Cleft and Craniofacial Multidisciplinary Team Clinic: A Look at Attrition Rates for Patients With Complete Cleft Lip and Palate and Nonsyndromic Single-Suture Craniosynostosis

Abstract

Objective:

To evaluate attrition rates prior to expected completion of team care for children with complete cleft lip and palate (cleft) or nonsyndromic single-suture craniosynostosis (synostosis).

Design:

A single-institution retrospective review of attendance data from 2002 to 2016.

Setting:

Single cleft and craniofacial center in the United States.

Patients/Participants:

A sample of 983 patients with either cleft or synostosis. Patients who were more than 2 to 3 years from their last visit were considered lost to follow-up. Patients with cleft older than 16 years or synostosis over 11 years were considered graduated from team care.

Results:

Survival analysis shows that in our patients with cleft, 25% leave before age 8 and over 60% are lost from team by age 16. In patients with synostosis, 25% leave before age 6 and 45% are lost by age 11. Cox regression showed underrepresented minorities being 1.7 times more likely to become lost in the cleft group (hazard ratio: 1.66, 95% confidence interval [CI]: 1.01–2.74).

Conclusions:

Overall, attrition rates were high at our institution. Many patients are lost to follow-up prior to receiving key medical interventions. Improved family education and personalized care are needed to help ensure continuity of care.

Introduction

Medical care of patients with cleft and craniofacial anomalies can be complex. Care requires input from multiple health-care providers, involves procedures spanning childhood, and requires family counseling and education, often in the setting of complex social situations (Cohen, 2016). In 1987, the US Surgeon General’s report acknowledged the importance of comprehensive care due to the need for coordinated, culturally sensitive, patient-centered, and readily accessible care for children with special health-care requirements (Koop, 1987).Multidisciplinary team clinics help streamline care so patients and their families can consolidate multiple appointments into a single visit. This consolidation lessens the burden of care by reducing the number of clinical visits, time parents must take off from work, additional expenses related to travel, and child care needs for siblings. This coordination ultimately helps minimize the disruption of daily life for families. Despite the benefits team clinics bring to the care of patients, we were concerned that patients were leaving the cleft-craniofacial clinic prior to expected completion of care.

This article focuses on children with complete cleft lip and palate (cleft) and nonsyndromic single-suture craniosynostosis (synostosis). In general, our care guidelines are as follows: patients with cleft are seen until age 16 to 20 years. They are followed annually until age 6 years and then biannually thereafter. Surgical management for patients with cleft has been standardized to occur at specific time periods based on the physical and psychosocial development of the child (Mathijssen, 2015; Daskalogiannakis and Antonarakis, 2016). Prior to graduating from team care, our patients undergo key standard interventions including cleft lip repair, palate repair, orthodontic procedures, and alveolar bone grafting. If indicated, patients may also undergo cleft rhinoplasty and orthognathic surgery at skeletal maturity. In addition to surgical care provided by plastic surgeons and otolaryngologists, patients also receive care from multiple nonsurgical providers including orthodontists, speech-language pathology, psychology, and audiology.

We follow patients with nonsyndromic synostosis until there has been sufficient stability in the patient’s condition such that the team providers deem it appropriate to graduate from team care. In general, we see these patients every 1 to 3 years at least through age 11. Although these patients are nonsyndromic, there has been evidence of cognitive, speech, or behavioral abnormalities, and therefore, these patients are best managed in a team setting (Cohen et al., 2004; Becker et al., 2005, Kelleher et al., 2006, Nischal, 2007). Long-term follow-up is also important to evaluate anthropometric outcomes and potential need for secondary procedures.

We sought to understand when and why patients and families stop coming to team clinic. We aimed to identify patients at risk of missing key interventions due to premature departure from team care. With this information, we can better educate our patients and modify our therapies to better suit the needs of patients and their families.

Patients and Methods

Following IRB approval, we retrospectively reviewed team clinic attendance data from January 2002 to September 2016. Patients who were more than 2 years from their last visit in the cleft group or 3 years out from their last visit in the synostosis group and had no follow-up appointment scheduled were considered inactive and lost to follow-up. These patients had become lost to all providers in the multidisciplinary team clinic. Clinic patients who were seen at or above the age of 16 years in the cleft group or 11 years of age in the synostosis group were considered graduated, as this is the youngest age that patients in both groups can graduate from team clinic. Patients younger than the age of 16 or 11 years, respectively, who had been seen in team clinic or had a cleft or synostosis-related procedure within the past 24 or 36 months were considered active patients.

We gathered patient age at last visit, sex, race, distance traveled to clinic based on zip code, and insurance status. We also excluded as outliers patients ≥ 30 years old at their final visit, as well as those living ≥ 300 miles from our institution in the cleft or ≥ 400 miles in the synostosis group. As a proxy for private socioeconomic status, we culled the appropriate median family income based on zip code and race from publicly available US census data via factfinder.census.gov (median income in the past 12 months, in 2015 inflation-adjusted dollars). Distance to clinic was obtained by querying the Google Maps Distance Matrix API running a script in visual basic for applications in Excel 2013 (Microsoft, Redmond, Washington).

To assess reasons for loss to follow-up, an anonymous brief survey was sent to parents of patients who failed to appear for scheduled visits for the past 3 years and had an e-mail address on file. The initial attempt was sent electronically via Survey-Monkey. Mailed surveys were sent to a subset of those who did not respond to the e-mailed survey. A final attempt by phone call was used on the remaining nonresponders. The survey attempted to understand the parent’s reason for leaving clinic and their perceived out-of-pocket costs (see Appendix A).

In addition, we queried our database 15 months after the initial query to determine whether any patients returned to clinic after being previously classified lost to follow-up. These patients’ charts were reviewed to identify reasons for return to team care.

SPSS (IBM SPSS Statistics for Windows, version 23.0; IBM Corp, Armonk, New York) was used for all data analysis. Proportions of categorical variables were compared using 2-sided Fisher exact tests. The means of continuous variables were compared using 2-tailed Student t tests or 1 - way analysis of variance (with Tukey Honestly Significant Difference post hoc), as appropriate. Kaplan-Meier survival analysis was used to evaluate the attrition of patients by age. Effects of parameters on the likelihood of being lost to follow-up were assessed using forced entry multivariable Cox proportional hazard models. In both the Kaplan-Meier and Cox models, age at final visit or graduation age is used as the time of event. Statistical significance was set a priori at P < .05 for all analysis. Outliers (in patient age and distance to clinic) were defined by the research team after reviewing scatterplots of the data and prior to analysis. There were distinct cut points in the data leading to the different categorizations.

We included African Americans, Hispanics, and Native Americans into the underrepresented minority group, while Caucasian and Asian patients, as well as those listed as “other” or “unknown” were included in the majority group. Asians were included in the majority group as, in almost every case, they had been adopted by Caucasian parents and/or lived in relatively high-ineome neighborhoods. The other/unknown patients were considered as part of the majority as the proportion of Caucasians in our cohort is much greater than that of all other groups. Mistakenly including a Caucasian in the Under-respresented minority (URM) group would have a greater effect than mistakenly including an URM in the majority groups.

Results

A total of 675 patients with complete cleft lip and palate were seen over this 15-year time period. We had a total of 529 (78%) patients classified as Caucasian, other, or unknown. There were 75 (11%) Asian patients, 51 (8%) African Americans, 19 (3%) Hispanics, and 1 Native American (0.1%). After excluding outliers, a total of 652 patients remained. Of those 652, 226 (35%) were still active patients, 123 (19%) had graduated from team care, and 303 (46%) were deemed lost to follow-up.

During that same time period, we saw a total of 344 patients with nonsyndromic synostosis. A total of 320 (93%) patients classified as Caucasian, other, or unknown. There were 4 (1%) Asian patients, 11 (3%) African Americans, and 9 (2.6%) Hispanics. After excluding outliers, we were left with a total of 331 patients. Of those remaining patients, 184 (56%) had sagittal, 73 (22%) metopic, 58 (18%) unicoronal, and 16 (5%) lambdoid synostosis. One hundred ninety-one (58%) were still active patients, 101 (31%) were considered lost, and 39 (12%) were graduated from team care.

Demographic information for patients who were lost and who graduated from team care can be seen in Table 1. For the synostosis population, the only demographic difference between groups was that graduates lived closer to clinic than those who were lost to follow-up (P = .006). No demographic differences were found in the cleft population.

Table 1.

Demographics of Patients With Complete Cleft Lip and Palate and Nonsyndromic Single-Suture Craniosynostosis Based on Whether Patients Graduated From Team Care or Were Lost to Follow-Up.

	Synostosis			Cleft
Demographics	Graduates	Lost to Follow-Up	P Value	Graduates	Lost to Follow Up	P Value
Sex
n (%)			.33			.26
Male	28 (72)	63 (62)		85 (31)	191 (63)
Female	11 (28)	38 (38)		38 (31)	112 (37)
Race
n (%)			.50			.14
Underrepresented minoritya	4 (10)	7 (7)		7 (6)	32 (11)
Majorityb	35 (90)	94 (93)		116 (94)	271 (89)
Medicaid
n (%)			.50			.91
Yes	14 (36)	44 (44)		53 (43)	128 (43)
No	25 (64)	57 (56)		70 (57)	173 (57)
Distance (miles), mean (SD)	71 (64)	116 (91)	<.01	87 (69)	86 (74)	.83
Income ($1000), mean (SD)	56 (21)	56 (22)	.97	54 (20)	53 (21)	.61
Diagnosis
n (%)			.25
Sagittal	18 (46)	60 (59)
Metopic	9 (23)	17 (17)
Unicoronal	8 (21)	21 (21)
Lambdoid	4 (10)	3 (3)

^aUnderrepresented minority: African American, Hispanic, and Native American.

^bMajority: Caucasians, Asians, other, and unknown.

Kaplan-Meier analysis of the cleft group estimates 25% of patients were lost to follow-up before 7.9 years of age, 50% were lost before 13.7 years of age, and 62% were lost prior to graduation (Figure 1A). For the synostosis group, 25% were estimated to be lost by 5.3 years of age and 45% prior to graduation (Figure 1B).

Figure 1.

The smoothed Kaplan-Meier survival functions showing retention of (A) cleft lip and palate and (B) synostosis patients. Age for active and graduated patients was capped at 16 years old for cleft and at 11 years old for synostosis.

Multivariable Cox proportional hazards analysis was performed to determine the effects of income, Medicaid status, sex, race, and distance to clinic on the likelihood of becoming lost or graduating from team clinic (Table 2). For the cleft group, race had a significant effect (P = .046), with underrepresented minorities being approximately 1.7 times more likely to become lost than patients in the majority group (hazard ratio: 1.66, 95% Cl: 1.01–2.74; Figure 2). Interestingly, no other factors significantly impacted whether a patient would be lost to follow-up or graduate (P ≥ .472). For the synostosis group, none of the variables considered significantly increased risk of loss to follow-up (P ≥ .127). Detailed results of the Cox analysis are in Table 2.

Table 2.

Multivariable Cox Proportional Hazards Analysis Showing the Effects of Income, Medicaid Status, Sex, Race, and Distance to Clinic on the Likelihood of Becoming Lost or Graduating From Team Clinic.^a

	Cleft Lip and Palate	Synostosis
	Hazard Ratio	Hazard Ratio
Demographics	(95% CI)	(95% CI)
Underrepresented Minority	1.664 (1.01–2.742)b	0.804 (0.247–2.618)
Sex	1.095 (0.856–1.400)	0.769 (0.504–1.173)
Distancec	1.029 (0.844–1.253)	1.107 (0.971–1.262)
Medicaid	1.056 (0.807–1.383)	1.102 (0.701–1.731)
Income	1.000 (1.000–1.000)	1.000 (1.000–1.000)
Overall χ2, significance	5.111–0.402	4.94–0.402

Abbreviation: CI, confidence interval.

^aReference categories for categorical variables: majority, male, and no Medicaid.

^bStatistically significant.

^cPer hundred miles

Figure 2.

Multivariable Cox proportional hazards analysis for the cleft group showing underrepresented minorities being approximately 1.7 times more likely to become lost to follow-up than patients in the majority group (hazard ratio: 1.66, 95% confidence interval [Cl]: 1.01–2.74).

Additional review of our patient data 15 to 18 months after initial inquiry revealed that 30 (9.9%) patients returned to clinic after being previously classified as lost in the cleft group and 6 (6%) patients originally categorized as lost to follow-up had returned to clinic in the synostosis group. Of these patients, the mean age at this new clinic visit was 11.2 years (±4.8) and 9.8 years (±2.3), respectively. On average, these patients were returning to clinic after 3.9 years (± 1.3) and 4.8 years (± 1.6). Patients presented back for surgical planning or concerns, including preparation for alveolar bone grafting in the cleft group or contour irregularities in the synostosis group. Other common concerns upon return were psychosocial (behavioral, emotional, and/or cognitive developmental concerns) or speech related (Table 3).

Table 3.

Reasons for Return to Clinic After Being Lost to Follow-Up.

Reason for Return	Number of Patients
Cleft
Surgical planning	21 (70%)
Psychologic concern	13 (43%)
Speech concern	8 (27%)
Routine visit	3 (10%)
Otolaryngology nonsurgical concern	2 (7%)
Other	2 (7%)
Synostosis
Psychologic concern	3 (50%)
Surgical concern	2 (33%)
Routine visit	2 (33%)

Survey

We sent 88 electronic surveys based on available e-mail addresses to families of patients with various cleft and craniofacial diagnoses that were deemed lost to follow-up from our clinic. Of those, 12 bounced back due to inactive e-mail accounts, and 4 parents declined to answer. Ultimately, we received 18 (20%) completed responses. A subset of 33 nonresponding families were mailed surveys, which yielded an additional 2 (6%) responses. Lastly, phone calls were made to the remaining 31 families who did not respond to the Mailed survey, which yielded 13 (42%) responses. Overall, 33 (38%) surveys were obtained.

Twenty (60%) surveys came from families who had a child with a cleft. The most common reasons for not returning to team clinic were seeing no reason to return (n = 7), cost being too high (n = 5), and not receiving a reminder letter or not thinking they were due to be seen (n = 5; Figure 3). Of those patients reporting costs were too high, their stated out-of-pocket costs ranged from $250 to over $1000.

Figure 3.

Pie chart for reasons families did not return to team care. Unlisted are items with zero responses: “weather issues” and “having no transportation.”

Thirteen (40%) surveys came from families who had a child with craniosynostosis. The most common reasons for not returning to team clinic were seeing no reason to return (n = 5) and cost being too high (n = 4; Figure 3). Of those who said cost was too high, their stated out-of-pocket cost ranged from $500 to over $1000.

Discussion

Multidisciplinary cleft and craniofacial team management has become the recommended method for providing complete and standardized care for patients affected with these conditions from infancy through adulthood (Eckstein et al., 2011; Warren et al., 2012; Birgfeld et al., 2015; Buchanan et al., 2017). Unfortunately, we have found that attrition rates are high within our institution. Our staff supplies information packets outlining the time line of care, reminder letters for appointments, and makes phone calls to patients to further educate families on the importance of continued care in the team setting. Wien patients are not able to be reached by phone, our staff will attempt to contact the primary care physician, emergency contact numbers, and any supplied e-mail addresses. Despite these efforts, many patients are still not able to be found due to changes in their mailing address and phone numbers.

Evaluation of our patients with complete cleft lip and palate indicated that over a 15-year time period, we lose 62% prior to graduation. For those patients with nonsyndromic single-suture synostosis, we lose 45% of patients prior to graduation. Although socioeconomic status, insurance status, and distance traveled to clinic did not appear to correlate with a patient’s likelihood for becoming lost to follow-up in the cleft group, underrepresented minorities were at greater risk to become lost to team care than Caucasian and Asian patients. We did not detect the same disparity in the synostosis group, which may be due to the very small number of underrepresented minorities with craniosynostosis included in the study (n = 11).

There is no single age interval at which our patients with cleft withdraw from our care. Rather, after a small spike in attrition after initial repair, the rate becomes and remains fairly steady all the way to graduation age. This suggests that patients are not all leaving following a particular milestone such as entering elementary school or obtaining an alveolar bone graft. Rather exogenous factors may be primarily responsible. This might include families moving outside of our clinical watershed, events which are less likely to be tied to patient age. We are unable to track family relocations aside from those indicated by requests to send records to outside institutions.

Fairly steady attrition was also seen in our synostosis patients with somewhat faster drop off at young ages beginning around 18 months. This, 18 months, is the age by which almost all synostosis patients have had their 1-year postoperative follow-up. The overall rate of attrition for patients with cleft and synostosis is similar through age 11, with the survival curves showing 60% retention at age 11 in patients with cleft and 55% at the same age for patients with synostosis. Given the very different care needs of the 2 groups, this similarity also points to exogenous factors playing a strong role.

Although the rates are similar, the consequences are not. A full quarter of patients with cleft are lost to our care by 8 years of age, during the time of mixed dentition when they should be undergoing preparation for alveolar bone grafting. With such a large proportion of patients lost prior to that age, these patients may not be obtaining this important surgical intervention. Failure to follow-up has been associated with abnormalities in adulthood that impact nasal/lip aesthetics, dentition, and speech (Truong et al., 2015). This age is also a crucial period for psychosocial development. Volpicelli et al. (2017) assessed age-related differences in psychosocial function of children with craniofacial anomalies and found that children between 8 and 10 years of age were at highest risk for distress and dysfunction. Of the 30 (9.9%) patients with cleft and 6 (6%) patients with synostosis previously classified as lost who returned to team clinic, the majority returned for surgical interventions/concerns, and psychosocial-related concerns were second most common. The long gap in care for these returning patients is still a concern. For that reason, we considered them lost to follow-up in the Kaplan-Meier and Cox models.

Our survey sought to identify reasons families forego returning to team care. The majority felt no medical reason to return and/or that costs were too high. This brings awareness to the importance of patient and family education and also the value of personalized care. Deuber et al. (2015) discussed a quality improvement initiative for postoperative care following cleft lip, palate, and alveolar bone grafting procedures. This effort involved regular teaching for the family, resident trainees, and inpatient care teams. They noted a significant reduction in the number of parent-initiated postoperative calls which they believe was potentially due to improvements in communication. Similarly, having better communication and education of our patients and families may help increase retention by empowering patients and families to be more invested in their own care time line. It is promising that in our contacting of families for the survey, it prompted a few of them to schedule appointments and return to care. Simply including additional follow-up by telephone to our protocol may extend the care of some patients. The educational aspect should also be considered. For example, parents need to be aware of the key time periods at which various providers involved in their care will become most relevant. Moreover, while providers may not have much control on the out-of-pocket costs families incur, we can strive to make team care more patient-specific and customized to best address each family’s unique needs. A more efficient patient care model that offers a higher level of specificity and customization might help control out-of-pocket expenses by providing patients only the care they need to optimize outcomes. In addition, social work can provide resources to help with financial or travel concerns to increase patient retention.

Understanding the burden of care that is placed on patients and their families is vital. The factors that must be considered are not only the financial burdens but also psychosocial stressors due to time off from school/work and undergoing multiple treatments. These factors must be weighed against the potential benefits of the given therapies, as increasing these burdens may result in impairing a family’s ability to adhere to the treatment regimen (Long, 2016).

Although overall attrition was high at our institution, our data suggest it is intensified among underrepresented minorities. Approaches focusing directly on changing impressions of the health-care system to these groups may reduce any disparity.

Although our synostosis graduates lived closer (on average) than those lost to follow-up, distance to clinic was not found to significantly impact risk in the proportional hazards model. This may be due to multicollinearity which shielded the impact of distance in the model. Given concerns to minimize patient burden of care as well as the increased distances lived by the patients lost in the synostosis group, there may be a role for use of telemedicine for those patients living far distances. Previous literature has shown the benefits of telemedicine in plastic surgery (Vyas et al., 2017).

Another strategy for increasing patient retention is to engage primary care physicians early in each patient’s care so that they may serve as a resource to remind families of the importance of continued team care follow-up. Further research evaluating these and other management strategies in team clinic may help families overcome the obstacles leading to early attrition.

Limitations

The retrospective nature of this study presented potential for confounding variables. The multivariable Cox regression models have somewhat low χ² values, indicating that the variables we assessed do not fully capture the factors leading to early attrition. Other factors that may also impact continuation of care such as primary provider or initial repair procedure were beyond the scope of this study. In addition, our proxy for socioeconomic status using census income data averages allows for misclassification.

Information was gathered from our institution’s database, which has potential for entry error. Although for our cleft group we looked at patients who failed to receive key surgical interventions such as lip and palate repair and alveolar bone graft surgery, we did not take into account those patients who did not receive appropriate rhinoplasty and orthognathic procedures. We were unable to identify patients who had become lost to our team clinic but had gone on to receive care with another institution. The survey we used was not validated and had a limited response rate, which may be biased for families that had either very positive or very negative experiences with our clinic. We had little minority representation in the graduated and lost groups limiting our generalizability and our power to detect differences between demographic subsets.

Conclusions

Team care for children with cleft and craniofacial anomalies is important in helping families overcome multiple obstacles to improve form, function, development, and integration into society. We identified a high rate of attrition in our cleft-craniofacial clinic. Reasons families fail to return to team clinic are multifaceted, and therefore, further in-depth research to examine burden of care will allow health-care teams to address issues that impact follow-up. Improved family education and personalized care are needed to help ensure continuity of care for these medically complex patients in order to decrease attrition rates.

Appendix A

Cleft and Craniofacial Team Care Survey

Why haven’t you returned to the Cleft (or Craniofacial) Team Clinic?

(Check all that apply.)

• □ Didn’t see a need

• □ Cost too high

• □ Trouble getting off work or school

• □ No transportation

• □ Personal/family issues

• □ Weather

• □ Our provider left

• □ We moved away

• □ Unhappy with care

• □ Didn’t like our provider(s)/the team

• □ Forgot to schedule an appointment or forgot to attend our appointment

• □ Never received a reminder card/didn’t think we were due

• □ Other:

  _______________________________________________

  _______________________________________________

  _______________________________________________

How much do you think your out-of-pocket cost was for your last team visit?

• □ $0–$100

• □ $100–$250

• □ $250–$500

• □ $500–$750

• □ $750–$1000

• □ >$1000

My child has

• □ Cleft lip or palate

• □ Craniosynostosis

• □ Another craniofacial deformity