Supplemental material, sj-docx-2-fac-10.1177_27325016241227614 for Revealing a Cleft in the Community: A Systematic Review of Sociodemographic Disparities Affecting the Care of Patients With Cleft Lip by Chioma G. Obinero, Alexandra Boyd, Jose Barrera, Alyssa Valenti, Naikhoba Munabi, Thomas Imahiyerobo and Matthew R. Greives in FACE

Footnotes

Cleft Abbreviations

CL = Cleft lip

CL/P = Cleft lip with or without cleft palate

CL+P = Combined cleft lip and palate

CP = Cleft palate

iCL = Isolated cleft lip

iCP = Isolated cleft palate

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.

Ethical Approval

Institutional Review Board approval was not required.

Informed Consent

Informed Consent was not required for this article

ORCID iDs

Chioma G. Obinero

Naikhoba Munabi

Matthew R. Greives

Supplemental Material

Supplemental material for this article is available online.

References

Mai

Isenburg

Canfield

, et al. National population-based estimates for major birth defects, 2010-2014. Birth Defects Res. 2019;111(18):1420-1435. doi:10.1002/bdr2.1589

Maarse

Pistorius

Van Eeten

, et al. Prenatal ultrasound screening for orofacial clefts. Ultrasound Obstet Gynecol. 2011;38(4):434-439. doi:10.1002/uog.8895

Smarius

Loozen

Manten

, et al. Accurate diagnosis of prenatal cleft lip/palate by understanding the embryology. World J Methodol. 2017;7(3):93-100. doi:10.5662/wjm.v7.i3.93

James

Costello

Ruiz

RL.

Management of cleft lip and palate and cleft orthognathic considerations. Oral Maxillofac Surg Clin North Am. 2014;26(4):565-572. doi:10.1016/j.coms.2014.08.007

Kobus

Kobus-Zaleśna

Timing of cleft lip and palate repair. Dev Period Med. 2014;18(1):79-83.

Hammoudeh

Imahiyerobo

Liang

, et al. Early cleft lip repair revisited: a safe and effective approach utilizing a multidisciplinary protocol. Plast Reconstr Surg Glob Open. 2017;5(6):e1340. doi:10.1097/GOX.0000000000001340

Lancaster

Lien

Chow

, et al. Early speech and language development in children with nonsyndromic cleft lip and/or palate: a meta-analysis. J Speech Lang Hear Res. 2020;63(1):14-31. doi:10.1044/2019_JSLHR-19-00162

McIntyre

Sethi

Schönbrunner

, et al. Number of surgical procedures for patients with cleft lip and palate from birth to 21 years old at a single children’s hospital. Ann Plast Surg. 2016;76:S205-S208. doi:10.1097/SAP.0000000000000765

Page

McKenzie

Bossuyt

, et al. The PRISMA 2020 statement: an updated guideline for reporting systematic reviews. BMJ. 2021;372:n71. doi:10.1136/bmj.n71

10.

Lynn

Ranganathan

Bageris

, et al. Sociodemographic predictors of patient age at time of cleft lip and palate repair. Cleft Palate Craniofac J. 2020;57(12):1402-1409. doi:10.1177/1055665620949119

11.

Ruff

Crerand

Sischo

, et al. Surgical care for school-aged youth with cleft: results from a multicenter, prospective observational study. Cleft Palate Craniofac J. Published online January 1, 2018. doi:10.1177/1055665618765776

12.

Wolfswinkel

Howell

MacDonald

Wilson

Howell

LK.

American Indian and Alaska Native accessibility to comprehensive cleft lip and palate treatment. Cleft Palate Craniofac J. 2023;60:1384. doi:10.1177/10556656221104942

13.

Wagner

Zimmerman

Barrero

, et al. Reduced socioeconomic disparities in cleft care after implementing a cleft nurse navigator program. Cleft Palate Craniofac J. 2022;59(3):320-329. doi:10.1177/10556656211005646

14.

Kalmar

Malphrus

Kosyk

Zapatero

Taylor

JA.

Socioeconomic disparities in cleft lip care. Cleft Palate Craniofac J. 2023;60(6):657-662. doi:10.1177/10556656221078488

15.

Williams

Mardon

Grove

, et al. Treatment of oralfacial clefts by state-affiliated craniofacial centers and cleft palate clinics. Birth Defects Res A Clin Mol Teratol. 2003;67(9):643-646. doi:10.1002/bdra.10115

16.

Cassell

Meyer

Farel

AM.

Predictors of referral to the North Carolina Child Service Coordination Program among infants with orofacial clefts. Cleft Palate Craniofac J. 2007;44(1):45-51. doi:10.1597/05-198

17.

Zaluzec

Rodby

Bradford

, et al. Delay in cleft lip and palate surgical repair: an institutional review on cleft health disparities in an urban population. J Craniofac Surg. 2019;30(8):2328-2331. doi:10.1097/SCS.0000000000005740

18.

Cassell

Strassle

Mendez

, et al. Barriers to care for children with orofacial clefts in North Carolina. Birth Defects Res A Clin Mol Teratol. 2014;100(11):837-847. doi:10.1002/bdra.23303

19.

Peck

Parsaei

Lattanzi

, et al. The geographic availability of certified cleft care in the United States: a national geospatial analysis of 1-hour access to care. J Oral Maxillofac Surg. 2021;79(8):1733-1742. doi:10.1016/j.joms.2021.02.043

20.

Cassell

Krohmer

Mendez

, et al. Factors associated with distance and time traveled to cleft and craniofacial care. Birth Defects Res A Clin Mol Teratol. 2013;97(10):685-695. doi:10.1002/bdra.23173

21.

Lynn

Ranganathan

Bageris

, et al. Sociodemographic predictors of missed appointments among patients with cleft lip and palate. Cleft Palate Craniofac J. 2018;55(10):1440-1446. doi:10.1177/1055665618764739

22.

Sharif-Askary

Bittar

Farjat

, et al. Geospatial analysis of risk factors contributing to loss to follow-up in cleft lip/palate care. Plast Reconstr Surg Glob Open. 2018;6(9):e1910. doi:10.1097/GOX.0000000000001910

23.

Kimia

Butler

Guajardo

, et al. Sociodemographic factors that influence the choice to pursue nasoalveolar molding: one Pediatric Hospital’s experience. Cleft Palate Craniofac J. 2020;57(9):1069-1077. doi:10.1177/1055665620936056

24.

Sischo

Chan

Stein

, et al. Nasoalveolar molding: prevalence of cleft centers offering NAM and who seeks it. Cleft Palate Craniofac J. 2012;49(3):270-275. doi:10.1597/11-053

25.

Gibson

Grayson

Shetye

PR.

Sociodemographic predictors of treatment success and difficulty in nasoalveolar molding. Cleft Palate Craniofac J. 2021;58(3):378-385. doi:10.1177/1055665620949791

26.

Cassell

Daniels

Meyer

RE.

Timeliness of primary cleft lip/palate surgery. Cleft Palate Craniofac J. 2009;46(6):588-597. doi:10.1597/08-154.1

27.

Mullen

Yan

Ford

Patel

Pecha

PP.

Racial and ethnic disparities in primary cleft lip and cleft palate repair. Cleft Palate Craniofac J. 2023;60(4):482-488. doi:10.1177/10556656211069828

28.

Stoneburner

Munabi

NCO

Nagengast

, et al. Factors associated with delay in cleft surgery at a tertiary children’s Hospital in a major US Metropolitan City. Cleft Palate Craniofac J. 2021;58(12):1508-1516. doi:10.1177/1055665621989508

29.

Broder

Wilson-Genderson

Sischo

Health disparities among children with cleft. Am J Public Health. 2012;102(5):828-830. doi:10.2105/AJPH.2012.300654

30.

Marston

Costello

Farhood

, et al. Association of pediatric patient demographic factors and scar anatomic features with scar outcomes after surgical repair of cleft lip. JAMA Facial Plast Surg. 2019;21(5):452-457. doi:10.1001/jamafacial.2019.0669

31.

Conrad

Albert

Crerand

, et al. Retrospective evaluation of number of surgeries and parent ratings of academic and behavioral functioning among children with isolated oral clefts. Cleft Palate Craniofac J. 2021;58(10):1294-1303. doi:10.1177/1055665620982807

32.

Hopper

Lewis

Umbdenstock

Garrison

Starr

JR.

Discharge practices, readmission, and serious medical complications following primary cleft lip repair in 23 U.S. Children’s Hospitals. Plast Reconstr Surg. 2009;123(5):1553-1559. doi:10.1097/PRS.0b013e3181a0746e

33.

Thompson

Heaton

Kelton

Sitzman

TJ.

National estimates of and risk factors for inpatient revision surgeries for orofacial clefts. Cleft Palate Craniofac J. 2017;54(1):60-69. doi:10.1597/15-206

34.

Bennett

Ranganathan

Patterson

, et al. Caregiver-reported outcomes and barriers to care among patients with cleft lip and palate. Plast Reconstr Surg. 2018;142(6):884e-891e. doi:10.1097/PRS.0000000000004987

35.

Damiano

Tyler

Romitti

, et al. Demographic characteristics, care, and outcomes for children with oral clefts in three states using participants from the National Birth Defects Prevention Study. Cleft Palate Craniofac J. 2009;46(6):575-582. doi:10.1597/07-237.1

36.

Patel

Edison

Carter

Pandya

NK.

The impact of race, insurance, and socioeconomic factors on pediatric knee injuries. Clin Sports Med. 2022;41(4):789-798. doi:10.1016/j.csm.2022.05.012

37.

Coker

Elliott

Toomey

, et al. Racial and ethnic disparities in ADHD diagnosis and treatment. Pediatrics. 2016;138(3):e20160407. doi:10.1542/peds.2016-0407

38.

Arant

Modest

Gil

Cruz

Jr.

What’s new in pediatric orthopaedic health care disparities?

J Pediatr Orthop. 2022;42(9):e954-e959. doi:10.1097/BPO.0000000000002224

39.

National Academies of Sciences, Engineering, and Medicine; Health and Medicine Division; Board on Population Health and Public Health Practice; Baciu

Negussie

Geller

, et al, eds. Communities in Action: Pathways to Health Equity. National Academies Press; 2017.

Supplementary Material

Please find the following supplemental material available below.

For Open Access articles published under a Creative Commons License, all supplemental material carries the same license as the article it is associated with.

For non-Open Access articles published, all supplemental material carries a non-exclusive license, and permission requests for re-use of supplemental material or any part of supplemental material shall be sent directly to the copyright owner as specified in the copyright notice associated with the article.

0.00 MB

0.03 MB

0.01 MB