Reply: Not All Clefts Are Created Equal

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We would to thank Guillaume, van Drie, and Breugem for their kind response to our article “Not All Clefts Are Created Equal: Patterns of Hospital-Based Care Use among Children with Cleft Lip and Palate within 4 Years of Initial Surgery.”1 In their insightful Letter to the Editor, they argue that our data underestimate the prevalence of feeding difficulties in the cleft population, quoting de Vries et al.,2 who reported that 67 percent of patients had feeding difficulties, and Spriestersbach et al.,3 who reported that 73 percent of isolated cleft palate patients had feeding difficulties. In addition, they correctly point out that feeding difficulties play a large role in the medical treatment of the cleft population both before and after surgery and is often a chief frustration of parents.2,4 Our group agrees with their emphasis on the importance of addressing feeding difficulties in children with cleft palate and recognize that our data may have underestimated the total care needed for these patients, as it used the Healthcare Cost and Utilization Project database.1
We conducted this population-based study of children with cleft lip, cleft palate, or both to determine and compare rates of hospital-based, acute care following their initial procedure. Secondarily, we sought to identify patient factors associated with high hospital-based acute care use. In addition, we wanted to test whether inherent patient factors may be associated with higher hospital-based care needs. Because of the database used and the focus on hospital-based care, we may have underestimated the rate of feeding difficulties, many of which may be managed on an outpatient basis.5–7
As Guillaume and colleagues mentioned, the reported prevalence of feeding difficulties varies across studies in part because of a lack of consensus regarding how this is defined for children with cleft lip and palate. In our study, failure to thrive and feeding difficulty was defined by standard administrative coding.5–9 Because we relied on International Classification of Diseases, Ninth Revision and Current Procedural Terminology coding in the current data set, and because the focus of the study was on hospital-based care, this likely identified the most serious cases that developed an acute issue or failed management in the outpatient setting.
Both our study and Guillaume’s commentary demonstrate that defining health care use and costs for children with cleft lip and palate can be challenging from a specificity/sensitivity standpoint. If all costs of care related to this population are considered, which has a moderately high rate of associated anomalies that are unrelated (e.g., heart, spine, craniofacial), the economic burden of care may be overestimated. If only those costs associated with hospitalization or surgery specifically related to the cleft are included, cost would be significantly underestimated. This patient population often requires home care, frequent visits to outpatient hospital departments, and even inpatient care close to home or at distant tertiary centers, the economic data for which may reside in disparate locations, making it difficult to paint a comprehensive picture of the problem. For that reason, we focused specifically on inpatient care, because it is this care that typically translates to the highest health care costs, and reliable databases are available.5–9 Doing this risks underestimating the actual costs because of a failure to capture care provided in other settings. Our findings, therefore, should be viewed as the hospital costs associated with care in this population and not the total costs of care during their first 4 years of life.
In conclusion, we thank Drs. Guillaume, van Drie, and Breugem for their response and agree that the overall disease burden, which translates to higher cost burden, is greater than our study suggests.
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